Edina H Moylett1, Javier Chinen, William T Shearer. 1. Section of Allergy and Immunology, Department of Pediatrics, Baylor College of Medicine and Texas Children's Hospital, 6621 Fannin Street, MC:FC330.01, Houston, TX 77030, USA.
Abstract
BACKGROUND: Chronic granulomatous disease is a genetically determined primary immunodeficiency disease in which phagocytic cells are unable to kill certain bacteria and fungi after ingestion. Manifestations include recurrent pyogenic infections caused by catalase-positive microbes. Trichosporon species are emerging as opportunistic agents that cause systemic disease in immunocompromised patients. Typically disease has been described in association with T beigelii in patients with secondary immunodeficiency, such as underlying malignancy. OBJECTIVE: The objective was to report the first 2 cases of T pullulans infection in 2 male children with chronic granulomatous disease. METHODS: The records of the 2 patients were reviewed. In addition, all cases of T pullulans infection reported in the English language literature are presented. RESULTS: This report brings to 7 the total number of cases of T pullulans reported and the first in patients with chronic granulomatous disease, one with invasive pneumonia and the other with an infected paronychium and localized cellulitis. In the 5 additional cases malignancy was the principal risk factor. CONCLUSION: T pullulans has rarely been reported as a fungal pathogen. The most prominent risk factor for the development of trichosporonosis is immunocompromise, most notably with neutropenia. Abnormally functioning neutrophils, such as with chronic granulomatous disease, may also predispose individuals to this opportunistic pathogen.
BACKGROUND:Chronic granulomatous disease is a genetically determined primary immunodeficiency disease in which phagocytic cells are unable to kill certain bacteria and fungi after ingestion. Manifestations include recurrent pyogenic infections caused by catalase-positive microbes. Trichosporon species are emerging as opportunistic agents that cause systemic disease in immunocompromised patients. Typically disease has been described in association with T beigelii in patients with secondary immunodeficiency, such as underlying malignancy. OBJECTIVE: The objective was to report the first 2 cases of T pullulans infection in 2 male children with chronic granulomatous disease. METHODS: The records of the 2 patients were reviewed. In addition, all cases of T pullulans infection reported in the English language literature are presented. RESULTS: This report brings to 7 the total number of cases of T pullulans reported and the first in patients with chronic granulomatous disease, one with invasive pneumonia and the other with an infected paronychium and localized cellulitis. In the 5 additional cases malignancy was the principal risk factor. CONCLUSION: T pullulans has rarely been reported as a fungal pathogen. The most prominent risk factor for the development of trichosporonosis is immunocompromise, most notably with neutropenia. Abnormally functioning neutrophils, such as with chronic granulomatous disease, may also predispose individuals to this opportunistic pathogen.
Authors: Patrick W Hickey; Deanna A Sutton; Annette W Fothergill; Michael G Rinaldi; Brian L Wickes; Howard J Schmidt; Thomas J Walsh Journal: J Clin Microbiol Date: 2009-08-05 Impact factor: 5.948
Authors: Cláudio Duarte-Oliveira; Fernando Rodrigues; Samuel M Gonçalves; Gustavo H Goldman; Agostinho Carvalho; Cristina Cunha Journal: Front Cell Infect Microbiol Date: 2017-04-07 Impact factor: 5.293