Corneal Deposits in a Patient With Anti–Interferon-γ Autoantibody Syndrome

Ahmad, Tessnim R; Ashraf, Davin C.; Seitzman, Gerami D.

doi:10.1001/jamaophthalmol.2019.4925

Cited by 3 publications

(3 citation statements)

References 9 publications

(21 reference statements)

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“…Long-term antibiotic treatment can lead to other complications, such as liver and renal toxicity. One author reported one AOID patient with posterior corneal deposits after long-term treatment with rifabutin [ 36 ]. Thus, this disease urged high clinical awareness.…”

Section: Discussionmentioning

confidence: 99%

Characteristics and Outcomes of Anti-interferon Gamma Antibody-Associated Adult Onset Immunodeficiency

et al. 2023

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Purpose Anti-interferon gamma antibody (AIGA) is a rare cause of adult onset immunodeficiency, leading to severe disseminated opportunistic infections with varying outcomes. We aimed to summarize the disease characteristics and to explore factors associated with disease outcome. Methods A systematic literature review of AIGA associated disease was conducted. Serum-positive cases with detailed clinical presentations, treatment protocols, and outcomes were included. The patients were categorized into controlled and uncontrolled groups based on their documented clinical outcome. Factors associated with disease outcome were analyzed with logistic regression models. Results A total of 195 AIGA patients were retrospectively analyzed, with 119(61.0%) having controlled disease and 76 (39.0%) having uncontrolled disease. The median time to diagnosis and disease course were 12 months and 28 months, respectively. A total of 358 pathogens have been reported with nontubercular mycobacterium (NTM) and Talaromyces marneffei as the most common pathogens. The recurrence rate was as high as 56.0%. The effective rates of antibiotics alone, antibiotics with rituximab, and antibiotics with cyclophosphamide were 40.5%, 73.5%, and 75%, respectively. In the multivariate logistic analysis, skin involvement, NTM infection, and recurrent infections remained significantly associated with disease control, with ORs of 3.25 (95% CI 1.187 ~ 8.909, P value = 0.022), 4.74 (95% CI 1.300 ~ 17.30, P value = 0.018), and 0.22 (95% CI 0.086 ~ 0.551, P value = 0.001), respectively. The patients with disease control had significant AIGA titer reduction. Conclusions AIGA could cause severe opportunistic infections with unsatisfactory control, particularly in patients with recurrent infections. Efforts should be made to closely monitor the disease and regulate the immune system.

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Section: Discussionmentioning

confidence: 99%

Characteristics and Outcomes of Anti-interferon Gamma Antibody-Associated Adult Onset Immunodeficiency

et al. 2023

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“…Before the introduction of AIGArelated immunotherapy, the overall improvement rate was 45.1%, and >50% of patients could not achieve remission; there was a 12.3% mortality rate, 8.7% recurrence rate, and 33.9% persistent infection rate. In this study, 67 patients underwent immunotherapy for AIGA (Supplementary Table 2) (6,7,(13)(14)(15)(16)(17)(18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30)(31), and the duration of anti-NTM or antifungal treatment in 29 patients was clearly described. The median duration of anti-NTM therapy was 15 (interquartile range: 7, 36) months, and in all of the 67 cases, infection was caused by persistent infection or deterioration after long-term regular antimicrobial therapy.…”

Section: Attempt Immunotherapy In Patients With Aiga Positivitymentioning

confidence: 99%

“…However, obvious skin damage, particularly sweet syndrome, can be observed in patients with high AIGA titers, which supports possible autoimmune diseases (35). Therefore, immunotherapy, including corticosteroids, immunosuppressive drugs, and biological agents (rituximab), should be considered when the infection continues to worsen (6,7,(13)(14)(15)(16)(17)(18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30)(31). However, these treatment options also affect protective immunity and often lead to serious side effects.…”

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confidence: 99%

Pathogen spectrum and immunotherapy in patients with anti-IFN-γ autoantibodies: A multicenter retrospective study and systematic review

Qiu

Fang

et al. 2022

Front. Immunol.

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BackgroundAnti-interferon-γ autoantibody (AIGA) positivity is an emerging immunodeficiency syndrome closely associated with intracellular infection in individuals without human immunodeficiency virus (HIV). However, the information on epidemiology, pathogen spectrum, and immunotherapy among these patients lack a systematic description of large data.MethodsThis systematic literature review and multicenter retrospective study aimed to describe the pathogen spectrum and review treatment strategies among patients with AIGA positivity.ResultsWe included 810 HIV-negative patients with AIGA positivity infected with one or more intracellular pathogens. Excluding four teenagers, all the patients were adults. The most common pathogen was nontuberculous mycobacteria (NTM) (676/810, 83.5%). A total of 765 NTM isolates were identified in 676 patients with NTM, including 342 (44.7%) rapid-grower mycobacteria, 273 (35.7%) slow-grower mycobacteria, and 150 (19.6%) unidentified NTM subtype. Even with long-term and intensive antimicrobial treatments, 42.6% of patients with AIGA positivity had recurrence and/or persistent infection. Sixty-seven patients underwent immunoregulatory or immunosuppressive therapy, and most (60) achieved remission. The most common treatment strategy was rituximab (27/67, 40.3%) and cyclophosphamide (22/67, 32.8%), followed by cyclophosphamide combined with glucocorticoids (8/67, 11.9%).ConclusionsIntracellular pathogen was the most common infection in patients with AIGA positivity. The predominant infection phenotypes were NTM, varicella-zoster virus, Talaromyces marneffei, and Salmonella spp., with or without other opportunistic infections. AIGA immunotherapy, including rituximab or cyclophosphamide, has yielded good preliminary results in some cases.

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Methylprednisolone acetate/rifabutin/rituximab

2020

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Corneal Deposits in a Patient With Anti–Interferon-γ Autoantibody Syndrome

Cited by 3 publications

References 9 publications

Characteristics and Outcomes of Anti-interferon Gamma Antibody-Associated Adult Onset Immunodeficiency

Characteristics and Outcomes of Anti-interferon Gamma Antibody-Associated Adult Onset Immunodeficiency

Pathogen spectrum and immunotherapy in patients with anti-IFN-γ autoantibodies: A multicenter retrospective study and systematic review

Methylprednisolone acetate/rifabutin/rituximab

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