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Invasive infection with Trichosporon inkin in 2 siblings with chronic granulomatous disease

Authors :
Sarah M. Wynne
Yvonne R. Shea
Steven M. Holland
Ashok Varma
Petrizia Lupo
Kyung J. Kwon-Chung
Armando C. Filie
Source :
Journal of Allergy and Clinical Immunology. 114:1418-1424
Publication Year :
2004
Publisher :
Elsevier BV, 2004.

Abstract

A 9-year-old girl with autosomal recessive chronic granulomatous disease (CGD) presented with asymptomatic bilateral pulmonary infiltrates on routine computed tomography. Fine-needle aspirate of the infiltrates was obtained and showed fungal cells resembling Trichosporon inkin . The specimen grew in culture, and testing by means of both API 20C and PCR amplification confirmed the diagnosis of T inkin . The infiltrates increased in size, despite sequential therapy with voriconazole, liposomal amphotericin B, caspofungin, and posaconazole. The patient required resection of the infected lung tissue, after which she recovered completely. While she was undergoing therapy, her 13-year-old brother, also with CGD, was given a diagnosis of bilateral T inkin –induced pulmonary infection. He also required bilateral pulmonary resection for cure. These cases demonstrate the predisposition of patients with CGD to have invasive infections with unusual fungal organisms, such as T inkin . They also illustrate the difficulty of treating invasive T inkin infections with antifungal agents alone. There are 9 previously reported cases of invasive infections caused by T inkin , 3 of which are in patients with CGD. All patients required removal of infected prosthetic devices or surgical resection of infected tissue for cure.

Details

ISSN :
00916749
Volume :
114
Database :
OpenAIRE
Journal :
Journal of Allergy and Clinical Immunology
Accession number :
edsair.doi.dedup.....60e82b64b0b6fcd1b010ab7b040b882c
Full Text :
https://doi.org/10.1016/j.jaci.2004.07.066