Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report
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 cla...
| Main Authors: | , , , , , , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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BMC
2020-07-01
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| Series: | AIDS Research and Therapy |
| Subjects: | |
| Online Access: | http://link.springer.com/article/10.1186/s12981-020-00295-y |
| _version_ | 1819229018634846208 |
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| author | Kazumasa Akagi Kazuko Yamamoto Asuka Umemura Shotaro Ide Tatsuro Hirayama Takahiro Takazono Yoshifumi Imamura Taiga Miyazaki Noriho Sakamoto Hirokazu Shiraishi Hideaki Takahata Yoshiaki Zaizen Junya Fukuoka Minoru Morikawa Kazuto Ashizawa Katsuji Teruya Koichi Izumikawa Hiroshi Mukae |
| author_facet | Kazumasa Akagi Kazuko Yamamoto Asuka Umemura Shotaro Ide Tatsuro Hirayama Takahiro Takazono Yoshifumi Imamura Taiga Miyazaki Noriho Sakamoto Hirokazu Shiraishi Hideaki Takahata Yoshiaki Zaizen Junya Fukuoka Minoru Morikawa Kazuto Ashizawa Katsuji Teruya Koichi Izumikawa Hiroshi Mukae |
| author_sort | Kazumasa Akagi |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-12-23T11:06:30Z |
| format | Article |
| id | doaj.art-f55667d5b8a4451ea043a417a5e95ded |
| institution | Directory Open Access Journal |
| issn | 1742-6405 |
| language | English |
| last_indexed | 2024-12-23T11:06:30Z |
| publishDate | 2020-07-01 |
| publisher | BMC |
| record_format | Article |
| series | AIDS Research and Therapy |
| spelling | doaj.art-f55667d5b8a4451ea043a417a5e95ded2022-12-21T17:49:28ZengBMCAIDS Research and Therapy1742-64052020-07-011711710.1186/s12981-020-00295-yHuman immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case reportKazumasa Akagi0Kazuko Yamamoto1Asuka Umemura2Shotaro Ide3Tatsuro Hirayama4Takahiro Takazono5Yoshifumi Imamura6Taiga Miyazaki7Noriho Sakamoto8Hirokazu Shiraishi9Hideaki Takahata10Yoshiaki Zaizen11Junya Fukuoka12Minoru Morikawa13Kazuto Ashizawa14Katsuji Teruya15Koichi Izumikawa16Hiroshi Mukae17Department of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalDepartment of Neurology and Strokology, Nagasaki University HospitalDepartment of Rehabilitation Medicine, Nagasaki University HospitalDepartment of Pathology, Nagasaki University HospitalDepartment of Pathology, Nagasaki University HospitalDepartment of Radiology, Nagasaki University HospitalDepartment of Clinical Oncology, Nagasaki University Graduate School of Biomedical SciencesAIDS Clinical Center, National Center for Global Health and MedicineInfection Control and Education Center, Nagasaki University HospitalDepartment of Respiratory Medicine, Nagasaki University HospitalAbstract 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.http://link.springer.com/article/10.1186/s12981-020-00295-yHIVAIDSEncephalopathyVacuolar myelopathyGranulomatous-lymphocytic interstitial lung diseaseAntiretroviral therapy |
| spellingShingle | Kazumasa Akagi Kazuko Yamamoto Asuka Umemura Shotaro Ide Tatsuro Hirayama Takahiro Takazono Yoshifumi Imamura Taiga Miyazaki Noriho Sakamoto Hirokazu Shiraishi Hideaki Takahata Yoshiaki Zaizen Junya Fukuoka Minoru Morikawa Kazuto Ashizawa Katsuji Teruya Koichi Izumikawa Hiroshi Mukae Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report AIDS Research and Therapy HIV AIDS Encephalopathy Vacuolar myelopathy Granulomatous-lymphocytic interstitial lung disease Antiretroviral therapy |
| title | Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report |
| title_full | Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report |
| title_fullStr | Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report |
| title_full_unstemmed | Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report |
| title_short | Human immunodeficiency virus-associated vacuolar encephalomyelopathy with granulomatous-lymphocytic interstitial lung disease improved after antiretroviral therapy: a case report |
| title_sort | human immunodeficiency virus associated vacuolar encephalomyelopathy with granulomatous lymphocytic interstitial lung disease improved after antiretroviral therapy a case report |
| topic | HIV AIDS Encephalopathy Vacuolar myelopathy Granulomatous-lymphocytic interstitial lung disease Antiretroviral therapy |
| url | http://link.springer.com/article/10.1186/s12981-020-00295-y |
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