Yang CH, Kuo FC, Lee CH. Pathological humerus fracture due to anti-interferon-gamma autoantibodies: A case report. World J Orthop 2021; 12(11): 938-944 [PMID: 34888154 DOI: 10.5312/wjo.v12.i11.938]
Corresponding Author of This Article
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
Research Domain of This Article
Orthopedics
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
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.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
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
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.
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.
