patient case study lymphadenitis

Case Study : Lymphadenitis

Male, J.W. 52 years old – Lymphadenitis

A patient was presented with severe inflammation of the one of the lymphatic nodes under his chin. It was very painful both to touch and swallowing, swollen to the size of an egg: 3.0 x 4.0 cm, accompanied with mild fever and malaise.

This patient is a therapist and used his professional InterX5002 Acute Presets directly on the swollen node and the back of the neck 3 times a day for 3 days, without the use of antibiotics. After an initial 3 days of treatments the inflammation went down and he felt much relief from pain and associated unpleasant symptoms.

After 3 days, the patient treated once every day for 2 more days, by which time, the symptoms of discomfort had gone and the swelling reduced to 1.0 x 1.0 cm. Over the following 2 weeks the swelling gradually decreased in size, and by week four all symptoms had gone.

Case report
Open access
Published: 16 December 2022

Tender cervical lymphadenitis as a herald of multi-system inflammatory syndrome in COVID-19 infection of children and adolescents: a report of two cases

Leonard Wanninayake 1 , 2 ,
Gayathree de Abrew 3 ,
Dinusha Logeshwaran 1 ,
Chathurika Weerasinghe 1 ,
Piyumi Gowinna 1 ,
Sachith Mettananda 3 , 4 &
Ranjan Premaratna ORCID: orcid.org/0000-0002-6588-9467 1 , 5

BMC Infectious Diseases volume 22 , Article number: 946 ( 2022 ) Cite this article

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Post-COVID-19 multisystem inflammatory syndrome (MIS) has been increasingly recognized but fever with isolated tender cervical lymphadenitis as the initial presentation has been rarely reported. We present 2 female patients one a child and the other an adolescent.

Case presentation

Case 1 was a 13-year-old girl who presented with tender cervical lymphadenopathy and fever 3-weeks post-COVID-19, and then developed features of MIS 5 days later. Case 2, also female, was 18 years old. She had no history of COVID-19 infection or immunization but had a serologic diagnosis of COVID-19. She similarly presented with fever and tender cervical lymphadenopathy, and then progressed rapidly to develop features of MIS. Both patients responded well to treatment with immunosuppressants and intravenous immunoglobulin.

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Introduction

Multi system inflammatory syndrome (MIS) following COVID 19 infection is still under study. It is mainly described in children (MIS-C) [ 1 ], however, recently a similar clinical entity has been reported in adults (MIS-A) [ 1 ]. MIS-C is described like Kawasaki disease or toxic shock syndrome. Its presentations vary and include persistent fever and combinations of symptoms related to gastrointestinal, cardiovascular, neurological, or haematological involvement. Severe most illness may lead to haemodynamic instability and development of shock. Elevated inflammatory markers and high antibody titres against COVID 19 infection helps in the diagnosis [ 2 , 3 ]. MIS also known to result in several dermatological manifestations and lymphadenopathy [ 1 , 3 ]. Recently reported case series of MIS-A highlight fever being the most common symptom and is documented in 96% of the patients [ 3 ]. Other reported clinical features are related to haematological involvement in 92%, hypotension in 60%, cardiac dysfunction in 54%, diarrhoea in 52% and the shortness of breath in 52% [ 1 , 3 , 4 , 5 ].

In pre COVID 19 era, acute fever with tender cervical lymphadenopathy has been a common clinical presentation where viral or bacterial aetiologies are considered and mostly managed as outpatients [ 6 , 7 ]. Although occurrence of lymphadenopathy together with symptoms such as conjunctival injection, skin rashes, vomiting, diarrhoea together with features of cardiovascular involvement and hypotension has been described in MIS [ 3 ] However, initial presentation with tender cervical lymphadenitis and fever has been rare [ 8 ]. We present 2 female adolescents who had fever with isolated tender cervical lymphadenitis as the initial presentation of MIS.

13-year-old healthy girl who had COVID-19 3 weeks before, had presented to the general practitioner with fever and painful right sided neck swelling for 3 days and received oral clarythromycin treatment based on high WBC (11.5 × 10 3 ) and c-reactive protein [CRP] (76 mg/dL). However, as there was no response, she presented to specialist care on the 5th day of illness with worsening symptoms. Examination revealed tender right cervical lymphadenitis with no tonsillar or pharyngeal inflammation. There were few 1–3 mm blisters on the trunk. After 2 days, she developed conjunctivitis, vomiting, diarrhoea and generalized erythematous macular rash and erythematous mucous membranes. Her pulse rate was 120/min and BP was 80/60 mmHg.

There was neutrophil leucocytosis with elevated CRP, ESR, serum ferritin, LDH, AST and ALT (Table 1 ), but serum procalcitonin and blood cultures were negative. Chest X-ray was normal, ECG showed sinus tachycardia and 2D transthoracic echo cardiogram showed a structurally normal heart with prominent and mildly dilated coronary arteries with no ectatic segments or aneurysms. The troponin I was normal. Anti-COVID 19 IgM and IgG were positive in very high titres. Her investigations on admission are summarized in Table 1 . Based on above features, a clinical diagnosis of severe MIS-C was made.

She was treated with intravenous fluid boluses, inotropic support [dobutamine], and the specific management was based on the updated clinical consensus on the management of MIS-C [ 6 ].

A 18-year-old otherwise healthy schoolgirl had a similar presentation to primary care with painful right sided neck lump for 4 days and high-grade intermittent fever with chills for 2 days. She had a mild cough with scanty non-purulent sputum. She did not have a history or contact with COVID 19 infection and did not receive vaccination against COVID 19. She had received treatment with oral antibiotics as for bacterial lymphadenitis. One day later she presented for specialist care with worsening symptoms and vomiting. She also had a pulse rate of 110/min and blood pressure BP 85/60 mmHg.

She had thrombocytopaenia with neutrophil predominance, elevated CRP, ferritin, LDH and D dimer. However, procalcitonin and blood cultures were negative. ECG showed sinus tachycardia and Trop I was normal. 2D Echo showed structurally normal heart with prominent and mildly dilated coronary arteries but no ectatic segments or aneurysms like first patient. She developed low arterial O 2 saturation (90%) on air on third day warranting supplementary O 2 via nasal prongs. Chest X-ray was normal and there were no inflammatory shadows or pleural effusion. CTPA which was negative for pulmonary embolism. HRCT showed evidence of extensive consolidation with ground-glass opacities in dependant areas of both lungs suggesting MIS associated with COVID 19 [ 7 ]. Her investigations are summarized in Table 1 .

A clinical diagnosis of MIS was made based on the multisystem involvement with strongly positive COVID 19 serology. Patient was given ICU care for 3 days and specific management was commenced like for the first patient and the heart rate was controlled with Ivabradine 2.5 mg twice daily. Inotropic support with given with noradrenaline 0.1 µg/kg/h during the first 2 days of ICU care. She had a gradual improvement over 72 h. On the 7th day of illness she developed a transient erythematous, non-scaly, blanching rash in upper limbs and in the torso and this was considered an Iv-Ig induced drug reaction [ 8 ]. After 11 days of institutional care, patient had a full recovery and was discharged on tapering doses of oral steroids. After 8 weeks of treatment, she had a remarkable recovery and was in good health.

Both above patients, who were previously well, presented with tender cervical lymphadenitis and rapidly progressed to involve other systems. The recent history of COVID-19 in the first patient together with few skin blisters, gastrointestinal and cardiovascular findings prompted the possibility of MIS. Although, the second patient had no history of COVID-19 or contact history, its likely her initial symptoms could have been overlooked at primary contact level and/or she had contacted the illness from an asymptomatic patient with COVID-19. However, it’s the experience of the first patient that helped in the diagnosis of the second patient, otherwise the illness of this would have been missed leading to a catastrophic ending.

In a recent publication, a 28-year-old man who has had fever and right sided submandibular lymphadenopathy was diagnosed having MIS-A by excluding all other possible aetiologies [ 5 ]. However, patient had no other systemic manifestations and has improved without immunosuppressive treatment [ 5 ]. Our second patient is 18 years, her illness would probably fall into MIS-A and she had an acute illness together with systemic involvement suggested by the tachycardia, hypotension, transaminitis and the lung involvement. She warranted immune-suppressive treatment; however, the treatment was based on MIS-C management guidelines [ 6 ] as there was no clear consensus in the management of MIS-A.

Fever with lymphadenitis is a common clinical presentation among children and adolescents. Common causes of such presentation include infectious mononucleosis, CMV infection, HIV, streptococcal pharyngitis, or tonsillitis, SLE and tuberculosis. However, the first three illnesses are viral aetiologies and results in normal inflammatory markers such as normal CRP together with reactive lymphocytosis in the blood picture. In bacterial infections, high inflammatory markers with raised neutrophil counts are expected. SLE (systemic lupus erythematosus) is known to cause a low WBC, high ESR and a low CRP level. Tuberculosis would result in a non-tender lymphadenopathy and other constitutional symptoms such as loss of appetite and weight loss. Although MIS associated with COVID-19 infection is mostly an illness of exclusion of above common aetiologies, presence of skin blisters or other dermatological manifestations, and systemic manifestations such as unexplained tachycardia or low blood pressure should prompt the suspicion of MIS [ 1 , 3 , 5 , 8 ] and investigated appropriately, as delay in treatment is likely to result in a deleterious outcome.

Tender cervical lymphadenopathy could be the herald of multi-system inflammatory syndrome following COVID-19 infection among children and adolescents, which the clinicians must have a good suspicion about. As this is an emerging clinical entity during the post COVID era, that may mimic common clinical scenarios we recommend careful assessment, follow up and documentation of similar illness to facilitate detection of MIS at an early stage.

Availability of data and materials

Data sharing is not applicable to this article as no datasets were generated or analysed during the current study. However, all clinical materials are available with the respective patients and their bed head tickets are available in the hospital record room maintaining confidentiality.

Abbreviations

Multisystem inflammatory syndrome in children

Multisystem inflammatory syndrome in adults

White blood cells

C reactive protein

Blood pressure

Neutrophils

Erythrocyte sedimentation rate

Aspartate transaminase

Alanine transaminase

Lactate dehydrogenase

Alkaline phosphatase

Polymerase chain reaction

Electrocardiogram

High resolution computed tomogram

Computed tomogram pulmonary angiogram

Acid fast bacilli

Tuberculosis

Intensive care unit

Intravenous

Intravenous immunoglobulin

Morris SB, Schwartz NG, Patel P, et al. Case series of multisystem inflammatory syndrome in adults associated with SARS-CoV-2 infection—United Kingdom and United States, March–August 2020. MMWR Morb Mortal Wkly Rep. 2020;69:1450–6.

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University Medical Unit, Colombo North Teaching Hospital, Ragama, Sri Lanka

Leonard Wanninayake, Dinusha Logeshwaran, Chathurika Weerasinghe, Piyumi Gowinna & Ranjan Premaratna

Department of Pharmacology, Faculty of Medicine, University of Colombo, Colombo, Sri Lanka

Leonard Wanninayake

University Paediatric Unit, Colombo North Teaching Hospital, Ragama, Sri Lanka

Gayathree de Abrew & Sachith Mettananda

Department of Paediatrics, Faculty of Medicine, University of Kelaniya, Ragama, Sri Lanka

Sachith Mettananda

Department of Medicine, Faculty of Medicine, University of Kelaniya, PO Box 6, Thalagolla Road, Ragama, Sri Lanka

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RP, DL and SM conceptualized the idea, RP, DL, LW, GA, CW, PG, SM participated in patient management at different stages, contributed to writing up and editing the manuscript. All authors read and approved the final manuscript.

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Correspondence to Ranjan Premaratna .

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Wanninayake, L., de Abrew, G., Logeshwaran, D. et al. Tender cervical lymphadenitis as a herald of multi-system inflammatory syndrome in COVID-19 infection of children and adolescents: a report of two cases. BMC Infect Dis 22 , 946 (2022). https://doi.org/10.1186/s12879-022-07943-w

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Received : 18 January 2022

Accepted : 12 December 2022

Published : 16 December 2022

DOI : https://doi.org/10.1186/s12879-022-07943-w

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Neck swelling and pain for several days.

Patient Data

Generalised subcutaneous oedema and swelling surrounding enlarged, but normal, lymph nodes.

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Lymphadenitis Clinical Presentation

Author: Elizabeth Partridge, MD, MPH, MS; Chief Editor: Russell W Steele, MD more...
Sections Lymphadenitis
Practice Essentials
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Physical Examination
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Questions & Answers

Patients with a clinical history of any of the following may be at risk for developing lymphadenitis:

Symptoms of an upper respiratory tract infection, sore throat, earache, coryza, conjunctivitis, or impetigo

Fever, irritability, or anorexia

Contact with animals, especially kittens or livestock

Recent dental care or poor dental health

Recent use of hydantoin and/or mesantoin

Enlarged lymph nodes can be asymptomatic, or they can cause local pain and tenderness. Overlying skin may be unaffected or erythematous.

Cervical lymphadenitis can lead to neck stiffness and torticollis .

Preauricular adenopathy is associated with several forms of conjunctivitis, including unilocular granulomatous conjunctivitis ( catscratch disease , chlamydial conjunctivitis, listeriosis, tularemia, or tuberculosis), pharyngeal conjunctival fever (adenovirus type 3 infection) and keratoconjunctivitis (adenovirus type 8 infection).

Retropharyngeal node inflammation can cause dysphagia or dyspnea.

Mediastinal lymphadenitis may cause cough, dyspnea, stridor, dysphagia, pleural effusion, or venous congestion.

Intra-abdominal (mesenteric and retroperitoneal) adenopathy can manifest as abdominal pain.

Iliac lymph node involvement may cause abdominal pain and limping.

Aspects of the physical examination are as follows:

Location - Depends on underlying etiology (see Causes section below)

Number - Single, local groupings (regional), or generalized (ie, multiple regions)

Size/shape - Normal lymph nodes range in size from a few millimeters to 2 cm in diameter; enlarged nodes are greater than 2-3 cm with regular/irregular shapes

Consistency - Soft, firm, rubbery, hard, fluctuant, warm

Tenderness - Suggestive of an infectious process but does not rule out malignant causes

Physical examination findings suggestive of malignancy are as follows:

Physical examination findings suggestive of infection are as follows:

Overlying erythema or streaking

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Carvalho AC, Codecasa L, Pinsi G, Ferrarese M, Fornabaio C, Bergamaschi V. Differential diagnosis of cervical mycobacterial lymphadenitis in children. Pediatr Infect Dis J . 2010 Jul. 29(7):629-33. [QxMD MEDLINE Link] .

Loizos A, Soteriades ES, Pieridou D, Koliou MG. Lymphadenitis by non-tuberculous mycobacteria in children. Pediatr Int . 2018 Dec. 60 (12):1062-1067. [QxMD MEDLINE Link] .

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Willemse SH, Oomens MAEM, De Lange J, Karssemakers LHE. Diagnosing nontuberculous mycobacterial cervicofacial lymphadenitis in children: A systematic review. Int J Pediatr Otorhinolaryngol . 2018 Sep. 112:48-54. [QxMD MEDLINE Link] .

Wang L, Wu W, Teng J, Zhong R, Han B, Sun J. Sonographic Features of Endobronchial Ultrasound in Differentiation of Benign Lymph Nodes. Ultrasound Med Biol . 2016 Dec. 42 (12):2785-93. [QxMD MEDLINE Link] .

Pecora F, Abate L, Scavone S, et al. Management of Infectious Lymphadenitis in Children. Children (Basel) . 2021 Sep 27. 8 (10): [QxMD MEDLINE Link] . [Full Text] .

Geake J, Hammerschlag G, Nguyen P, Wallbridge P, Jenkin GA, Korman TM, et al. Utility of EBUS-TBNA for diagnosis of mediastinal tuberculous lymphadenitis: a multicentre Australian experience. J Thorac Dis . 2015 Mar. 7(3):439-48. [QxMD MEDLINE Link] . [Full Text] .

Dulin MF, Kennard TP, Leach L, Williams R. Management of cervical lymphadenitis in children. Am Fam Physician . 2008 Nov 1. 78(9):1097-8. [QxMD MEDLINE Link] .

Haimi-Cohen Y, Markus-Eidlitz T, Amir J, Zeharia A. Long-term Follow-up of Observation-Only Management of Nontuberculous Mycobacterial Lymphadenitis. Clin Pediatr (Phila) . 2016 Oct. 55 (12):1160-4. [QxMD MEDLINE Link] .

Jensen FN, Nielsen AB, Dungu KHS, et al. Distinct clinical parameters were associated with shorter spontaneous resolution in children with non-tuberculous mycobacterial lymphadenitis. Acta Paediatr . 2024 Jan 16. [QxMD MEDLINE Link] .

Guss J, Kazahaya K. Antibiotic-resistant Staphylococcus aureus in community-acquired pediatric neck abscesses. Int J Pediatr Otorhinolaryngol . 2007 Jun. 71(6):943-8. [QxMD MEDLINE Link] .

Howard-Jones AR, Al Abdali K, Britton PN. Acute bacterial lymphadenitis in children: a retrospective, cross-sectional study. Eur J Pediatr . 2023 May. 182 (5):2325-33. [QxMD MEDLINE Link] . [Full Text] .

A lymph node biopsy is performed. Note that a marking pen has been used to outline the node before removal and that a silk suture has been used to provide traction to assist the removal.

Contributor Information and Disclosures

Elizabeth Partridge, MD, MPH, MS Assistant Clinical Professor of Pediatrics, Division of Pediatric Infectious Diseases, Allergy and Immunology, Medical Director of Hospital Epidemiology and Infection Prevention, UC Davis Children’s Hospital, UC Davis Medical Center Elizabeth Partridge, MD, MPH, MS is a member of the following medical societies: American Academy of Pediatrics , American Medical Association , California Medical Association , Infectious Disease Association of California, Infectious Diseases Society of America , Pediatric Infectious Diseases Society , Society for Healthcare Epidemiology of America Disclosure: Nothing to disclose.

Dean A Blumberg, MD Associate Professor of Pediatrics and Chief, Section of Pediatric Infectious Disease, University of California Davis Children's Hospital Dean A Blumberg, MD is a member of the following medical societies: American Academy of Pediatrics , American Society for Microbiology , American Society of Tropical Medicine and Hygiene , California Medical Association , Infectious Diseases Society of America , Pediatric Infectious Diseases Society , Sierra Sacramento Valley Medical Society Disclosure: Received grant/research funds from Novartis for clinical research investigator; Received speaking fees paid to university, not self from Merck for speaking and teaching; Received speaking fees paid to university, not self from sanofi pasteur for speaking and teaching.

Mary L Windle, PharmD Adjunct Associate Professor, University of Nebraska Medical Center College of Pharmacy; Editor-in-Chief, Medscape Drug Reference Disclosure: Nothing to disclose.

Larry I Lutwick, MD, FACP Editor-in-Chief, ID Cases; Moderator, Program for Monitoring Emerging Diseases; Adjunct Professor of Medicine, State University of New York Downstate College of Medicine Larry I Lutwick, MD, FACP is a member of the following medical societies: American Association for the Advancement of Science , American Association for the Study of Liver Diseases , American College of Physicians , American Federation for Clinical Research , American Society for Microbiology , Infectious Diseases Society of America , Infectious Diseases Society of New York, International Society for Infectious Diseases , New York Academy of Sciences , Veterans Affairs Society of Practitioners in Infectious Diseases Disclosure: Nothing to disclose.

Russell W Steele, MD Clinical Professor, Tulane University School of Medicine; Staff Physician, Ochsner Clinic Foundation Russell W Steele, MD is a member of the following medical societies: American Academy of Pediatrics , American Association of Immunologists , American Pediatric Society , American Society for Microbiology , Infectious Diseases Society of America , Louisiana State Medical Society , Pediatric Infectious Diseases Society , Society for Pediatric Research , Southern Medical Association Disclosure: Nothing to disclose.

Gary J Noel, MD Professor, Department of Pediatrics, Weill Cornell Medical College; Attending Pediatrician, New York-Presbyterian Hospital Gary J Noel, MD is a member of the following medical societies: Pediatric Infectious Diseases Society Disclosure: Nothing to disclose.

The authors and editors of Medscape Reference gratefully acknowledge the contributions of previous author, Ulfat Shaikh, MD, MPH, to the development and writing of this article.

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Lymphadenitis
Mesenteric Lymphadenitis
Mesenteric Lymphadenitis (Adenitis) Imaging
Mycobacterium Avium Complex (MAC) (Mycobacterium Avium-Intracellulare [MAI])
Toxoplasmosis Pathology
Mycobacterium haemophilum Infection
Cutaneous Kikuchi Disease

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http://orcid.org/0000-0001-7837-9733 Elpis Mantadakis 1 ,
Chrysostomos Soultanidis 2 ,
Athanasia Christidou 3 and
Sofia Maraki 3
1 Department of Pediatrics , Democritus University of Thrace , Alexandroupolis , Greece
2 Department of Pediatric Surgery , University General Hospital of Alexandroupolis , Alexandroupolis , Greece
3 Department of Clinical Microbiology and Microbial Pathogenesis , University Hospital of Heraklion, Crete, Greece , Heraklion , Greece
Correspondence to Professor Elpis Mantadakis; emantada{at}med.duth.gr

An 8-year-old girl with subacute submandibular lymphadenitis and no other complaints is described. After failure of parenteral antistaphylococcal therapy, she underwent incision and drainage of the involved lymph node. The responsible pathogen was identified as Mycobacterium malmoense by GenoType CM assay and sequencing of the 16S ribosomalRNA (rRNA) gene. The patient remains healthy, 11 months after surgery, even though it took approximately 4 months for the surgical incision to heal completely. While M. malmoense is a relatively common cause of non-tuberculous mycobacteria (NTM) lymphadenitis in Northern Europe, this is the first reported case from Greece. We conclude that in a young child with lymphadenitis without systemic symptoms, the microbiology laboratory should be notified in advance in order to extend the duration of mycobacterial cultures. Application of molecular methods will increase the number of reported cases of rare NTM in the future.

otolaryngology / ENT

https://doi.org/10.1136/bcr-2020-234657

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Contributors EM had the idea of writing the case report, performed the literature search, wrote the manuscript and provided medical care for the patient. CS operated on the patient and helped with literature search. AC and SM performed the microbiological investigations and helped revise the manuscript prior to its submission.

Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

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Kikuchi Fujimoto lymphadenitis: case report and literature review

Affiliation.

1 Department of Internal Medicine, San Camillo Hospital, Rome, Italy. [email protected]
PMID: 12949892
DOI: 10.1002/ajh.10335

We describe a young woman with two severe episodes of Kikuchi Fujimoto disease occurring 16 years apart. Both episodes were proven by biopsy, and on the second occasion the patient remained dependent on high-dose prednisone for more than 6 months in order to control inflammation and achieve a reduction in cervical lymph node size. The second lymph node biopsy showed leukocytoclastic vasculitis in addition to the typical features of Kikuchi Fujimoto disease, but, even though the clinical interpretation of this finding was unclear, we documented no clinical or laboratory evidence of the development of other serious systemic disease over 20 years of follow-up. Kikuchi Fujimoto disease is considered a disorder with a self-limited course and a favorable outcome. However, on the basis of our experience with this patient and data from peer-reviewed literature, we suggest that this generally accepted postulate should be revised.

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Case Report
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Published: 27 October 2021

Adult-onset Still’s disease with multiple lymphadenopathy: a case report and literature review

Zhonghua Huang ORCID: orcid.org/0000-0001-9491-3377 1 ,
Qinqin Min 1 ,
Zhenguo Li 1 ,
Jiaxin Bi 1 ,
Lingyun Liu 1 &
Yingying Liang 1

Diagnostic Pathology volume 16 , Article number: 97 ( 2021 ) Cite this article

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Adult-onset Still’s disease (AOSD) often presents with systemic multiple lymphadenopathy. In addition to the common paracortical and mixed patterns in AOSD lymph node histopathological features, other morphological patterns include diffuse, necrotic, and follicular patterns. However, to date, there have been few reports on the histopathological description of AOSD lymph nodes.

Case presentation

An 18-year-old woman presented 2 months earlier with pain in her large joints with painless rash formation; bilateral posterior cervical lymph node, left supraclavicular lymph node, and left posterior axillary lymph node enlargement, and no tenderness. Left cervical lymph node resection was performed for pathological examination. The lymph node structure was basically preserved, and subcapsular and medullary sinus structures were observed. Many histiocytes in the sinus were observed, the cortical area was reduced, a few lymphoid follicles of different sizes were observed, and some atrophy and hyperplasia were noted. The lymphoid tissue in the paracortical region of the lymph node was diffusely proliferative and enlarged, mainly comprising histiocytes with abundant cytoplasm, immunoblasts and numerous lymphocytes with slightly irregular, small- to medium-sized nuclei. Nuclear karyorrhexis was easily observed, showing a few nuclear debris and the “starry sky” phenomenon, accompanied by abundantly branching high endothelial small vessels with few scattered plasma cells and eosinophil infiltration. Lymphoid follicle immunophenotype with reactive proliferative changes was observed. Approximately 40% of the cells in the paracortical region were positive for Ki-67, and the histiocytes expressed CD68, CD163, and some expressed S-100, with the absence of myeloperoxidase. The immunoblasts expressed CD30 and CD20, not ALK or CD15. Background small- to medium-sized T cells expressed CD2, CD3, CD5, CD7, CD4, and CD8; the number of CD8-positive T cells was slightly predominant, and a small number of T cells expressed granzyme B and T-cell intracellular antigen 1. The patient received a comprehensive medical treatment after the operation, and her condition was stable without progression at the 11-month follow-up evaluation.

Conclusions

The pathological features of AOSD lymphadenopathy raises the awareness of AOSD among pathologists and clinicians and aids in the diagnosis and differential diagnosis of AOSD lymphadenopathy from other reactive lymphadenopathies (lupus lymphadenitis, etc.) and lymphomas.

Adult-onset Still’s disease (AOSD) is a rare group of systemic autoinflammatory diseases with complex, incompletely defined etiology and pathogenesis, mainly characterized by intermittent hyperthermia, transient skin rash, elevated blood leukocyte levels (neutrophils > 80%), polyarthritic pain, and multiple lymphadenopathy, and a predilection for young adults [ 1 ]. In 1971, Bywaters first described 14 cases of Still’s disease observed in patients aged 17–35 years with clinical features significantly similar to those of childhood Still’s disease, mainly characterized by high fever, multiple skin rashes, and polyarthritis, thus defining AOSD [ 2 ]. AOSD is often accompanied by liver and spleen enlargement and lymphadenopathy. Its clinical manifestations are complex and unspecific, sometimes similar and overlapping with lymphoma in clinical manifestations and histopathology [ 3 ], which may easily lead to misdiagnosis or missed diagnosis. In this case report, we present a case of AOSD and review the relevant literature to explore the clinical and pathomorphological features of enlarged lymph nodes and immunophenotypes, with the aim of improving the level of pathological diagnosis of the disease to prevent the misdiagnosis of lymphoma or other associated lymphadenopathies (lupus lymphadenitis, etc.).

Our patient was an 18-year-old woman. Two months earlier, she presented with pain in the large joints of all extremities, with no evident trigger for its development, and pruritic and painless rash formation on the skin of the dorsum of the shoulders (Fig. 1 ) and both wrists and both sides of the thighs, which worsened with increasing symptoms. Febrile and night sweats were observed on the sixth day upon admission. The patient was admitted to the hospital for blood analysis, and the blood test results were as follows: ferritin, 466.4 ng/ml; C-reactive protein (CRP) level, 82.7 mg/l; interleukin-6 level, 85.12 pg/ml; white blood cell count, 15.55 × 10 9 /L; and neutrophils, 89.3%. The patient’s antinuclear antibody was positive (titer 1:100), but her rheumatoid factor,anti double stranded DNA antibody, Anti smooth muscle antibody,anti-extractable nuclear antigen antibodies and anti-neutrophil cytoplasm antibodies were negative. Since symptom onset, the patient lost 5 kg of body weight. Examination revealed the presence of bilateral posterior cervical lymph nodes, left supraclavicular lymph nodes, and left posterior axillary lymph nodes without tenderness. According to the new classification of systemic lupus erythematosus (SLE) published by the European alliance against Rheumatism (EULAR) and the American Society of Rheumatology (ACR) in 2019 [ 4 ], clinicians have excluded the diagnosis of SLE. Following the clinical suspicion of lymphoma, left neck lymph node resection was performed for pathological examination.

Pruritus and painless rash developed on the back and shoulder skin of the patient

Pathological findings

One lymph node was resected, measuring approximately 2.5 × 2 × 1.2 cm, with its cut surface gray white and gray red in color and medium in texture.

The lymph node structure was partially preserved (Fig. 2 a),the subcapsular and medullary sinuses were slightly dilated, and the sinus comprised a larger amount of histiocytes (Fig. 2 b). The cortical areas were atrophic and smaller (Fig. 2 c), and a few lymphoid follicles of various sizes were observed (Fig. 2 d). Some of the follicular germinal centers were atrophic and smaller (Fig. 2 e), some follicles germinal centers were hyperplastic and enlarged with the phenomenon of “starry sky”.The paracortical areas of the lymph nodes were diffusely hyperplastic and enlarged,mainly comprised abundant histiocytes, immunoblasts and medium-sized T lymphocytes with slightly irregular nuclei,against a background scattered with a higher amount of plasma cells and a few infiltrating eosinophils (Fig. 2 f). In some areas, histiocytic hyperplasia was patchy,and a few proliferating histiocytes had distorted and elongated nuclei with irregular morphology (Fig. 3 a). In some areas, immunoblasts proliferated in a “mottled” manner, with a small amount of apoptotic nuclear debris and phagocytosis of nuclear debris by histiocytes. Medium-sized T lymphocytes were actively proliferative,karyorrhexis was easily observed, and the proliferation of high endothelial venules in the paracortical area showed a complex branching pattern (Fig. 3 b).

Microscopic features. a . Lymph node structure is partially preserved, and residual lymph follicles can be seen (HE, × 40); b . The subcapsular and medullary sinuses are slightly expanded, and the sinus comprises a larger amount of histiocytes (HE, × 100); c . The cortical areas are atrophic and smaller (HE, × 40); d . A few lymphoid follicles of various sizes are observed (HE, × 40); e . Some of the follicular germinal centers are atrophic and smaller (HE, × 100);f. Lymph node paracortical areas are diffusely proliferative and enlarged,and consist mainly of histiocytes, immunoblasts, and medium-sized T lymphocytes (HE, × 400)

Microscopic features. a . A small number of histiocytes (black arrows) have distorted and elongated nuclei with irregular shapes (HE, × 400); b . High endothelial venule proliferation in the paracortical area (HE, × 40); c . The proliferating histiocytic fraction of the paracortical area expresses S-100(Envison, × 100); d . Proliferating CD8-positive T cells in the paracortical zone (Envison, × 100)

Immunohistochemical findings

Proliferating histiocytes expressed CD68 and CD163, and some expressed S-100 (Fig. 3 c) and myeloperoxidase. The proliferating immunoblasts were strongly and weakly heterogeneously positive for CD30 and CD20,and negative for CD15.The proliferating medium-sized T lymphocytes in the paracortical areas expressed CD3, CD5, CD4, and CD8, with a slight predominance of CD8-positive cells (Fig. 3 d), and some T cells expressed granzyme B and T-cell intracellular antigen 1. The proliferation index of Ki-67 in the hot spots of paracortical areas was about 40%, and plasma cells expressed CD138 but not immunoglobulin 4(IgG4). Lymphoid follicles expressed CD20, CD10, and BCL-6,and did not express BCL-2,the proliferation index of Ki-67 was approximately 80%. CD123, CD56, TdT, ALK, and CK antibodies were not detected.

The final diagnosis was adult-onset Still’s disease (AOSD), combined with clinical manifestations, laboratory examination and lymph node pathological biopsy. The patient received a comprehensive medical treatment after the operation, including symptomatic treatment, oral prednisone acetate tablets and methotrexate. Her condition was stable without progression at the 11-month follow-up evaluation.

Discussion and conclusions

Clinically, AOSD is relatively rare, with an incidence between approximately 1 and 34 per 1 million population, with an equal incidence in both sexes, and a “bimodal” age of onset of 15–25 years and 36–46 years [ 5 ]. AOSD often has four major clinical features: transient rash in the proximal limbs or trunk at the peak of fever, high fever of 39 °C or more, elevated peripheral white blood cell count and neutrophil proportion greater than 80%, and generalized polyarticular pain or arthritis. Other clinical manifestations include pharyngeal pain, myalgia, myositis, lymphadenopathy, splenomegaly, pericarditis, myocarditis, pleuritis, lung disease, hepatitis, increased erythrocyte sedimentation rate and CRP levels, increased ferritin level, decreased glycosylated ferritin level, and coagulopathy [ 6 ]. AOSD can be easily misdiagnosed clinically as infectious lesions or other autoimmune diseases,such as systemic lupus erythematosus,because patients with systemic lupus erythematosus often have joint pain and rash. Clinicians are not aware of the possibility of AOSD when conservative treatment fails. After futile antibiotic treatment such as in the case presented here, AOSD was finally diagnosed by pathological examination of biopsied lymph nodes. Studies have shown that the general time from the appearance of symptoms or signs to the final diagnosis of AOSD ranges from 1.5 to 4 years [ 7 ].

AOSD usually presents with high fever, arthralgia, and rash and is often accompanied by multiple lymphadenopathy and hepatosplenomegaly [ 8 ]. When malignant lymphoma is easily suspected clinically after conservative medical treatment is ineffective, pathological biopsy of lymph nodes is the inevitable choice, combined with pathomorphological features, immunohistochemistry, and molecular biological examination to confirm the diagnosis. Jeon et al. [ 9 ] have summarized the pathohistomorphological changes in 12 AOSD enlarged lymph nodes and classified the lymphadenopathy into four morphological types. The first atypical paracortical hyperplasia pattern, characterized by hyperplasia in the paracortical areas of the lymph nodes with abundant high endothelial vessels, comprised mainly of reactive proliferating T lymphocytes, scattered large activated B/T immunoblasts, and few plasma cells and eosinophils, with a ratio of CD4- to CD8-positive T lymphocytes of approximately 3:2. Moreover, mildly hyperplastic histiocytes and focally hyperplastic monocytoid B cells were observed. Furthermore, the second burnt out histiocytic pattern was characterized by hyperplasia of the paracortical areas, high endothelial vascularity and sinus histiocytic proliferation with no remnants of lymphoid follicles. Histiocytes expressed CD68 and S-100 and were often clustered in a mottled pattern in the paracortical areas. Additionally, the third exuberant immunoblastic reaction pattern was characterized by patchy or diffuse proliferation of numerous immunoblasts in the paracortical area, predominant immunoblasts with numerous mitotic figures and a Ki-67 proliferation index of up to 90%, which is most easily confused with malignant lymphoma. The fourth follicular hyperplasia pattern was characterized by numerous lymphoid follicles of various sizes distributed throughout the lymph nodes, some with enlarged germinal centers, some with atrophy of germinal centers, and vascular hyalinization with widening of the mantle or marginal zones. The histomorphological features of AOSD lymphadenopathy are complex and diverse and change dynamically with the course of the disease.

Kim et al. [ 10 ] have performed histological observation of lymphadenopathy in 48 patients with AOSD and summarized six morphological patterns of lymph nodes. These include follicular pattern, dominated by extensive hyperplasia of lymphoid follicles; paracortical areas pattern, with proliferation and expansion of the paracortical areas and only a few small remnants of lymphoid follicles; diffuse pattern, characterized by diffuse hyperplasia of the paracortical areas, with no lymphoid follicular structures observed; necrotic pattern, characterized by proliferative expansion of the paracortical areas, focal scattered necrosis and nuclear fragmentation; mixed patterns of lymphoid follicles and paracortical areas; and a mixed pattern of diffuse and paracortical areas. It is also mentioned in the text that in almost all morphological patterns, moderate to severe hyperplasia of histiocytes is observed, and there are more CD8-positive T cells than CD4-positive T cells. The morphological features of lymph nodes in our case should belong to the mixed pattern of lymphoid follicles and paracortical areas, which were expanded with proliferative and atrophic lymphoid follicular structures, and a slight predominance of CD8-positive T lymphocytes.

Patients with AOSD often present with multiple enlarged lymph nodes, and when the diagnosis is still difficult to establish by a combination of clinical manifestations, laboratory tests, and imaging studies, surgical resection with pathological biopsy of lymph nodes is the inevitable choice for a definitive diagnosis. Previous studies have shown that most lymph node lesions histologically exhibit reactive hyperplasia in the paracortical region, characterized by the proliferation of immunoblasts and high endothelial venules [ 10 , 11 ]. The histomorphological features of AOSD lymphadenopathy are complex and diverse and change dynamically with the course of the disease, which still needs to be differentiated from the following diseases. (1) Angioimmunoblastic T-cell lymphoma (AITL) is a T-cell lymphoma formed by the proliferation of mature follicular helper T cells with prominent hyperplasia of high endothelial venules and follicular dendritic cells, often showing generalized lymphadenopathy, hepatosplenomegaly, systemic symptoms, polyclonal hypergammaglobulinemia, and a rash with pruritus. There are many similarities between AITL and AOSD in clinical presentation and histomorphological features, with the former neoplastic cells often expressing CXCL13, PD1, CD10, BCL-6, and ICOS, most of which often show Epstein-Barr virus (EBV)-positive B cells, and an irregular proliferation of CD21-positive follicular dendritic cells surrounding high endothelial venules. (2) Dermatopathic lymphadenopathy is a special type of proliferative lesion of the paracortical region of lymph nodes that usually presents as lymphadenopathy in the drainage area with chronic skin irritation and often shows a pale nodular appearance in the paracortical region, which is mainly composed of proliferating interdigitated dendritic cells, Langerhans cells, and pigment-laden histiocytes. Some studies have shown that in dermatopathic lymphadenopathy lesions [ 12 ], the paracortical areas of lymph nodes contain at least three subsets of dendritic cells with different immunophenotypes: interdigitated dendritic cells (S-100 positive, CD1a sparsely positive, langerin negative), Langerhans cells (S-100 positive, CD1a positive, langerin positive), and few dendritic cells (S-100 positive, CD1a negative, langerin negative). (3) Infectious mononucleosis is an EBV infection-induced proliferative lesion of the lymph nodes and tonsils that is commonly observed in adolescents and young adults and has a short disease course. Its histologic features vary with disease duration. Lymphofollicular hyperplasia predominates early in the disease, with monocytoid B-cell and histiocytic hyperplasia. The later stages of the disease show proliferative expansion in the paracortical areas, comprising proliferating immunoblasts, small- to medium-sized lymphocytes, and plasma cells with a mottled appearance, dominated by CD8-positive T cells. Moreover, the immunoblasts often show Epstein-Barr virus-encoded small RNA positivity. (4) Histiocytic necrotizing lymphadenitis (Kikuchi’s disease), also known as Kikuchi-Fujimoto lymphadenitis, usually has a self-limited predilection for young adults, especially young Asian women. It is classified into three subtypes: proliferative, necrotic, and xanthomatous. The early stage is dominated by the proliferation of immunoblasts, crescentic histiocytes, and plasmacytoid dendritic cells in the paracortical region. The necrotic phase shows patchy necrosis without neutrophil infiltration in the paracortical area, with a large number of nuclear debris. The xanthoma stage contains a large number of foamy histiocytes and few immunoblasts. When AOSD lymphadenopathy appears in a necrotic pattern, it needs to be differentiated from Kikuchi’s disease; moreover, the simultaneous appearance of both lesions has also been documented [ 13 ]. (5) Systemic lupus erythematosus (SLE) is an autoimmune connective tissue disease. Up to 60% of patients have generalized or local lymphadenopathy, and the most common is involving neck and mesenteric lymph nodes. When lymph nodes are involved, it is called systemic lupus erythematosus (SLE) - associated lymphadenitis (also known as lupus lymphadenitis). Histologically, hematoxylin body and different degrees of coagulation necrosis is the characteristic morphological change of lupus lymphadenitis. Hematoxylin bodies are often seen in necrotic areas and sinuses;The necrotic area is usually large, mainly composed of lymphoid cells, abundant nuclear fragments and residual shadows of histiocytes. Sometimes there are a large number of plasma cell infiltration in germinal center and medullary cord, and neutrophil infiltration will also be encountered. Kikuchi’s disease and lupus lymphadenitis (LL) often show the same immunophenotype and have overlapping histological characteristics. Histologically, it is almost impossible to distinguish between Kikuchi’s disease and lupus lymphadenitis. Therefore, it is necessary to further integrate clinicopathological information and apply C4d immunohistochemical staining to distinguish lupus lymphadenitis from Kikuchi’s disease. Lupus lymphadenitis will show C4d deposition [ 14 ].

Depending on the course of the disease, patients with AOSD have been clinically classified into three different clinical patterns (monocyclic, multicyclic, and slowly progressive) [ 15 , 16 ]. The chronic progression pattern is most commonly characterized by the occurrence of at least one persistent symptom lasting more than 1 year; it is mainly characterized by stable disease progression, persistent inflammation, and often erosion of the affected joint, followed by a multicyclic pattern, manifesting as periodic recurrences with unpredictable deterioration months or years later. A monocyclic pattern, manifesting as a single episode over 2 months but less than 1 year, persists in remission with no recurrence throughout follow-up. A new approach has divided patients with AOSD into two phenotypes: those with systemic features and those with chronic arthritis as the predominant feature [ 17 ].

AOSD often presents as a chronic passage, and patients may develop different complications within the course of the disease, which affects their clinical condition, treatment, and prognosis. Secondary hemophagocytic lymphohistiocytosis, also known as macrophage activation syndrome, is the most severe complication associated with high mortality. Its common complications include coagulopathy with multiorgan involvement, including the heart, lung, liver, spleen, and other sites [ 17 , 18 ], and these patients often require more intensive treatment and have a worse prognosis. It has been shown that more than 20% of patients with AOSD experience recurrence and that patients with severe disease at the initial stage of the disease may be at an increased risk of recurrence, which requires intensive treatment and close follow-up [ 19 ].

Availability of data and materials

The datasets used and/or analyzed during the current study are available from the corresponding author upon reasonable request.

Abbreviations

Adult-onset Still’s disease

C-reactive protein

Angioimmunoblastic T-cell lymphoma

Dermatopathic lymphadenopathy

Infectious mononucleosis

Hemophagocytic lymphohistiocytosis

Macrophage activation syndrome

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Acknowledgements

We would like to thank director Mumin Shao from Pathology Department of Shenzhen Hospital of traditional Chinese medicine for his diagnostic assistance.

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ZHH conceptualized and wrote the manuscript. HX collected clinical data. QQM performed pathological diagnosis and immunohistochemical analyses. All authors have read and approved the final manuscript prior to submission.

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Huang, Z., Xu, H., Min, Q. et al. Adult-onset Still’s disease with multiple lymphadenopathy: a case report and literature review. Diagn Pathol 16 , 97 (2021). https://doi.org/10.1186/s13000-021-01159-3

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DOI : https://doi.org/10.1186/s13000-021-01159-3

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Pre-operative calcitonin and cea values may predict the extent of metastases to the lateral neck lymph nodes in patients with medullary thyroid cancer.

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1. introduction, 2. materials and methods, 2.1. patients, 2.2. treatment and follow-up, 2.3. serum markers and pathological examination, 2.4. statistical analysis, 3.1. clinical and histopathological characteristics in patients with mtc, 3.2. pre-operative ctn and cea levels, 3.3. cut-off values of pre-operative ctn and cea levels predictive of the n1b status, 4. discussion, 5. conclusions, author contributions, institutional review board statement, informed consent statement, data availability statement, acknowledgments, conflicts of interest.

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Click here to enlarge figure

Parameter	N = 87
Age, years (mean ± SD)	48.3 ± 18.6
Gender, female n. (%)	59 (67.8)
Sporadic MTC, n. (%)	52 (59.8)
Tumor size (mm), median (25–75 IQR)	10 (6–17)
Total thyroidectomy with central LN dissection, n. (%)	60 (69)
Total thyroidectomy with both central and lateral LN dissection, n. (%)	27 (31)
Bilateral tumor, n. (%)	17 (19.5)
Multifocal tumor, n. (%)	21 (24.1)
N0, n. (%)	70 (80.5)
N1a, n. (%)	11 (12.6)
N1b, n. (%)	6 (6.9)
LN = lymph node

Tumor Size (mm)	Ctn Value (Median and IQR Range, pg/mL)	CEA Value (Median and IQR Range, ng/mL)
≤10	60 (24–94)	5.5 (1.3–7)
11–20	234 (125–472)	17.9 (13.9–50.7)
21–39	630 (503–722)	43.7 (28–67.1)
≥40	739 (447–1189)	52.0 (37.6–59.9)

0	88.4 (27–282)	7.0 (1.9–28)
1–5	108.0 (57.4–269)	9.6 (1.8–35.2)
>5	520.5(272–657.5)	43.2 (19.2–67.1)

N0	88.4 (27–282)	7 (1.9–28)
N1a	98.1 (57.4–145)	2.6 (1–16.5)
N1b	520.5 (118–722)	53.8 (19.2–67.1)

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Share and Cite

Prinzi, A.; Frasca, F.; Russo, M.; Pellegriti, G.; Piticchio, T.; Tumino, D.; Belfiore, A.; Malandrino, P. Pre-Operative Calcitonin and CEA Values May Predict the Extent of Metastases to the Lateral Neck Lymph Nodes in Patients with Medullary Thyroid Cancer. Cancers 2024 , 16 , 2979. https://doi.org/10.3390/cancers16172979

Prinzi A, Frasca F, Russo M, Pellegriti G, Piticchio T, Tumino D, Belfiore A, Malandrino P. Pre-Operative Calcitonin and CEA Values May Predict the Extent of Metastases to the Lateral Neck Lymph Nodes in Patients with Medullary Thyroid Cancer. Cancers . 2024; 16(17):2979. https://doi.org/10.3390/cancers16172979

Prinzi, Antonio, Francesco Frasca, Marco Russo, Gabriella Pellegriti, Tommaso Piticchio, Dario Tumino, Antonino Belfiore, and Pasqualino Malandrino. 2024. "Pre-Operative Calcitonin and CEA Values May Predict the Extent of Metastases to the Lateral Neck Lymph Nodes in Patients with Medullary Thyroid Cancer" Cancers 16, no. 17: 2979. https://doi.org/10.3390/cancers16172979

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Medicine (Baltimore)
v.95(29); 2016 Jul

A case report of isolated lymphadenopathy revealing localized leishmanial lymphadenopathy in an asthenic 25-year-old man

Quentin hurlot.

a Department of Pathology, Institut Universitaire du Cancer

Judith Fillaux

b Department of Parasitology, Toulouse University Hospital

Camille Laurent

c INSERM, Centre de Physiopathologie Toulouse-Purpan, Toulouse

Antoine Berry

Paul hofman.

d Department of Pathology, Nice University Hospital, Nice

Bruno Marchou

e Department of Infectious and Tropical Diseases, Toulouse University Hospital

Pierre Delobel

Pierre brousset.

f CRCT INSERM U1037, Toulouse

g Laboratoire d’Excellence Toulouse Cancer LABEX-TOUCAN, France.

Guillaume Martin-Blondel

Background:.

Visceral leishmaniasis (VL) is endemic in large areas of the tropics, the subtropics, and the Mediterranean basin. Besides classical VL presentation, exceptional cases of a limited form of VL have been reported. Here we describe the challenges of diagnosis and management of this intriguing entity.

Case summary:

A 25-year-old French Caucasian man presented with marked asthenia that had lasted 6 months and was strictly isolated except for a 2-cm left cervical lymphadenopathy. The rest of the clinical examination and extensive biological exploration were unremarkable.

Histological examination of the cervical lymphadenopathy showed a reactive lymphoid hyperplasia with granulomatous organization associated with small particles in the cytoplasm of epithelioid histiocytes and giant cells evocative of Leishman–Donovan bodies. Polymerase chain reaction (PCR) performed on the tissue confirmed the presence of Leishmania donovani/infantum DNA. Direct examination of a bone marrow aspiration, together with blood and bone marrow PCR, did not find other evidence for VL. Serology for leishmaniasis was unreactive. Extensive work-up for other causes of granulomatous lymphadenitis was negative. A diagnosis of localized leishmanial lymphadenopathy was made. Intravenous liposomal amphotericin B (20 mg/kg in five infusions) was initiated and well tolerated. Asthenia disappeared promptly and the patient fully recovered.

Conclusion:

Localized lymph node enlargement because of leishmanial infection should be included in the differential diagnosis of lymphadenopathy of unknown origin in patients who stayed or visited, even a long time ago and for a short period, endemic areas for leishmaniasis such as the Mediterranean basin. Fine-needle aspiration cytology and/or PCR for Leishmania sp of the lymphadenopathy might contribute to the diagnosis. A low-dose liposomal amphotericin B treatment might be effective, and deserves further study.

1. Introduction

Visceral leishmaniasis (VL) is a zoonotic vector-borne disease caused by the Leishmania donovani protozoa complex (L donovani and Leishmania infantum ). [ 1 ] VL is endemic in large areas of the tropics, subtropics, and the Mediterranean basin and can occur in travelers coming back from affected countries. The complex parasite–host interactions that predispose some individuals to developing the disease or to controlling the infection influence a spectrum of clinical manifestations ranging from asymptomatic to fatal visceral infections. [ 2 ] Here we report an unusual case of VL presenting as an isolated cervical lymphadenopathy in an immunocompetent host.

2. Case presentation

A 25-year-old French Caucasian man presented in December 2014 with marked asthenia that had lasted 6 months. He had no fever, weight loss, or night sweats and was asymptomatic except for this profound asthenia. He had no past medical history. This patient was born and lived in Paris, France, and had moved 2 years before Toulouse, in the southwestern part of France. He traveled to Egypt for 1 week in 2009, spent 1 month in China and Vietnam in July 2013, and 2 weeks in Corsica in September 2013. The clinical examination was unremarkable except for a firm and mobile 2 cm left cervical lymphadenopathy without inflammatory signs. In particular, he had no other lymphadenopathy, nor any hepatosplenomegaly, and the otorhinolaryngology examination was normal. Laboratory blood tests showed normal cell count, creatinine, C-reactive protein, hepatic enzyme, and lactate dehydrogenase levels. Erythrocyte sedimentation rate, as well as serum protein electrophoresis, was normal. Serologies for human immunodeficiency virus (HIV), syphilis, and Bartonella henselae were unreactive. Serology for toxoplasmosis showed IgG without IgM. A computed tomography scan of the neck, chest, abdomen, and pelvis showed no other lymphadenopathy, and no evidence for sarcoidosis, tuberculosis, or cancer. The cervical lymphadenopathy was biopsied. The histologic examination of the lymphadenopathy revealed a reactive lymphoid hyperplasia with granulomatous organization. Hematoxylin and eosin (Fig. (Fig.1A), 1 A), Periodic Acid–Schiff and Grocott (not shown) colorations showed small particles in the cytoplasm of epithelioid histiocytes and giant cells evocative of Leishman–Donovan bodies. Polymerase chain reaction (PCR) performed on the biopsy confirmed the presence of L donovani/infantum DNA [ 3 ] and was negative for Toxoplasma gondii . Immunohistochemistry using a monoclonal antibody p19–11 raised against the Leishmania homologue of receptors for activated C-kinase was also positive (diluted 1:80, [ 4 ] Fig. Fig.1B). 1 B). Immunohistochemistry for T gondii ( T gondii Ab-1 Rabbit polyclonal antibody) and HIV (anti-HIV-1 p24 antibody), and in situ hybridization for Epstein–Barr virus-encoded RNA, were negative. Direct examination of a bone marrow aspiration, together with blood and bone marrow PCR, did not find other evidence for VL. Serology for leishmaniasis was unreactive by indirect fluorescent assay ( vircell leishmania indirect fluorescent antibody IgG L infantum ) and immunochromatography (BioRad-IT Leish “ L infantum ”), but homemade L infantum -specific Western blot showed 14- and 18-kDa bands. [ 5 ] Extensive work-up for other causes of granulomatous lymphadenitis, including PCR in blood, stool, and saliva for Tropheryma whipplei , was negative. A diagnosis of localized leishmanial lymphadenopathy was made. Intravenous liposomal amphotericin B (20 mg/kg in 5 infusions) was initiated and well tolerated. Asthenia disappeared promptly and the patient fully recovered.

An external file that holds a picture, illustration, etc.
Object name is medi-95-e3932-g001.jpg

Histopathology of the cervical lymphadenopathy. (A) Hematoxylin and eosin coloration (magnification ×1000) of the lymphadenopathy showing granulomatous organization with giant cells and epithelioid histiocytes. Arrows show Leishman–Donovan bodies. (B) Immunohistochemistry of the lymphadenopathy using a monoclonal anti-Leishman antibody (magnification ×1200), demonstrating leishmanial parasites with their nuclei (arrows).

3. Discussion

After being transmitted by the bite of phlebotomine sand flies, leishmanial amastigotes disseminate through the lymphatic and vascular systems and infect the reticulo-endothelial system, resulting in infiltration of the bone marrow, liver, spleen, and lymph nodes. [ 1 ] Approximately 0.2 to 0.4 million new VL cases occur each year, with an overall case fatality rate of 10%. The most common presentation of VL includes signs of persistent systemic infection (high-grade fever, asthenia, anorexia, and weight loss) and of parasitic invasion of the blood and reticulo-endothelial system (lymphadenopathy, hepatomegaly, splenomegaly, pancytopenia, and hypergammaglobulinemia). VL symptoms often persist for several weeks to months before patients either seek medical care or die from bacterial co-infections, massive bleeding, or severe anemia. Liposomal amphoterin B is used as first-line treatment in Europe and the United States. [ 6 ] However, asymptomatic infections are far more frequent than symptomatic VL cases in immunocompetent hosts, demonstrating that many people infected with visceral leishmanial species develop an effective immune response and do not manifest the clinical disease. [ 7 ] In between, rare cases of a limited form of VL presenting as isolated lymphadenopathy without other evidence of VL have been described as localized leishmanial lymphadenopathy. [ 8 ] The clinical presentation of this emerging entity has been recently defined in 17 consecutive patients presenting with localized leishmanial lymphadenopathy during a large outbreak of L infantum VL that occurred in Spain. [ 9 ] Localized leishmanial lymphadenopathy exclusively affects immunocompetent hosts. As in the case described here, patients presented with indolent lymphadenopathy affecting mainly the cervical area (65%) without fever or systemic symptoms. Diagnosis can be made by fine-needle aspiration cytology of the lymphadenopathy, showing granulomatous lymphadenitis with leishmanial parasites. PCR for L donovani/infantum DNA on aspiration might also be a useful tool. Serological testing was not reliable, as half of the patients in this study had negative results. Although no consensus on therapeutic management of localized leishmanial lymphadenopathy has been formulated, [ 6 ] patients were usually treated for VL. Regarding that form of the disease, in the Spanish study, 2 patients displayed lymphadenopathies affecting different areas, and 1 had clinical and biological signs evocative of early stages of typical VL, suggesting that localized leishmanial lymphadenopathy might evolve toward a systemic disease. [ 9 ] However, treatments received were heterogeneous, and liposomal amphoterin B was used, with total dose varying from 10 to 30 mg/kg. Overall, the outcome was favorable, regardless of the dose used. Because patients suffering from localized leishmanial lymphadenopathy are not immunocompromised and the disease is still limited to the lymph node, the use of lower total doses of liposomal amphoterin B (10 mg/kg) has been advocated.

4. Conclusion

Localized lymph node enlargement because of leishmanial infection should be included in the differential diagnosis of lymphadenopathy of unknown origin in patients who stayed or visited, even a long time ago, endemic areas for leishmaniasis such as the Mediterranean basin (Table (Table1). 1 ). Fine-needle aspiration cytology and/or PCR for Leishmania sp of the lymphadenopathy might contribute to the diagnosis. A low-dose liposomal amphotericin B treatment might be effective and deserves further study.

Main causes of localized lymphadenopathy (enlargement of a single node or multiple contiguous nodal regions, adapted from [ 10 ] ).

An external file that holds a picture, illustration, etc.
Object name is medi-95-e3932-g002.jpg

Acknowledgments

The authors would like to thank David and Virginia Clark for their kind help in English editing.

Abbreviations: HIV = human immunodeficiency virus, PCR = polymerase chain reaction, VL = visceral leishmaniasis.

The authors have no conflicts of interest to disclose.

Open access
Published: 31 August 2024

The links between symptom burden, illness perception, psychological resilience, social support, coping modes, and cancer-related worry in Chinese early-stage lung cancer patients after surgery: a cross-sectional study

Yingzi Yang ORCID: orcid.org/0000-0003-4242-0444 1 , 2 na1 ,
Xiaolan Qian 1 na1 ,
Xuefeng Tang 1 ,
Chen Shen 2 ,
Yujing Zhou 3 ,
Xiaoting Pan 2 &
Yumei Li 4

BMC Psychology volume 12 , Article number: 463 ( 2024 ) Cite this article

1 Altmetric

Metrics details

This study aims to investigate the links between the clinical, demographic, and psychosocial factors and cancer-related worry in patients with early-stage lung cancer after surgery.

The study utilized a descriptive cross-sectional design. Questionnaires, including assessments of cancer-related worry, symptom burden, illness perception, psychological resilience, coping modes, social support and participant characteristics, were distributed to 302 individuals in early-stage lung cancer patients after surgery. The data collection period spanned from January and October 2023. Analytical procedures encompassed descriptive statistics, independent Wilcoxon Rank Sum test, Kruskal-Wallis- H - test, Spearman correlation analysis, and hierarchical multiple regression.

After surgery, 89.07% had cancer-related worries, with a median (interquartile range, IQR) CRW score of 380.00 (130.00, 720.00). The most frequently cited concern was the cancer itself (80.46%), while sexual issues were the least worrisome (44.37%). Regression analyses controlling for demographic variables showed that higher levels of cancer-related worry (CRW) were associated with increased symptom burden, illness perceptions, and acceptance-rejection coping modes, whereas they had lower levels of psychological resilience, social support and confrontation coping modes, and were more willing to obtain information about the disease from the Internet or applications. Among these factors, the greatest explanatory power in the regression was observed for symptom burden, illness perceptions, social support, and sources of illness information (from the Internet or applications), which collectively explained 52.00% of the variance.

Conclusions

Healthcare providers should be aware that worry is a common issue for early stage lung cancer survivors with a favorable prognosis. During post-operative recovery, physicians should identify patient concerns and address unmet needs to improve patients’ emotional state and quality of life through psychological support and disease education.

Peer Review reports

Introduction

Lung cancer is a common malignant neoplasm that often causes considerable psychological distress to patients and their families [ 1 ]. Due to the increasing public awareness of health screening in recent years and the use and promotion of low-dose computed tomography (CT) screening for early screening and detection of lung cancer, the incidence of early-stage lung cancer has been increasing [ 2 ]. According to the clinical diagnostic criteria for lung cancer, early-stage non-small cell lung cancer refers to a tumor that is confined to the lung and has not metastasized to distant organs or lymph nodes, generally referring to stage I and II [ 3 , 4 ]. For individuals diagnosed with early-stage non-small cell lung cancer, radical surgical resection offers the most beneficial treatment option for extended survival [ 5 , 6 ]. It can be said that after diagnosis and active treatment of early-stage lung cancer patients, the recurrence rate 5 years after surgery is low. Although the survival rate after radical resection has improved, a decline in lung function is inevitable. According to studies [ 7 , 8 ], lung function experienced a steep decline at 1 month after lung resection, partially recovered at 3 months, and stabilized at 6 months after surgery. Patients who have undergone surgery for lung cancer often experience post-treatment symptoms, including pain, dyspnea, and fatigue, which negatively affect their quality of life [ 9 ]. A previous study conducted by our team [ 10 ] found that post-operative lung cancer patients had various unmet needs during their recovery, including physiological, safety, family and social support, and disease information. The psychological distress experienced by patients, including worry, anxiety, and fear, increases due to unmet needs after cancer treatments [ 11 , 12 ].

In recent years, there has been an increasing focus on Cancer-related worry (CRW) as a form of psychological distress experienced by cancer patients. CRW refers to the uncertainty of cancer patients’ future after cancer diagnosis. It encompasses areas of common concern to cancer patients, such as cancer itself, disability, family, work, economic status, loss of independence, physical pain, psychological pain, medical uncertainty, and death. The purpose of CRW is to reflect the unmet needs or concerns of cancer patients [ 13 , 14 , 15 ]. Unlike anxiety, worry primarily reflects the patient’s repetitive thoughts about the uncertainty of the future [ 13 ]. It is also a cognitive manifestation of the uncertainty of disease prognosis [ 16 ].

Currently, measurement scales such as the State Train Anxiety Inventory (STAI) and the Hospital Anxiety and Depression Scale (HADS) are frequently used to evaluate physical symptoms caused by autonomic nervous activity in patients. However, they do not assess patients’ concerns or anxiety content [ 17 ]. Some scholars [ 13 , 17 , 18 ] have developed tools to measure the degree and content of cancer-related worries. The CRW questionnaire is a tool for measuring and evaluating the anxiety status of patients. It can also detect their needs or preferences through convenient means, allowing for the design of personalized care [ 13 ]. These questionnaires were primarily utilized to assess the level and nature of worry among cancer patients diagnosed with breast, prostate, skin, and adolescent cancers [ 19 , 20 , 21 , 22 ], However, it has not been employed in the post-operative population for early-stage lung cancer.

Theory framework

Mishel’s Uncertainty in Illness Theory [ 23 ] defines illness uncertainty as a cognitive state that arises when individuals lack sufficient information to effectively construct or categorize disease-related events. The theory explains how patients interpret the uncertainty of treatment processes and outcomes through a cognitive framework. It consists of three main components (see Fig. 1 ): (a) antecedents of uncertainty, (b) appraisal of uncertainty, and (c) coping with uncertainty. The antecedents of uncertainty include the stimulus frame (such as symptom burden), cognitive capacity (like disease perception), and structure providers (including social support and sources of disease information). Managing uncertainty requires coping modes such as emotional regulation and proactive problem-solving. Studies [ 24 ] have shown that there is a correlation between the way patients manage their emotions and the coping modes they experience when faced with difficulties. Patients with positive emotional coping modes are less likely to worry, while patients with avoidance coping modes are more likely to experience emotional distress. Furthermore, previous research [ 25 ] has shown a strong correlation between psychological resilience and cancer-related worries. Psychological resilience reflected an individual’s ability to adapt and cope with stress or adversity, and was an important indicator of a patient’s psychological traits [ 26 ]. Furthermore, it had a significant impact on their mental state and quality of life. Specifically, higher levels of cancer worries have been linked to lower levels of psychological resilience.

Based on the theoretical framework and literature research presented, it is hypothesized that factors such as psychological resilience, antecedents of disease uncertainty (symptom burden, disease perception, social support and sources of disease information), and coping modes will correlate with the level of cancer-related worries in postoperative patients with early-stage lung cancer. Therefore, the aim of this study was to investigate the potential correlates of cancer-related worry in early-stage lung cancer patients after surgery. This analysis will aid medical professionals in comprehending the worry state and unmet needs of early-stage lung cancer patients after surgery, and in tailoring rehabilitation programs and psychological interventions accordingly.

Mishel’s uncertainty in Illness theory framework

Study design

The study was cross-sectional in design. The Stimulating the Reporting of Observational Studies in Epidemiology (STROBE) checklist was completed (see S1 STROBE Checklist).

Setting and participants

This study included patients who underwent surgical treatment for lung cancer at a general hospital in Shanghai between January and October 2023. The study inclusion criteria were: (1) age over 18 years; (2) clinical diagnosis of early-stage primary non-small cell lung cancer (NSCLC), Tumor node metastasis classification (TNM) I to II [ 27 , 28 ], and received video-assisted thoracic surgery; (3) recovery time after surgery was within 1 month. Exclusion criteria were: (1) Postoperative radiotherapy and chemotherapy or second surgery may be required. (2) If other malignant tumors are present, they may also need to be treated. (3) Patients with severe psychological or mental disorders may not be candidates for this survey. (4) Patients with speech communication difficulties or hearing and visual impairments may require additional accommodations.

Sample size was calculated using G*Power software version 3.1.9 [ 29 ]. Calculate power analysis using F-test and linear multiple regression: fixed model, R2 increase as statistical test, and “A priori: Calculate required sample size - given alpha, power, and effect size” as type of power analysis. Cohen’s f2 = 0.15, medium effect size, α = 0.05, power (1-β) = 0.80, number of tested predictors = 8, total number of predictors = 10 as input parameters. The analysis showed that the minimum required sample size was 109 adults. Finally, the required sample size was determined to be over 120 adults with a probability of non-response rate of 10%.

Research team and data collection

The research team consisted of two nursing experts, four nursing researchers (one of whom had extensive experience in nursing psychology research), a group of clinical nurses, and several research assistants. To ensure the scientific quality and rigor of the research, the team was responsible for overseeing the quality of the project design and implementation process. Prior to conducting the formal survey, all research assistants received consistent training and evaluation to ensure consistent interpretation of questionnaire responses.

One month after surgery marks the first stage of the early recovery process for lung cancer patients and is a critical period for psychological adjustment [ 30 ]. Understanding a patient’s psychological state is critical to providing excellent medical care. During this period, patients typically need to visit the outpatient clinic for wound suture removal and dressing changes, further emphasizing the importance of postoperative recovery. Therefore, we chose this time frame for our research to gain a more comprehensive understanding of patients’ psychological well-being.

Prior to the start of the study, the hospital management and department head worked with us to provide comprehensive and standardized training to all participating investigators to ensure the reliability and consistency of the study. Research assistants rigorously screened patients based on predefined inclusion and exclusion criteria, which were designed to ensure a representative sample of lung cancer patients in the early recovery phase.

To obtain informed consent, patients were provided with detailed information and explanations and asked to complete the questionnaire voluntarily. To accommodate the different needs and preferences of patients, we used a combination of electronic and paper questionnaires. The electronic questionnaires were administered via the Questionnaire Star platform ( https://www.wjx.cn/ ), allowing patients to scan a two-dimensional (QR) code to access the survey. For those who preferred paper questionnaires, research assistants provided physical copies and assisted with completion as needed. The questionnaire was designed with consistent instructions to ensure that patients had a full understanding of the questions. Upon completion, each questionnaire was carefully reviewed and checked for accuracy. In addition, to reduce attrition, we obtained patients’ consent to keep their contact information, which allowed us to communicate with them further and collect additional data.

Ethical considerations

The study was approved by the hospital ethics committee. All participants gave informed consent prior to enrollment.

Sociodemographic questionnaire

We developed a sociodemographic survey based on a literature review and expert consultation. The survey includes patient demographic data such as age, sex, residence, lifestyle, education, marital status, childbearing history, religion, insurance, economic status (annual household income), smoking habits, employment status, sources of disease information, and previous psychological counseling. Clinical case information includes physical comorbidity, clinical tumor stage, and cancer type.

Cancer-related worry

The study measured participants’ cancer-related worry using the Brief Cancer-related Worry Inventory (BCWI), which was originally designed by Hirai et al. [ 13 ] to evaluate distinct concerns and anxiety levels among individuals with cancer. For this research, we used the 2019 Chinese edition of the BCWI, as introduced and updated by He et al. [ 31 ] (see Supplementary Table 2 ). The BCWI was comprised of 16 items, which were divided into three domains: (1) future prospects, (2) physical and symptomatic problems, and (3) social and interpersonal problems. Participants were asked to assess their cancer-related worries on a scale ranging from 0 to 100. Worry severity was determined by summing the scores for each item. The higher the total score, the more intense the patient’s cancer-related worry. The BCWI provided a concise evaluation of cancer-related worries in cancer patients. With only 16 items, it was able to differentiate them from symptoms of anxiety, depression, and post-traumatic stress disorder. In this study, the Cronbach’s alpha coefficient for this scale was 0.96.

Symptom burden

The M.D. Anderson Symptom Assessment Scale [ 32 ] was a widely used tool for evaluating symptom burden in cancer patients. The Chinese version of MDASI-C was translated and modified by researchers at the M.D. Anderson Cancer Center. The questionnaire included 13 multidimensional symptom items, such as pain, fatigue, nausea, restless sleep, distress, and shortness of breath, forgetfulness, loss of appetite, lethargy, dry mouth, sadness, vomiting, and numbness. Six additional items were used to evaluate the impact of these symptoms on work, mood, walking, relationships, and daily enjoyment. Patients rated the severity of their symptoms over the past 24 h on a scale of 0 (absent) to 10 (most severe), providing a comprehensive assessment of symptom burden across multiple dimensions. The Cronbach’s alpha coefficient for this study was 0.95.

Illness perception

The Brief Illness Perception Questionnaire (BIPQ) was used to measure participants’ illness perception. The scale, developed by Broadbent [ 33 ], was later revised by Mei [ 34 ] to include a Chinese version. It consisted of eight items divided into cognition, emotion, and comprehension domains as well as one open-ended question (What are the three most important factors in the development of lung cancer, in order of importance? ). The study utilized a 10-point Likert scale to rank items one through eight, with a range of 0–80 points. The ninth item required an open response. A higher total score indicated a greater tendency for individuals to experience negative perceptions and perceive symptoms of illness as more severe. In this study, the Cronbach’s alpha coefficient for this scale of eight scoring items was found to be 0.73.

Psychological resilience

The study measured participants’ psychological resilience using the 10-item Connor Davidson Resilience Scale (CD-RISC-10), originally developed by Connor and Davidson [ 35 ], and later revised by Campbell based on CD-RISC-25. The scale was designed to assess an individual’s level of emotional resilience in a passionate environment. It comprised 10 items, rated on a 5-point Likert scale (0 = never, 1 = rarely, 2 = sometimes, 3 = often, 4 = always). The total score ranged from 0 to 40 points, with higher scores indicating greater resilience. The study utilized the Chinese version of CD-RISC10, which was translated and revised by Ye et al. [ 36 ], to measure psychological resilience. The Cronbach’s alpha coefficient of the scale in this study was 0.96.

Coping modes

The study measured participants’ coping modes using the Medical Coping Modes Questionnaire (MCMQ), a specialized tool for measuring patient coping modes. The MCMQ was first designed by Feifel in 1987 [ 37 ] and was translated and revised into Chinese by Shen S and Jiang Q in 2000 [ 38 ]. It consisted of 20 items and three dimensions: confrontation (eight items), avoidance (seven items), and acceptance-resignation (five items). The study utilized a 4-point scoring system to evaluate coping events, with scores ranging from 1 to 4 based on the strength of each event. Eight items (1, 4, 9, 10, 12, 13, 18, and 19) were negatively scored, resulting in a total score range of 20 to 80 points. A higher score indicated a more frequent use of this coping mode. The three dimensions of this scale can be split into three scales for separate use. The reliability coefficients of the Confrontation Coping Mode Scale, Avoidance Coping Mode Scale, and Acceptance-resignation Coping Mode Scale were 0.69, 0.60, and 0.76.

Social Support

The study used the Perceived Social Support Scale (PSSS) developed by Zimet [ 39 ] to assess the level of self-understanding and perceived social support in postoperative lung cancer patients. The Chinese version of the PSSS, as adapted by Chou [ 40 ], showed satisfactory reliability and validity. The 12-item scale comprised of three dimensions: family, friend, and other support, and was rated using a 7-point Likert scale (1 = strongly disagree, 7 = strongly agree). The scale’s total score ranged from 12 to 84, with a higher score indicating a greater subjective sense of social support received by individuals. The Cronbach’s alpha coefficient for this scale in this study was 0.94.

Statistical analyses

The questionnaire was validated and double-checked before being input into Excel to ensure accuracy. Statistical analysis and processing of the questionnaire data were conducted using Statistical Product and Service Solutions (SPSS) 26.0 software. All statistical tests were two-sided with a significance level of α = 0.05. The quantitative data were presented using means and standard deviations, while the qualitative data were represented by frequency distributions. The data that conforms to normal distribution was analyzed using independent sample t-test for comparison between two groups and one-way ANOVA analysis for comparison between multiple groups. Non-normal distribution data were analyzed using non-parametric Wilcoxon rank sum test for comparison between two groups and Kruskal-Wallis - H -Test for comparison between multiple groups. Additionally, Spearman correlation analysis was used to study the correlations among CRW, MDASI-C, BIPQ, CD-RISC-10, MCMQ, and PSSS. A hierarchical linear regression analysis was performed to identify the multidimensional factors affecting CRW. All variables significantly correlated with the outcome variable ( p < 0.05) were included in the corresponding hierarchical regression analysis. Using Mishel’s Uncertainty in Illness Theory as a framework, a four-step model was adopted to study the factors influencing cancer-related concerns. The model includes individual factors such as sociodemographic and disease-related data, as well as psychological resilience, stimulus frame (symptom burden, illness perception, social support, and sources of disease information), and coping modes.

In this study, 307 questionnaires were distributed (227 electronic, 80 paper). Five paper questionnaires were invalid, resulting in 302 valid questionnaires and a recovery rate of 98.37%. The participants were 302 postoperative lung cancer patients, with 36.75% male and 63.25% female, aged 18–83 years (mean age 52.73, SD 13.07). Most patients (90.07%) were married. Additional demographic and clinical information is in Table 1 .

89.07% of people had cancer-related worries after surgery and median (interquartile range, IQR) score for CRW was 380.00 (130.00, 720.00) with a range of 0-1600. 86.42% reported worry about future prospects, 84.11% worry about physical and symptomatic problems, 79.80% worry about social and interpersonal problems. Among the 16 worry items of BCWI, the highest frequency of patient worries was “About cancer itself” (80.46%), followed by “About whether cancer might get worse in the future” (79.14%); the lowest frequency concern was “about sexual issues” (44.37%). According to the average score of the items in the three dimensions of CRW, it could be seen that patients had the highest standardized score (standardized score = median score/the total score of the dimension*100%) in future prospects (30.00%), the second standardized score in physical and symptomatic problems (20.00%), and the lowest in social and interpersonal problems (15.00%) as it is shown in Table 2 .

On the total score of the CRW scale, patients’ cancer-related worry was significantly correlated with their gender ( p = 0.009) and annual family income ( p = 0.018; see Table 1 ). The results suggested that gender and annual family income were related to the level of concern patients had after developing cancer. Additionally, there was a correlation between patients who received information about their disease from the internet or applications and their level of CRW ( p = 0.024; see Table 1 ).

The correlation analysis between various scales revealed a significant correlation ( p < 0.05; see Table 3 ) between CRW and MDASI-C, BIPQ, CD-RISC-10, and two dimensions of MCMQ (excluding avoidance). It was worth noting that avoidance coping modes did not show a correlation with CRW. In addition, according to the summary of the ninth open-ended question on the BIPQ, patients believed that the main causes of lung cancer were genetics, fatigue, stress from work, family or life, negative emotions (anger, worry) and unhealthy lifestyle (diet, work and rest, smoking), environmental factors (poor air quality, secondhand smoke, cooking fumes), and new coronavirus pneumonia (including vaccination, new coronavirus infection), etc. Further stratified linear regression analysis was conducted to determine the correlation between the dependent and independent variables (Table 4 ).

Table 4 presented the results of the hierarchical linear regression analysis for CRW in early-stage lung cancer patients. First, all scales included in the hierarchical regression analysis were tested for collinearity (variance inflation factor, VIF). The average VIF value was slightly above 1, indicating that the results were acceptable [ 41 ]. Second, the core research variables were divided into four levels according to the theoretical research, and the variables included in each level were analyzed separately. Model 1 included personal characteristics as independent variables and explained 5% of the variance in CRW. The analysis identified only two variables associated with CRW: being female (compared to male) and having low income (compared to high income). In model 2, psychological resilience was identified as an individual psychological characteristic and placed in the second level, resulting in a 9% increase in explanatory power. Model 3 added the antecedents of uncertainty, including symptom burden, illness perception, social support, and source of disease information, at the third level. This significantly increased the explanatory power of the overall regression model by 53%. The addition of coping modes, specifically confrontation coping mode and acceptance-resignation coping mode, to model 4 only increased the explanatory power of the overall regression model by 1%. The overall model demonstrated a total explanatory power of 68%. In Model 4, factors that significantly correlated with CRW included middle income( β = 2.17), psychological resilience( β =-2.42), symptom burden( β = 12.62), illness perception( β = 9.17), social support( β =-3.27), and source of disease( β = 2.01), as well as confrontation coping mode ( β =-1.98) and acceptance-resignation coping mode( β = 2.77), see Table 4 .

This study analyzed data from 302 patients to investigate the clinical, demographic, and psychosocial factors that correlated with cancer-related worry in patients with early-stage lung cancer after surgery. The study extended our understanding of the specific content and relevant factors of psychological distress in post-operative patients with early-stage lung cancer, a relationship that had not been fully investigated.

The study revealed that Chinese patients with early stage lung cancer were primarily concerned about their future prospects related to the disease itself, while sexual life problems caused by cancer were of least concern. This finding was consistent with previous studies [ 42 ], but this study provided more specific information on patients’ cancer-related worries. Lung cancer was widely perceived as a serious illness by the public due to its high cancer-specific mortality rate and low survival rate after diagnosis [ 43 ]. Consequently, patients with lung cancer often experience significant psychological distress after diagnosis. Even if the tumor was successfully removed, patients might still face challenges during recovery [ 44 ]. According to Reese’s research [ 45 ], long-term survivors of lung cancer experienced mild sexual distress. They also noted that sexual distress was significantly associated with physical and emotional symptoms. Although this study found that patients were least concerned about sexual distress, it should be noted that this study was based on the early stages of recovery after cancer surgery. Due to the postoperative repair of their body and emotions, patients were primarily focused on meeting their physiological and safety needs [ 10 ]. Further validation and exploration are required as there are limited studies on sexual distress in post-operative patients with early-stage lung cancer.

This study found that gender and annual family income were associated with the CRW of early-stage lung cancer patients. Among them, women and patients from low-income families had higher CRW scores, which was similar to the results of CRW in other cancer studies [ 14 , 46 ]. The reason might be that women were more conscious about uncomfortable symptoms than men, and women were more concerned about the duration of the disease and subsequent treatment effects than men [ 47 ]. Moreover, lower annual family income might cause patients to face more financial pressure in terms of medical expenses and treatment, thereby increasing their concerns about the consequences of the disease [ 48 ]. In addition, several other studies have found that education level, smoking status, and tumor stage had an impact on cancer-related worry scores [ 14 , 47 ], but this study did not show statistical significance.

After controlling for demographic covariate factors, the study found a significant negative relationship between psychological resilience and CRW. In a study conducted by Chen et al. [ 49 ], lower levels of psychological resilience were observed in post-operative lung cancer patients, which had a direct impact on their emotional state. In the context of treatment and recovery after lung cancer surgery, medical professionals should prioritize enhancing patients’ psychological resilience. This could be achieved through psychological support and appropriate interventions to improve emotional health and quality of life.

The antecedents in the illness uncertainty theoretical framework, such as symptom burden, illness perception, social support, and sources of disease information, were shown to be significantly associated with patients’ cancer worries in this study. The theory of uncertainty in illness [ 50 ] posited that uncertainty was caused by stimulus frames, cognitive abilities, and structural providers. In this study, these antecedents corresponded to symptom burden, illness perception, social support, and sources of disease information, and they correlated with patient worry related to cancer. Patients with a high symptom burden, high illness perception, low social support, and excessive attention to disease information on the Internet were more likely to have high cancer-related concerns. Previous studies [ 40 , 51 ] have verified the relationship between symptom burden, illness perception, and social support with psychological distress in lung cancer patients. However, there have been few studies on this patient group after surgery for early-stage lung cancer, particularly based on the uncertainty theoretical framework. Additionally, when analyzing the antecedents of structured providers, we included the sources from which patients receive information about their disease, particularly statistics from medical staff and online platforms. Our study found that patients who frequently accessed disease information on the internet had higher cancer-related worry scores. This was consistent with previous qualitative studies [ 10 ] which had shown that patients often turn to the internet for disease-related knowledge due to a perceived lack of effective information from medical staff.

This study also explored the association of CRW with coping modes. According to the theoretical framework of uncertainty in illness, coping modes are key for managing uncertainty, as uncertainty influences patients’ coping methods. Previous research [ 51 ] has shown that different coping modes can affect patients’ emotional states. Because this study focused primarily on the factors correlated with CRW, we included coping modes as independent variables in the linear regression analysis. The results indicated that CRW was negatively correlated with the coping mode of confrontation and positively correlated with the coping mode of acceptance-resignation. Acceptance-resignation, a negative coping mode, has been shown [ 52 ] to be associated with patients’ fear of disease progression and negative attitudes toward the disease, which decreases their confidence in treatment. Poręba-Chabros et al. [ 53 ] found that negative coping patterns were significantly associated with depression. When patients adopt an acceptance-resignation coping mode, their compliance behaviors decrease as they succumb to the disease. Conversely, confrontation, a positive coping mode, can enhance patients’ psychosocial adaptability, buffer psychological distress, and improve quality of life [ 52 ]. Interestingly, avoidance coping modes did not show a correlation with CRW in the postoperative population of early stage lung cancer, which is inconsistent with previous studies [ 54 ]. This lack of correlation may be due to several factors. The focus of our study on the first month after surgery may mean that patients are more focused on immediate physical recovery rather than engaging in avoidance behaviors. The nature of avoidance coping may temporarily alleviate worry without addressing underlying concerns, resulting in no measurable effect on CRW. Sample characteristics, measurement limitations, and individual differences in coping modes may also contribute to bias. In addition, strong psychological resilience, robust support systems, positive surgical outcomes, and increased health education may make avoidance modes less relevant or impactful on CRW in this population. Although the two coping modes were found to be associated with cancer-related worry in our study, hierarchical regression analysis showed that their influence on patients’ worry was small. Future research should explore the mechanisms of cancer-related worry and coping modes based on the theoretical framework of illness uncertainty. Given the association between cancer-related worry and coping modes, psychological intervention for patients should be emphasized in clinical practice. Healthcare professionals should conduct comprehensive psychological assessments and provide effective emotional support and education to help cancer patients develop more effective coping modes and reduce worry caused by uncertainty.

Strengths and limitations

This study presented evidence for CRW and the influencing factors that postoperative patients with early-stage lung cancer face. Based on the results, healthcare providers could identify the specific unmet needs of these patients more precisely and develop effective intervention strategies to improve their emotional state and quality of life. While the existing literature has extensively discussed psychological symptoms such as anxiety, depression, and fear in patients with mid-to-late stage lung cancer, relatively little research has been conducted on the mental health of postoperative patients with early-stage lung cancer, who are an important group of long-term lung cancer survivors [ 55 ]. As the number of patients diagnosed with early-stage lung cancer increases, so does concern about their mental health and unmet needs [ 56 , 57 ].

Nonetheless, this study also has some limitations. First, the single-center cross-sectional design and relatively small sample size may limit the generalizability of the findings to the broader population of early-stage lung cancer patients.

Second, while the study adopted a theoretical framework, it primarily conducted basic factor analysis without delving into the interaction mechanisms between the identified factors. This limitation restricts our understanding of how these factors interplay to influence cancer-related worry (CRW) in postoperative patients. In addition, while the CRW variable is largely operationalized in a similar way to the BIPQ and psychological resilience, we found no high correlation between these scales, as indicated by the Variance Inflation Factor (VIF). This means that multicollinearity, or the overlap between the variables, is not an issue in our study. However, we recognize that the scales we used may have limitations and might not fully capture the specific experiences of our sample. Therefore, future research should use different methods to measure CRW and related factors to better understand their individual effects on cancer-related worry.

Third, CRW is a dynamic process [ 20 ], and this study only focused on the patients’ situation within one month after surgery. This snapshot approach may not reflect the evolving nature of CRW over time. Longitudinal studies are needed to provide a more comprehensive understanding of how CRW and its influencing factors change throughout the postoperative recovery period.

These limitations suggest that future research should adopt a longitudinal design to analyze and verify the identified factors over an extended period. Additionally, expanding the study to include multiple centers and larger, more diverse sample sizes would enhance the generalizability of the findings. Exploring the interaction mechanisms between factors using advanced analytical methods could provide deeper insights into the complexities of CRW. By addressing these limitations, future research can build on our findings to offer more robust and generalizable evidence on the psychological distress experienced by postoperative early-stage lung cancer.

Implications for practice

The results of the study indicated that early-stage lung cancer patients in China had significant concerns about their future prospects, particularly regarding the disease itself, with less attention paid to the impact of cancer on sexual life. To facilitate postoperative recovery effectively, healthcare providers must promptly identify and address these specific concerns by incorporating routine psychological assessments and developing tailored intervention strategies, such as cognitive-behavioral therapy and resilience training, to improve patients’ mental health and overall well-being.

Moreover, this study identified psychological resilience, symptom burden, illness perception, social support, sources of disease information (from the Internet or applications), and coping modes of confrontation and acceptance-resignation as key predictors of cancer-related worry in postoperative early-stage lung cancer patients. Managing patients’ postoperative emotional states and enhancing their quality of life requires a deep understanding and proactive intervention by healthcare professionals. Specific strategies include providing psychological assessments, developing individualized care plans, facilitating support groups, and utilizing technology for continuous support.

The study provided insight into cancer-related worry among Chinese patients after surgery for early-stage lung cancer. The results showed that patients were most concerned about their future prospects, particularly the disease itself, while relatively little attention was paid to their sexual distress. The study identified several key factors that correlated with cancer-related worry, including psychological resilience, symptom burden, illness perception, social support, and sources of disease information from the internet, as well as coping modes. These findings emphasized the significance of healthcare providers identifying and addressing the individual needs of patients during post-operative recovery. It is important to improve the emotional state and quality of life of patients through psychological support and disease education. This study provides guidance for post-operative care of patients with early stage lung cancer and suggests avenues for future research. Specifically, further exploration of the mechanisms of these relationships and development of effective interventions are needed.

Data availability

The data that supported the findings of this study are available on request from the corresponding author. The data are not publicly available due to privacy or ethical restrictions.

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Acknowledgements

The authors would like to thank the nurses (Especially Gu Jingzhi and Liu Jing) and doctors at Shanghai Pulmonary Hospital for facilitating this study and all the patients who kindly participated in the survey.

Scientific clinical research project of Tongji University, JS2210319; Key disciplines of Shanghai’s Three-Year Action Plan to Strengthen Public Health System Construction (2023–2025), GWVI-11.1-28; The National Key Research and Development Plan Project of China, 2022YFC3600903.

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Yingzi Yang and Xiaolan Qian share first authorship.

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Department of Health Care, Shanghai Health and Medical Center, No. 67, Dajishan, Wuxi City, Jiangsu Province, 214063, People’s Republic of China

Yingzi Yang, Xiaolan Qian & Xuefeng Tang

School of Medicine, Tongji University, 1239 Siping Road, Shanghai, 200092, People’s Republic of China

Yingzi Yang, Chen Shen & Xiaoting Pan

Department of Thoracic Surgery, Shanghai Pulmonary Hospital, Tongji University, No.507, Zhengmin Road, Shanghai, 200433, People’s Republic of China

Yujing Zhou

Department of Nursing, Shanghai Pulmonary Hospital, Tongji University, No.507, Zhengmin Road, Shanghai, 200433, People’s Republic of China

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Y.Y. and Y. L designed the study. Y.Y., Y.Z., C.S and X.P. ran the study and collected the data. Y.Y., X.Q., X.T. and C.S. analyzed the data; Y.Y., and X. Q. interpreted the results and drafted the paper. Y.Y. wrote the main manuscript text and X.Q. prepared Tables 1 , 2 , 3 and 4 . Y.Y., X.T. and Y.L revised the manuscript. All authors read and approved the final version of the manuscript.

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Correspondence to Xuefeng Tang or Yumei Li .

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The study was approved by the institutional review board at the Shanghai Pulmonary Hospital (Q23–396). The procedures used in this study adhere to the tenets of the Declaration of Helsinki. Consent for publication Informed consent was obtained from all individual participants included in the study.

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Yang, Y., Qian, X., Tang, X. et al. The links between symptom burden, illness perception, psychological resilience, social support, coping modes, and cancer-related worry in Chinese early-stage lung cancer patients after surgery: a cross-sectional study. BMC Psychol 12 , 463 (2024). https://doi.org/10.1186/s40359-024-01946-9

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Published : 31 August 2024

DOI : https://doi.org/10.1186/s40359-024-01946-9

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Patients had no prior CRC and had no prior antidiabetic medication prescriptions between matched cohorts in the overall study population (A) and in patients with obesity/overweight (B). Kaplan-Meier analysis was used to estimate the probability of the outcome (first diagnosis of CRC) at daily time intervals with censoring applied within a 15-year time window starting from the index event (first prescription of glucagon-like peptide 1 receptor agonists [GLP-1RAs] vs other non–GLP-1RA antidiabetic medications). The cohorts were propensity score matched for demographics, adverse socioeconomic determinants of health, preexisting medical conditions, personal and family history of cancers such as CRC and colonic polyps, benign neoplasms of the colon and rectum, lifestyle factors (exercise, diet, smoking, and alcohol drinking), medical encounters, and procedures such as colonoscopy. Overall risk is defined as the number of incidence cases among the number of patients in each cohort at the beginning of the time window. A plus sign (+) indicates that a patient was prescribed a GLP-1RA or non–GLP-1RA antidiabetic medication, while a minus sign (−) indicates that they were not. AGI indicates alpha-glucosidase inhibitors; DPP-4, dipeptidyl-peptidase 4 inhibitors; SGLT2, sodium-glucose cotransporter-2 inhibitors; SU, sulfonylureas, TZD, thiazolidinediones.

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Wang L , Wang W , Kaelber DC , Xu R , Berger NA. GLP-1 Receptor Agonists and Colorectal Cancer Risk in Drug-Naive Patients With Type 2 Diabetes, With and Without Overweight/Obesity. JAMA Oncol. 2024;10(2):256–258. doi:10.1001/jamaoncol.2023.5573

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GLP-1 Receptor Agonists and Colorectal Cancer Risk in Drug-Naive Patients With Type 2 Diabetes, With and Without Overweight/Obesity

1 Center for Science, Health, and Society, Case Western Reserve University School of Medicine, Cleveland, Ohio
2 Departments of Internal Medicine, Pediatrics, and Population and Quantitative Health Sciences and the Center for Clinical Informatics Research and Education, The MetroHealth System, Cleveland, Ohio
3 Center for Artificial Intelligence in Drug Discovery, Case Western Reserve University School of Medicine, Cleveland, Ohio
4 Case Comprehensive Cancer Center, Case Western Reserve University School of Medicine, Cleveland, Ohio

Glucagon-like peptide 1 receptor agonists (GLP-1RAs) are approved by the US Food and Drug Administration for treating type 2 diabetes (T2D). GLP-1RAs have pleiotropic effects on lowering plasma glucose, inducing weight loss, and modulating immune functions. 1 Because overweight/obesity is a major risk factor for colorectal cancer (CRC), 2 we hypothesize that GLP-1RAs are associated with a decreased risk for CRC in patients with T2D compared with non–GLP-1RA antidiabetics. We conducted a nationwide, retrospective cohort study among drug-naive patients with T2D comparing GLP-1RAs with 7 non–GLP-1RA antidiabetics, including metformin and insulin, which are suggested to influence CRC risk. 3

We used the TriNetX platform to access deidentified electronic health records of 101.2 million patients, including 7.4 million with T2D from 59 health care organizations across 50 states. 4 TriNetX built-in analytic functions allow for patient-level analyses while only reporting population-level data. The MetroHealth System institutional review board determined that using data from TriNetX is not human subject research and therefore exempt from approval. The TriNetX platform has been shown to be useful for retrospective cancer cohort studies. 5 , 6

The study population comprised 1 221 218 patients with T2D who had medical encounters for T2D and were subsequently prescribed antidiabetic medications from 2005 to 2019, no prior antidiabetic medication use (drug naive), and no prior CRC diagnosis. GLP-1RAs were compared with insulin, metformin, alpha-glucosidase inhibitors, dipeptidyl-peptidase 4 (DPP-4) inhibitors, sodium-glucose cotransporter-2 (SGLT2) inhibitors, sulfonylureas, and thiazolidinediones. The time of 2005 to 2019 (except for a starting year of 2013 for SGLT2 inhibitors and 2006 for DPP-4 inhibitors) was chosen based on the year drugs were first approved. The study population was divided into exposure and comparison cohorts for each comparison.

Cohorts were propensity score matched (1:1, using nearest neighbor greedy matching) for demographics, adverse socioeconomic determinants of health, preexisting medical conditions, family and personal history of cancers and colonic polyps, lifestyle factors (exercise, diet, smoking, and alcohol drinking), and procedures such as colonoscopy 2 ( Table ). The outcome was the first diagnosis of CRC that occurred within 15 years starting from the index event (first prescription of GLP-1RAs vs non–GLP-1RA antidiabetics). With censoring applied, Kaplan-Meier analysis with hazard ratios (HRs) and 95% CIs were used to compare time to event rates at daily time intervals. Separate analyses were performed in patients stratified by the status of obesity/overweight and sex but not by age groups and race and ethnicity due to limited sample sizes. Data were collected and analyzed on September 13, 2023, within the TriNetX Analytics Platform using built-in functions (R, version 4.0.2 [R Project for Statistical Computing]), with statistical significance set at a 2-sided P < .05. More details are available in the eMethods in Supplement 1.

During a 15-year follow-up in 1 221 218 drug-naive patients with T2D, GLP-1RAs were associated with decreased risk for CRC compared with insulin (HR, 0.56; 95% CI, 0.44-0.72), metformin (HR, 0.75; 95% CI, 0.58-0.97), SGLT2 inhibitors, sulfonylureas, and thiazolidinediones, and with lower but not statistically significant risk compared with alpha-glucosidase or DPP-4 inhibitors ( Figure , A). Consistent findings were observed in women and in men. GLP-1RAs were associated with a lower risk for CRC in patients with obesity/overweight compared with insulin (HR, 0.50; 95% CI, 0.33-0.75), metformin (HR, 0.58; 95% CI, 0.38-0.89), or other antidiabetics ( Figure , B).

In this cohort study, GLP-1RAs were associated with reduced CRC risk in drug-naive patients with T2D with and without obesity/overweight, with more profound effects in patients with obesity/overweight, suggesting a potential protective effect against CRC partially mediated by weight loss and other mechanisms not related to weight loss. Study limitations include potential unmeasured or uncontrolled confounders, self-selection, reverse causality, and other biases inherent in observational studies, and that results need validation from other data and study populations. Further research is warranted to investigate the effects in patients with prior antidiabetic treatments, underlying mechanisms, potential differential effects within GLP-1RAs, and effects of GLP-1RAs on other obesity-associated cancers.

Accepted for Publication: September 24, 2023.

Published Online: December 7, 2023. doi:10.1001/jamaoncol.2023.5573

Corresponding Authors: Rong Xu, PhD, Center for Artificial Intelligence in Drug Discovery ( [email protected] ), and Nathan A. Berger, MD, Case Comprehensive Cancer Center ( [email protected] ), Case Western Reserve University School of Medicine, 10900 Euclid Ave, Cleveland, OH 44106.

Author Contributions: Drs Xu and Berger had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.

Concept and design: W. Wang, Kaelber, Xu, Berger,.

Acquisition, analysis, or interpretation of data: L. Wang, W. Wang, Xu, Berger.

Drafting of the manuscript: W. Wang, Xu, Berger.

Critical review of the manuscript for important intellectual content: All authors.

Statistical analysis: L. Wang, W. Wang, Xu.

Obtained funding: Xu.

Administrative, technical, or material support: Kaelber, Xu, Berger.

Supervision: Xu, Berger.

Conflict of Interest Disclosures: Dr Kaelber reported grants from the National Institutes of Health during the conduct of the study. Dr Xu reported grants from the National Institutes of Health during the conduct of the study. Dr Berger reported grants from the National Institutes of Health during the conduct of the study. No other disclosures were reported.

Funding/Support: This work was supported by the National Cancer Institute Case Comprehensive Cancer Center (CA221718, CA043703), the American Cancer Society (RSG-16-049-01-MPC), the Landon Foundation–American Association for Cancer Research (15-20-27-XU), the National Institutes of Health Director’s New Innovator Award Program (DP2HD084068), the National Institute on Aging (AG057557, AG061388, AG062272, AG07664), and the National Institute on Alcohol Abuse and Alcoholism (AA029831).

Role of the Funder/Sponsor: The funders had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.

Data Sharing Statement: See Supplement 2 .

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Case report
Open access
Published: 31 August 2024

Does gait influence biomechanics in a distal femoral osteotomy? An early post operative fracture after DFO above a Tomofix ® plate in a multiple sclerosis and low-density bone affected patient: choose a longer plate—a case report

Antongiulio Favero 1 ,
Domenico Alesi 1 , 2 ,
Vito Gaetano Rinaldi 1 , 2 ,
Tosca Cerasoli 1 ,
Stefano Zaffagnini 1 &
Giulio Maria Marcheggiani Muccioli 1 , 2

Journal of Medical Case Reports volume 18 , Article number: 400 ( 2024 ) Cite this article

Metrics details

Distal femur osteotomies are a well known and valuable treatment option to manage valgus malalignment with unicompartmental arthritis. Early postoperative complications are well known, and risk factors, such as pulmonary diseases, smoke, high dependent functional status, and body mass index, have been studied, but no study is available about osteotomies when gait is abnormal because of neurodegenerative conditions or when mineral density is below the normal rate.

Case presentation

We report the case of a 44 year-old female Mediterranean patient who underwent a biplanar distal femur opening wedge osteotomy surgery following a lateral meniscus total removal, which led to the subsequent development of lateral compartment osteoarthritis and pain, despite general comorbidities, such as multiple sclerosis. Additionally, 2 months later a supracondylar femur fracture above the previously applied Tomofix ® plate was reported. Fracture was treated by applying a LCP condylar 16 hole (336 mm) plate, a structural fibular graft, and strut fibular graft on the opposite side.

The overall aim of this case report is to provide a lesson to surgeons who want to perform a realignment surgery of the lower limb in patients with abnormal gait. Not only mechanical axes are to be considered, but also bone density, patient’s gait, and load force distribution along the bone stock. Emerging literature on three-dimensional cutting guides fails to account for these factors, thus promoting a standardized approach to surgery across all patients. The present case highlights a patient with low bone density and abnormal force distribution resulting from a pathologic neurodegenerative gait. In such cases, treatment decisions must carefully consider the biomechanical vulnerabilities of the native bone and the distribution of vector forces. These conditions must lead the choice toward a longer plate if an osteotomy is indicated, because surgery is more likely to fail.

Peer Review reports

Introduction

Varus and valgus malalignment with isolated medial or lateral wear of knee compartments can be treated in young and active patients by performing a high tibial osteotomy (HTO) or a distal femoral osteotomy (DFO), shifting the mechanical axis of the lower limb, and thus distributing load equally on knee compartments.

Early postoperative complications include recurrence of deformity, infection, joint contracture, hemartrosis, loss of posterior slope, compartment syndrome, neurovascular injury, hardware failure, delayed union, nonunion, and fractures [ 4 ]. The latter ones are among the rarest complications, and usually occur after falls [ 2 ].

Preoperative planning of osteotomies should include anterior–posterior weight bearing X-rays of the lower limbs and lateral projections of the knee [ 1 , 2 , 4 , 6 , 7 ], but no study is available regarding the correlation between gait, distribution of forces, and mechanical axis surgical adjustments.

A 44 year-old Mediterranean woman (weight of 81 kg, height of 178 cm, body mass index of 25,6 kg/m 2 ) affected by a relapsing remitting form of multiple sclerosis in an initial phase, first presented to our outpatient clinic complaining of lateral left knee pain. Nothing relevant was found in the family history. In 2021, the patient underwent an arthroscopic subtotal lateral meniscectomy of the knee owing to a nonspecified lateral meniscal tear; in 2022 she then underwent an arthroscopic meniscal regularization because of residual pain. Physical examination revealed lateral pain during walking and lateral compartment tenderness at palpation and in response to valgus stress; ligaments were stable at stress maneuvers; range of movement (ROM) reported was 0–110°, limited by pain. Radiographs (Fig. 1 , left) revealed lateral compartment knee osteoarthritis and valgus femoral deformity. Therefore, an opening wedge DFO with an allograft wedge was scheduled. Initial planning showed lateral distal femoral angle (mLDFA) of 86,71° and medial proximal tibial angle (MPTA) of 87,01°; we therefore estimated a 5 mm medial opening cut to restore a neutral axis (Fig. 2 ).

X rays showing preoperative imaging, postoperative imaging, and post fracture imaging

From left to right: preoperative standing X-ray imaging, post distal femoral osteotomy standing X-ray imaging, and posttraumatic fracture fixation non-weight bearing panoramic X ray of the lower limb

Surgery was performed without any intraoperative complications. The patient developed postoperative anemia requiring a transfusion. No other complications were reported. The patient was discharged after 4 days. The rehabilitation program consisted in a knee brace blocked in extension for 30 days, which was removable to perform mobilization exercises; isometry exercises were allowed after 10 days, along with passive leg mobilization with kinetec device (starting with 0–35°). Flexion over 90° was not permitted in the first month. Finally, no weight bearing walking was allowed on the operated limb, and the blocked brace had to always remain on.

The patient came to visit as scheduled 30 days later in our outpatient clinic: knee ROM was 0–90°, X-rays were taken (Fig. 1 center), and she was prescribed progressive weight bearing for 30 days, physiotherapy (muscular reinforcement) two times a week, and maximum permitted-kinetec-assisted passive flexion for 45 days.

Then, 20 days later the patient presented to our emergency room (ER) referring a sudden failure of the knee while walking, followed by pain, swelling, and functional impotence. X-rays (Fig. 1 , right) revealed a displaced distal femur fracture. The patient was therefore hospitalized, and surgery was scheduled. The patient also brought to our attention a recent preoperative dual-energy X-ray absorptiometry (DEXA) scan showing a −2,5 T score, which was not considered before, because of her young age.

At 6 days after admission, surgery was performed. By laterally accessing the femur, after retracting fascia lata, muscular fascia, and lateral vastus, a multifragmentary displaced fracture, proximal to the previously applied plate was reported. By protecting the previous osteotomy site with k-wires, we removed the plate and its screws. A structural fibular graft was then placed inside the femoral canal. A LCP condylar 16 hole (336 mm) plate was placed after anatomical reduction of the stumps. We placed both locking and nonlocking screws, using a second contralateral fibular graft. Surgery was performed without any complications.

During hospitalization venous bleeding led to multiple transfusions. No other complications were reported. Physiotherapy started 3 days later: no weight bearing on the affected limb and knee brace blocked in extension.

The patient was dismissed 5 days after surgery.

The patient came to visit in our outpatient clinic after 21 days. Stitches were removed and the surgical wound had healed properly. She was given indication to start rehabilitation: progressive weight bearing with crutches. Postoperative X-rays at 1 month showed good progression in bone healing and good positioning of the plate (Fig. 3 ). The patient was very satisfied with the result. After 6 months from the first surgery, the patient would like to correct the other limb alignment too.

X-ray imaging showing plate fixation at 1 month follow up

Discussion and conclusion

No superiority between opening and closing wedge DFO has been found in literature; however, Rosso et al . [ 7 ] reported higher intraoperative precision regarding correction and less postoperative plate intolerance with open wedge techniques. Literature reports high variability in complications after opening wedge DFO procedures, ranging from 0% to 30%, the main consisting of hardware related issues [ 2 ]. This one is included among early postoperative complications and can compromise surgery, because, as Chahla et al . [ 2 ] reported in his systematic review, bone healing time can range from 3 to 6 months. Berk et al . (2023) reported higher rates of early complications in DFO when compared with HTO (11.6% versus 21.5%), suggesting that this procedure may need a strict follow up, especially in the first months. This may be because DFO are performed closer to diaphyseal bone when compared with HTO, and growth potential differs between these areas [ 6 ]. We presented the case of a distal femoral fracture after an opening wedge DFO, which occurred only 2 months after the procedure. Despite that, alignment, and healing of the bone in the site of osteotomy were not compromised; still, this complication must be defined as early and hardware related, because the length of the plate caused anomalous distribution of force vectors along the bone stock, defining a locus minoris resistantiae just above the plate itself. According to a 2013 systematic review of Vena et al . analyzing complications after osteotomies, fractures usually happen around the site of wedge opening or just below, with the fracture line directed toward the articular portion and are seen intraoperatively or as late complications. To our knowledge, there are no other cases describing an early fracture above the opening wedge in a low-density bone patient. No studies considered the link between osteoporosis and osteotomies owing to the relatively young age of patients undergoing this procedure (better outcomes in patients < 60 years) [ 9 ]. This study could suggest that bone density should be considered in patients who are at risk of osteoporosis. Our case lacked the recognition of preoperative preexisting osteoporosis, which is well known to be linked to multiple sclerosis, such as some fracture patterns, as Yazdan Panah et al . [ 11 ] highlighted. Moreover, we must consider that the population is growing progressively older in first world countries and patients might refuse a knee replacement asking for an axis correction (as in osteotomies) if their functional requests are high.

Well known associated risk factors are age > 45 years, diabetes mellitus, chronic obstructive pulmonary disease, and smoking [ 4 ] and are related to failure of the procedure, mainly influencing soft tissue healing but also favoring infection. Moreover, high body mass index, hypertension, and dependent functional status were found to be risk factors [ 3 ]. Our patient did not present any of these risk factors, apart from dependent functional status. This evidence could suggest that we should improve our knowledge in this field to avoid complications such as in this case.

According to Berk et al . The most common complications after DFO are anemia requiring transfusion (14%) and readmission (4%). Our patient developed both.

Existing literature already evaluated outcomes of DFO in patients affected by monocompartimental osteoarthritis. Gait analysis is a valuable tool in this field. Regarding valgus correction, it is known that it can cause an increase in the abduction moment and a lateral shift in the dynamic knee joint loading. Varus osteotomies have less literature regarding these aspects, but recent studies seem to suggest that knee adduction moment can increase similarly. It is also well known that the peak knee flexion moment is strongly related to walking speed [ 10 ]. No literature exists regarding abnormal distribution of forces on the bone stock after surgeries in patients affected by neurodegenerative diseases. More specifically, no studies evaluated the link between abnormal gait and complications after DFO. Current literature suggests that coronal forces could prevail on other plane vectors during normal gait, above all when a slow gait or an intra-external rotated lower limb axis is considered. In our case, we had both a slow and wide gait, which could have led coronal forces to strongly overcome bone elastic module. Moreover, out patient’s bone was osteoporotic. We chose a Tomofix ® plate, because, as Rosso et al . [ 7 ] reported that, despite no clinical superiority being found, biomechanically it has greater axial and torsional stability. Positioning a too short plate, such as Tomofix ® ones commonly used for osteotomies, on an osteoporotic bone, can elicit peri-implant fractures. Abnormal gait, including poor coordination and balance issues, eventual spasticity, and widening of the docking station typical of neurodegenerative condition affected patients, may add a further risk factor. In these cases, we suggest applying a longer plate and eventually, if other risk factors are highlighted, a contralateral fibular strut on the other side primarily favoring bone regrowth.

Custom guides for osteotomies are becoming more and more popular among orthopedic surgeons, focusing above all on axis realignment, leaving aside how force vectors will distribute after our cuts. New vectors could destabilize previous bone deficiencies leading to surgical failures.

Increasing literature is emerging about distal femur fractures treated with fibular grafts and plating, showing promising results. Wen Chin Su [ 8 ] and Ibrahim [ 5 ] show that this application may have lower bone healing time and postoperative nonunion or femur collapse in varus rates, particularly when patients are older than 50 years, with a lower bone density (not necessarily pathological). Our patient resembled these risk factors; therefore, we applied the same type of cautions.

The overall aim of this case report is to provide a lesson to surgeons who want to perform a realignment surgery of the lower limb in patients with abnormal gait. Not only mechanical axes are to be considered, but also bone density, patient’s gait and load force distribution along the bone stock. Emerging literature on three-dimensional cutting guides fail to account for these factors, thus promoting a standardized approach to surgery across all patients. The present case highlights a patient with low bone density and abnormal force distribution resulting from a pathologic neurodegenerative gait. In such cases, treatment decisions must carefully consider the biomechanical vulnerabilities of the native bone and the distribution of vector forces. These conditions must guide the choice toward a longer plate if an osteotomy is indicated, because surgery is more likely to fail.

This case report has some limitations. First, follow up time is limited: a long-term follow could provide better clinical and radiographical data. Moreover, we expect this patient to experience a progressive neurological decay, which will affect our outcome in a nonpredictable and measurable way. We also did not report clinical scores prior to surgery.

Availability of data and materials

All the data discussed in the manuscript are in the databases of Istituto Ortopedico Rizzoli.

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Antongiulio Favero, Domenico Alesi, Vito Gaetano Rinaldi, Tosca Cerasoli, Stefano Zaffagnini & Giulio Maria Marcheggiani Muccioli

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Each author contributed to the article: GMMM performed the surgery, conceived and designed the paper, and revised the paper; AF contributed data and wrote the paper; SZ conceived and designed the paper and revised the paper.

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Favero, A., Alesi, D., Rinaldi, V.G. et al. Does gait influence biomechanics in a distal femoral osteotomy? An early post operative fracture after DFO above a Tomofix ® plate in a multiple sclerosis and low-density bone affected patient: choose a longer plate—a case report. J Med Case Reports 18 , 400 (2024). https://doi.org/10.1186/s13256-024-04739-1

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Distal femoral osteotomy
Knee fracture
Distal femur fracture
Low density bone
Neurodegenerative gait
Lateral compartment osteoarthritis
Fibular strut
Lower limb malalignment
Gait analysis

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A case report of isolated lymphadenopathy revealing localized leishmanial lymphadenopathy in an asthenic 25-year-old man

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