期刊
JOURNAL OF CLINICAL IMMUNOLOGY
卷 39, 期 5, 页码 512-518出版社
SPRINGER/PLENUM PUBLISHERS
DOI: 10.1007/s10875-019-00652-1
关键词
Anti-interferon-gamma autoantibodies; non-tuberculous mycobacteria; immunodeficiency
类别
资金
- Clinician Scientist Award (CSA), Individual Research Grant (IRG), Bedside Bench (BB)
- National Medical Research Council (NMRC), Singapore
- National University Health System
- Microbiome BIGHEART grant of the National University of Singapore
Immunodeficiency secondary to anti-interferon-gamma (anti-IFN-gamma) autoantibodies was first described in 2004 as an acquired defect in the IFN-gamma pathway leading to susceptibility to multiple opportunistic infections, including dimorphic fungi, parasites, and bacteria, especially tuberculosis and non-tuberculous mycobacterium (NTM) species. It has so far only been described in adult patients. We present 2 cases of disseminated NTM infections in otherwise immunocompetent children. A 16-year-old girl with Sweet's syndrome-like neutrophilic dermatosis developed recurrent fever and cervical lymphadenitis secondary to Mycobacterium abscessus. A 10-year-old boy with a history of prolonged fever, aseptic meningitis, aortitis, and arteritis in multiple blood vessels developed thoracic vertebral osteomyelitis secondary to Mycobacterium avium complex. Both patients were found to have positive serum neutralizing anti-IFN gamma autoantibodies. Testing for anti-IFN gamma autoantibodies should be considered in otherwise healthy immunocompetent hosts with recurrent or disseminated NTM infection. This represents a phenocopy of primary immunodeficiency which has been recently described only in adults. We report the first two cases of this phenomenon to affect children.
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