A non-HIV case with disseminated Mycobacterium kansasii disease associated with strong neutralizing autoantibody to interferon-γ

Nei, Takahito; Okabe, Masahiro; Mikami, Iwao; Koizumi, Yasuo; Mase, Hiroshi; Matsuda, Kuniko; Yamamoto, Takeshi; Takeda, Satoru; Tanaka, Keiji; Dan, Kazuo

doi:10.1016/j.rmcr.2012.11.003

Cited by 13 publications

(14 citation statements)

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“…Negative changes in a positive control are caused by individual background factors, such as an immunocompromised status, including acquired immunodeficiency syndrome or immunosuppressant intake. The level of anti-IFN-γ autoantibodies, as reported in severe infectious diseases, including sepsis caused by opportunistic pathogens (18) or disseminated nontuberculous mycobacterium infection (19,20), is also high in this situation.…”

Section: Discussionmentioning

confidence: 76%

Miliary Tuberculosis with Indeterminate Interferon Gamma Release Assay Results

et al. 2013

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Recently, interferon gamma release assays (IGRAs) have become an important clinical tool for detecting latent tuberculosis. However, IGRA results may impede making a diagnosis. We herein present an interesting case of miliary tuberculosis with a nonspecific IGRA reaction due to hemophagocytosis. Case ReportA 64-year-old man with advanced chronic kidney disease due to nephrosclerosis and ischemic heart disease was admitted to our hospital with an intermittent high-grade fever lasting for one week. The plan was to initiate hemodialysis therapy, and the patient underwent surgery for internal shunt ostomy in the left upper arm two months prior to admission. He was given neither steroids nor other immunosuppressants. On admission, he was free from respiratory symptoms, such as a productive cough. His laboratory data and radiological findings were evaluated. The clinical laboratory data obtained on admission confirmed a high alkaline phosphatase level (above 2,000 IU/mL) and a decreased hemoglobin (Hgb) level (below 10.0 g/dL). The serum C-reactive protein and procalcitonin levels were elevated, indicating an inflammatory reaction. The soluble interleukin-2 receptor (sIL-2R) level was elevated to 18,986 U/mL. Both anti-HIV and anti-human T-cell leukemia virus (HTLV) antibodies were negative. He had no history of hematological diseases. Chest roentogenography performed on admission showed no abnormalities, except cardiomegaly ( Figure A).We initially suspected a bloodstream infection, such as infectious endocarditis, and thus performed blood cultures and serological surveillance. However, these microbiological inspections yielded no useful data. On the other hand, chest computed tomography revealed slight density changes in both lung fields ( Figure B); however, the patient had no respiratory symptoms. Although we recognized a slight density elevation in the pulmonary alveolar field, radiologists in our hospital did not mention these findings, and miliary tuberculosis was not suspected. The detection of a column appearance depends on the type of monitor used. We attempted to obtain respiratory samples to detect pulmonary infections, including tuberculosis. However, due to the lack of sputum production, we were unable to obtain respiratory samples. The interferon gamma release assay (IGRA, QuantiFERON-TB TM Gold In-tube (QFT-3G)) results showed values for both negative and positive controls as well as the patient's samples exceeding 8.01 IU/mL. According to previous guidelines for using IGRAs (1), our patient's results were invalid due to the nonspecific reaction of the negative control (a positive reaction in the negative control).Despite conducting further studies, a final diagnosis was not reached, and the patient's condition deteriorated. Hemo-

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Section: Discussionmentioning

confidence: 76%

Miliary Tuberculosis with Indeterminate Interferon Gamma Release Assay Results

et al. 2013

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“…Recent case reports have shown high titers of neutralizing anti-interferon-gamma (IFN-g) antibodies in the plasma of disseminated cases with normal immunity, affected downstream immunity induced by IFN-g (Doffinger et al, 2004;Hoflich et al, 2004;Kampmann et al, 2005;Patel et al, 2005;Koya et al, 2009;Nei et al, 2013). Based on these studies, immunoadsorption therapy or intravenous immunoglobulin therapy would appear to be hopeful for these patients with severe disseminated NTM due to anti-IFN-g antibodies.…”

Section: November 3 2016mentioning

confidence: 99%

Disseminated Mycobacterium kansasii infection with cutaneous lesions in an immunocompetent patient

Zhang

Feng

2017

International Journal of Infectious Diseases

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A case of disseminated Mycobacterium kansasii infection involving the skin and soft tissue in a 57-year-old male farmer who presented with recurrent fever, respiratory syndromes, and skin lesions is reported. The positive findings of syndromes, laboratory examinations, and identification of M. kansasii in puncture fluid indicated the diagnosis of disseminated M. kansasii infection involving the skin and soft tissue, lungs, and mediastinal lymph nodes. After applying the standard HRE regimen (isoniazid 300mg/day, rifampicin 600mg/day, and ethambutol 750mg/day), the patient's temperature normalized and his symptoms improved gradually. No notable adverse drug reactions occurred and the skin lesions had healed after 4 months of follow-up. Disseminated M. kansasii infections occur mainly in immunocompromised patients. Moreover, disseminated infections with skin lesions is rare in immunocompetent patients. Following a review of the literature, only eight similar cases were identified as of disseminated M. kansasii infection with cutaneous lesions, and thecase presented here appears to be the second involving an immunocompetent individual. Special attention should be paid to a persistent and chronic rash following a chronic respiratory syndrome in order to exclude skin disease caused by non-tuberculous mycobacteria.

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“…These included NEMO deficiency (one patient) (13) and defects in the IFN-g/IL-12 pathway: IFN-g receptor deficiency (one patient) (14), IL-12 receptor deficiency (one patient) (15), and autoantibodies to IFN-g (one patient) (16). Disseminated disease has been reported in a patient with a known GATA2 mutation (3) and a patient whose clinical history is compatible with GATA2 deficiency (4).…”

Section: Review Of the Literaturementioning

confidence: 99%

Mediastinal and Disseminated Mycobacterium kansasii Disease in GATA2 Deficiency

et al. 2016

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Rationale: Mycobacterium kansasii usually causes chronic pulmonary infections in immunocompetent patients. In contrast, disseminated M. kansasii disease is commonly associated with advanced human immunodeficiency virus infection, but is reported infrequently in other immunocompromised patients.Objectives: To identify common clinical manifestations and potential risk factors for M. kansasii infection in patients with GATA2 deficiency.Methods: We reviewed M. kansasii disease associated with GATA2 deficiency at one institution and disease associated with primary and other immunodeficiencies reported in the literature. Measurements and Main Results:Nine patients with GATA2 deficiency developed M. kansasii infections. Six patients developed disseminated disease. All patients presented with significant mediastinal lymphadenopathy or abscesses. Seven patients had pulmonary risk factors, including six smokers. The majority of patients had low numbers of neutrophils, monocytes, B cells, CD4 1 T cells, and natural killer cells. Other conditions associated with disseminated M. kansasii disease were thymic disorders and IFN-g/IL-12 defects.Conclusions: Disseminated M. kansasii disease involving mediastinal lymph nodes is surprisingly common in GATA2 deficiency, but also occurs in defects of IFN-g synthesis and response. Disseminated M. kansasii should be considered a marker indicating a need to evaluate for immunodeficiency syndromes.

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A non-HIV case with disseminated Mycobacterium kansasii disease associated with strong neutralizing autoantibody to interferon-γ

Cited by 13 publications

References 10 publications

Miliary Tuberculosis with Indeterminate Interferon Gamma Release Assay Results

Miliary Tuberculosis with Indeterminate Interferon Gamma Release Assay Results

Disseminated Mycobacterium kansasii infection with cutaneous lesions in an immunocompetent patient

Mediastinal and Disseminated Mycobacterium kansasii Disease in GATA2 Deficiency

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