Author:
Akagi Kazumasa,Yamamoto Kazuko,Umemura Asuka,Ide Shotaro,Hirayama Tatsuro,Takazono Takahiro,Imamura Yoshifumi,Miyazaki Taiga,Sakamoto Noriho,Shiraishi Hirokazu,Takahata Hideaki,Zaizen Yoshiaki,Fukuoka Junya,Morikawa Minoru,Ashizawa Kazuto,Teruya Katsuji,Izumikawa Koichi,Mukae Hiroshi
Abstract
Abstract
Background
Vacuolar encephalomyelopathy, a disregarded diagnosis lately, was a major neurological disease in the terminal stages of human immunodeficiency virus (HIV)-1 infection in the pre-antiretroviral therapy (ART) era. Granulomatous-lymphocytic interstitial lung disease (GLILD) was classically identified as a non-infectious complication of common variable immunodeficiency; however, it is now being recognized in other immunodeficiency disorders. Here, we report the first case of GLILD accompanied by vacuolar encephalomyelopathy in a newly diagnosed HIV-infected man.
Case presentation
A 40-year-old Japanese man presented with chronic dry cough and progressing paraplegia. Radiological examination revealed diffuse pulmonary abnormalities in bilateral lungs, focal demyelinating lesions of the spinal cord, and white matter lesions in the brain. He was diagnosed with GLILD based on marked lymphocytosis detecting in bronchoalveolar lavage, and transbronchial-biopsy proven T-cellular interstitial lung disease with granulomas. Microbiological examinations did not reveal an etiologic agent. The patient was also diagnosed with HIV-associated vacuolar encephalomyelopathy on the basis of an elevated HIV viral load in cerebrospinal fluid. After initiating ART, the brain lesions and paraplegia improved significantly, and interstitial abnormalities of the lungs and cough disappeared.
Conclusion
This report highlights that even in the post-ART era in developed countries with advanced healthcare services, HIV-associated vacuolar encephalomyelopathy should be considered in the differential diagnosis of a progressive neurological disorder during the first visit. Furthermore, GLILD may represent an HIV-associated pulmonary manifestation that can be treated by ART.
Publisher
Springer Science and Business Media LLC
Subject
Pharmacology (medical),Virology,Molecular Medicine
Cited by
2 articles.
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