Pathological humerus fracture due to anti-interferon-gamma autoantibodies: A case report

doi:10.5312/wjo.v12.i11.938

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World Journal of Orthopedics

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Case Report

World J Orthop. Nov 18, 2021; 12(11): 938-944
Published online Nov 18, 2021. doi: 10.5312/wjo.v12.i11.938

Pathological humerus fracture due to anti-interferon-gamma autoantibodies: A case report

Cheng Hsun Yang, Feng-Chih Kuo, Chen-Hsiang Lee

Cheng Hsun Yang, Division of Infection Disease, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital, Kaohsiung 833, Taiwan

Feng-Chih Kuo, Department of Orthopaedic Surgery, Kaohsiung Chang Gung Memorial Hospital, Kaohsiung 833, Taiwan

Chen-Hsiang Lee, Division of Infection Disease, Department Internal Medicine, Chang Gung University, College Medicine, Chang Gung Memorial Hospital, Kaohsiung Medicine Centre, Kaohsiung 833, Taiwan

Author contributions: Yang CH reviewed the literature and contributed to manuscript drafting; Guo FZ was the patient’s orthopedist and contributed to manuscript drafting; Lee CH was responsible for the revision of the manuscript for important intellectual content; All authors issued final approval for the version to be submitted.

Informed consent statement: All study participants, or their legal guardian, provided informed written consent prior to study enrollment.

Conflict-of-interest statement: The authors declared no conflict of interest in our case report.

CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016) Statement, and the manuscript was prepared and revised according to the CARE Checklist (2016) Statement.

Corresponding author: Chen-Hsiang Lee, MD, Professor, Division of Infection Disease, Department Internal Medicine, Chang Gung University, College Medicine, Chang Gung Memorial Hospital, Kaohsiung Medicine Centre, 123 Ta Pei Road, Kaohsiung 833, Taiwan. lee900@adm.cgmh.org.tw

Received: June 8, 2021
Peer-review started: June 8, 2021
First decision: July 28, 2021
Revised: August 6, 2021
Accepted: September 15, 2021
Article in press: September 15, 2021
Published online: November 18, 2021
Processing time: 160 Days and 4.7 Hours

Abstract

BACKGROUND

Various etiologies contribute to pathological fractures, including bone infections. Recently, non-tuberculosis Mycobacterium-related bone infections among patients with anti-interferon-gamma autoantibody-induced adult-onset immunodeficiency has raised concerns in Southeast Asia, with the common presentations including osteomyelitis. However, it also rarely manifests as traumatic fractures, as reported in this case.

CASE SUMMARY

A diabetic female fractured her humerus after a traumatic accident and received fixation surgery. Abnormal necrotic bone tissue and abscess formation were noted, and she was diagnosed with a pathological fracture due to non-tuberculosis Mycobacterium infection. Multiple bone involvement was also revealed in a bone scan. Anti-interferon-gamma autoantibodies were then checked due to an unexplained immunocompromised status and found to be positive. Her humerus fracture and multiple bone infections healed after steroid and anti-non-tuberculosis Mycobacterium medication treatment following fixation surgery.

CONCLUSION

Comprehensive preoperative evaluations may help identify pathological fractures and guide the treatment course.

Keywords: Immunocompromised status; Neutralizing anti-interferon-gamma autoantibody; Non-tuberculosis mycobacterium; Pathological fracture; Pre-operative evaluations; Case report

Core Tip: Identifying neutralizing anti-interferon-gamma autoantibody-related non-tuberculosis Mycobacterium bone infections requires careful history taking and physical examinations. While a biopsy of the bone lesion is the gold standard for diagnosis, it is advisable to check co-existing lymphadenopathy, dermatoses, and lung and blood-stream infections, as they provide easily accessible specimens for culturing and cytopathology. Serum tests of immune profiles are also important for atypical or opportunistic infections. These pre-operative evaluations may guide the choice of surgical modality, medical treatment that accompanies surgery, and decide the prognosis of healing.