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Endobronchial and Pulmonary Tuberculosis Associated With Selective Immonuglobulin A Deficiency FREE TO VIEW

Gilmar Zonzin, MD; Jaime Correia, MD; Mariana Silva; Marcelle Tavares; Juliana Silveira; Henrique Rossi
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Pneumology, Centro Universitário de Volta Redonda, Volta Redonda, Rio de Janeiro, Brazil

Chest. 2015;148(4_MeetingAbstracts):171A. doi:10.1378/chest.2265718
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SESSION TITLE: Tuberculosis Global Case Reports

SESSION TYPE: Global Case Report Poster

PRESENTED ON: Tuesday, October 27, 2015 at 01:30 PM - 02:30 PM

INTRODUCTION: Selective deficiency of immunoglobulin A (IgA) is the most common of the primary immunodeficiencies. It occurs in 1:500 live births approximately. Reduced IgA production, which remains in abnormally low levels, characterizes the disease. Usually, patients are asymptomatic, but some may develop infections in the upper and lower airways, chronic diarrhea and autoimmune phenomena. This work presents the case of a young woman with IgA deficiency, associated with the development of endobronchial and pulmonary tuberculosis.

CASE PRESENTATION: LRM, 25 years old, resident in Rio de Janeiro State, Brazil, seeks medical treatment in Pulmonology reporting recurrent respiratory symptoms that were presented about 40 days ago. Her main complaints were productive cough with purulent sputum, pleuritic chest pain on the right side, mild dyspnea and fever of 38°C (100.4°F). She had been diagnosed with pneumonia before and treated with levofloxacin 500 mg/day for 10 days, systemic corticosteroids and inhaled bronchodilators. Reported similar episodes since childhood, diagnosed as asthma crisis, sinusitis and recurrent pneumonia. She was in good general health condition, axillary temperature of 37.5°C (99.5°F), pulmonary auscultation with crackles in both lung bases. Blood pressure of 125/75mmHg, heart rate of 86 beats/min and respiratory rate of 22 breaths/min. The chest radiography showed bilateral bronchopneumonic infiltrates. Due to her well-being, home treatment was chosen and clarithromycin 500mg/day was prescribed. At this moment, several additional tests were requested: chest computed tomography (CT), complete blood count, inflammatory markers, serologies, autoantibodies, serum immunoglobulin, HIV test, tuberculin skin test (TST), among others. CT scan revealed peribronchovascular thickening with mucoid impaction and the tree-in-bud sign with bilateral small centrilobular nodules, some of them confluents. Laboratory results negative for specific infections (inluding sputum smear for acid-fast bacilli (AFB) and TST). IgA levels came below 7mg/dL, characterizing IgA deficiency. However, pulmonary infiltrates progressed. Bronchoscopy was performed and showed bronchial mucosa edema with pronounced hyperemia and granulation suggestive of endobronchial tuberculosis (EBTB). Bronchoalveolar lavage (BAL) negative for AFB, histopathological finds were compatible with EBTB and positive culture for Mycobacterium tuberculosae (MTB), defining diagnosis of pulmonary tuberculosis (PT) and EBTB. With specific treatment addressed, patient evolved positively to clinical cure and complete radiological resolution.

DISCUSSION: IgA deficiency is not commonly associated with tuberculosis. There are few reports about it in literature, however this form of immunodeficiency facilitates the occurrence of respiratory diseases. In the present case, the first imaging studies were striking because of the significant pulmonary involvement along the relatively mild clinic the patient had. The sputum test came negative for tuberculosis as well as the TST, but the IgA deficit identified with no clinical and imaginologic improvement led to bronchoscopy request, where the histopathological findings and the culture of BAL set the diagnosis of PT and EBTB. Probably, the diagnosis has been hampered by the previous use of quinolones and the low sensitivity of sputum examination due to paucibacillary condition.

CONCLUSIONS: The endobronchial form of tuberculosis is not frequent in adults and is most commonly related to primary pulmonary form as encountered in children. We did not find data in literature suggesting that IgA deficiency may predispose neither PT nor EBTB. The clinical and imaginologic evaluation led to the request of bronchoscopy whose findings were consistent with edematous, hyperemic and granular type EBTB, diagnosis confirmed microbiologically and histopathologicaly. The treatment is similar for both EBTB and PT, which is done with specific antibiotic therapy and periodic monitoring to evaluate treatment response.

Reference #1: Jorgensen GH et al,Clinical symptoms in adults with selective IgA deficiency: a case-control study,J Clin Immunol.2013 May;33(4):742-7.doi: 10.1007/s10875-012-9858-x.Epub 2013 Feb 7.

Reference #2: Singh K, et al, IgA deficiency and autoimmunity.Autoimmun Rev.2014 Feb;13(2):163-77. doi:10.1016/j.autrev.2013.10.005.Epub 2013 Oct 21.

Reference #3: Solhpour A.et al.Subphrenic abscess and focal recurring lesions due to tuberculosis in a patient with IgA deficiency.Journal of Infection,Vol. 54, no.1, p. e9-e12,2007.

DISCLOSURE: The following authors have nothing to disclose: Gilmar Zonzin, Jaime Correia, Mariana Silva, Marcelle Tavares, Juliana Silveira, Henrique Rossi

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