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Clinical manifestations, course, and outcome of patients with neutralizing anti-interferon- γ autoantibodies and disseminated nontuberculous mycobacterial infections

View Article: PubMed Central - PubMed

ABSTRACT

Neutralizing anti-interferon-γ autoantibody (nAIGA)-associated immunodeficiency is an emerging medical issue worldwide. In the present study, we describe and discuss the clinical features and outcomes of patients with nAIGAs and disseminated infections by nontuberculous mycobacteria (dNTM).

We thoroughly reviewed the medical records of all patients. Microorganisms and nAIGAs were identified using previously described methods with modifications. All data were calculated and analyzed using SPSS software.

Among 46 adult patients with dNTM infections, we identified 45 cases (97.8%) with nAIGAs. The average patient age was 58.6 years, and there was no sex predominance. Cervical lymphadenitis (81.8%) was the most common clinical manifestation. Endocrine disorder was the leading comorbidity (7 cases). Malignancies were found in 4 patients, and all of the malignancies originated from the T-cell/macrophage lineage. More than half of the identifiable isolates were slow-growing NTMs. Twenty-eight (62.2%) and 18 (40.0%) patients had a history of zoster and salmonellosis, respectively. A high proportion of patients with recurrent episodes of NTM infection or a history of zoster and dNTM infection had initial nAIGA titers ≥10–5 dilution (P < 0.05). Twenty-seven patients (60.0%) required long-term antimycobacterial therapy and had at least 1 episode of recurrent NTM disease. No mortality was related to dNTM infection.

In Taiwan, nAIGAs are a recently recognized mechanism of dNTM infection. Long term of antibiotic treatment and adherence to medical advice are necessary to improve the clinical outcome of patients with nAIGAs.

No MeSH data available.


Related in: MedlinePlus

A, Number of patients with different titers of nAIGA. B, Patients with NTM infection and a history of HZ had a significantly higher initial nAIGA titer (≥10–5 dilution) than those with NTM infection alone. C, Patients with recurrent NTM infections, particularly those with ≥5 episodes, had a higher initial nAIGA titer. D, Patients with persistent infection tended to have a higher initial nAIGA titer (≥10–5 dilution). HZ = herpes zoster, nAIGA = neutralizing anti-interferon-γ autoantibodies, NTM = nontuberculous mycobacteria, Sal = salmonellosis. ∗P < 0.05.
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Figure 2: A, Number of patients with different titers of nAIGA. B, Patients with NTM infection and a history of HZ had a significantly higher initial nAIGA titer (≥10–5 dilution) than those with NTM infection alone. C, Patients with recurrent NTM infections, particularly those with ≥5 episodes, had a higher initial nAIGA titer. D, Patients with persistent infection tended to have a higher initial nAIGA titer (≥10–5 dilution). HZ = herpes zoster, nAIGA = neutralizing anti-interferon-γ autoantibodies, NTM = nontuberculous mycobacteria, Sal = salmonellosis. ∗P < 0.05.

Mentions: The distribution of the initial nAIGA titers according to the types of infections, the recurrence frequency of NTM infections, and the patient outcomes is shown in Fig. 1. Most cases (95.6%) had an initial nAIGA titer ≥10–4 dilution (Fig. 2A). In terms of pathogens and histories of infection, a significantly higher proportion of patients with a history of HZ and dNTM infection had an initial nAIGA titer ≥10–5 dilution compared with the proportion of those with dNTM infection alone (Fig. 2B; P < 0.05). A higher proportion of patients with recurrent episodes, particularly those with >5 episodes, had an initial nAIGA titer ≥10–5 dilution (Fig. 2C). Patients with persistent NTM infection tended to have higher initial nAIGA titers (≥10–5 dilution) than the cured cases, but the difference was not significant (Fig. 2D). In brief, a high proportion of patients with recurrent episodes of NTM infection or a history of HZ and dNTM infection had initial nAIGA titers ≥10–5 dilution.


Clinical manifestations, course, and outcome of patients with neutralizing anti-interferon- γ autoantibodies and disseminated nontuberculous mycobacterial infections
A, Number of patients with different titers of nAIGA. B, Patients with NTM infection and a history of HZ had a significantly higher initial nAIGA titer (≥10–5 dilution) than those with NTM infection alone. C, Patients with recurrent NTM infections, particularly those with ≥5 episodes, had a higher initial nAIGA titer. D, Patients with persistent infection tended to have a higher initial nAIGA titer (≥10–5 dilution). HZ = herpes zoster, nAIGA = neutralizing anti-interferon-γ autoantibodies, NTM = nontuberculous mycobacteria, Sal = salmonellosis. ∗P < 0.05.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4998320&req=5

Figure 2: A, Number of patients with different titers of nAIGA. B, Patients with NTM infection and a history of HZ had a significantly higher initial nAIGA titer (≥10–5 dilution) than those with NTM infection alone. C, Patients with recurrent NTM infections, particularly those with ≥5 episodes, had a higher initial nAIGA titer. D, Patients with persistent infection tended to have a higher initial nAIGA titer (≥10–5 dilution). HZ = herpes zoster, nAIGA = neutralizing anti-interferon-γ autoantibodies, NTM = nontuberculous mycobacteria, Sal = salmonellosis. ∗P < 0.05.
Mentions: The distribution of the initial nAIGA titers according to the types of infections, the recurrence frequency of NTM infections, and the patient outcomes is shown in Fig. 1. Most cases (95.6%) had an initial nAIGA titer ≥10–4 dilution (Fig. 2A). In terms of pathogens and histories of infection, a significantly higher proportion of patients with a history of HZ and dNTM infection had an initial nAIGA titer ≥10–5 dilution compared with the proportion of those with dNTM infection alone (Fig. 2B; P < 0.05). A higher proportion of patients with recurrent episodes, particularly those with >5 episodes, had an initial nAIGA titer ≥10–5 dilution (Fig. 2C). Patients with persistent NTM infection tended to have higher initial nAIGA titers (≥10–5 dilution) than the cured cases, but the difference was not significant (Fig. 2D). In brief, a high proportion of patients with recurrent episodes of NTM infection or a history of HZ and dNTM infection had initial nAIGA titers ≥10–5 dilution.

View Article: PubMed Central - PubMed

ABSTRACT

Neutralizing anti-interferon-&gamma; autoantibody (nAIGA)-associated immunodeficiency is an emerging medical issue worldwide. In the present study, we describe and discuss the clinical features and outcomes of patients with nAIGAs and disseminated infections by nontuberculous mycobacteria (dNTM).

We thoroughly reviewed the medical records of all patients. Microorganisms and nAIGAs were identified using previously described methods with modifications. All data were calculated and analyzed using SPSS software.

Among 46 adult patients with dNTM infections, we identified 45 cases (97.8%) with nAIGAs. The average patient age was 58.6 years, and there was no sex predominance. Cervical lymphadenitis (81.8%) was the most common clinical manifestation. Endocrine disorder was the leading comorbidity (7 cases). Malignancies were found in 4 patients, and all of the malignancies originated from the T-cell/macrophage lineage. More than half of the identifiable isolates were slow-growing NTMs. Twenty-eight (62.2%) and 18 (40.0%) patients had a history of zoster and salmonellosis, respectively. A high proportion of patients with recurrent episodes of NTM infection or a history of zoster and dNTM infection had initial nAIGA titers &ge;10&ndash;5 dilution (P&#8202;&lt;&#8202;0.05). Twenty-seven patients (60.0%) required long-term antimycobacterial therapy and had at least 1 episode of recurrent NTM disease. No mortality was related to dNTM infection.

In Taiwan, nAIGAs are a recently recognized mechanism of dNTM infection. Long term of antibiotic treatment and adherence to medical advice are necessary to improve the clinical outcome of patients with nAIGAs.

No MeSH data available.


Related in: MedlinePlus