TY - JOUR
T1 - Leukoencephalopathy resolution after atypical mycobacterial treatment
T2 - A case report
AU - Oliveira, Marcos C.B.
AU - Douglas Kazutoshi, Sato
AU - Soares-Neto, Herval R.
AU - Lucato, Leandro T.
AU - Callegaro, Dagoberto
AU - Nitrini, Ricardo
AU - Medeiros, Raphael S.S.
AU - Misu, Tatsuro
AU - Fujihara, Kazuo
AU - Castro, Luiz H.
N1 - Funding Information:
This study was partially supported by KAKENHI (22229008) of The Ministry of Education, Culture, Sports, Science and Technology (MEXT) of Japan, and by the Health and Labor Sciences Research Grant on Intractable Diseases (Neuroimmunological Diseases) from the Ministry of Health, Labor and Welfare of Japan. The funding sources had no role in the design, collection, analysis, or interpretation of the study, nor in the writing of the article or decision to submit.
Funding Information:
Dr. Oliveira has no conflicts of interest to disclose. Dr. Castro has no conflicts of interest to disclose. Dr. Sato is an associated editor of the Arquivos de Neuropsiquiatria (official journal of the Brazilian Academy of Neurology), receives scholarship from the Ministry of Education, Culture, Sports, Science and Technology (MEXT) of Japan, has received research support from Ichiro Kanehara Foundation (2011), and speaker honorarium from Novartis. Dr. Soares-Neto has no conflicts of interest to disclose. Dr.
Funding Information:
Lucato has no conflicts of interest to disclose. Dr. Callegaro has no conflicts of interest to disclose. Dr. Medeiros has no conflicts of interest to disclose. Prof. Misu has received speaker honoraria from Bayer Schering Pharma, Biogen Idec Japan, Mitsubishi Tanabe Pharma Corporation, Asahi Kasei Medical Co., and Astellas Pharma Inc. and has received research support from Bayer Schering Pharma, Biogen Idec Japan, Asahi Kasei Kuraray Medical Co., The Chemo-Sero-Therapeutic Research Institute, Teva Pharmaceutical K.K., Mitsubishi Tanabe Pharma Corporation, Teijin Pharma, and Grants-in-Aid for Scientific Research from the Ministry of Education, Science and Technology, and the Ministry of Health, Labor and Welfare of Japan. Prof. Fujihara serves on scientific advisory boards for Bayer Schering Pharma, Biogen Idec, Mitsubishi Tanabe Pharma Corporation, Novartis Pharma, Chugai Pharmaceutical, Ono Pharmaceutical, Nihon Pharmaceutical, Merck Serono, Alexion Pharmaceuticals, Medimmune and Medical Review; has received funding for travel and speaker honoraria from Bayer Schering Pharma, Biogen Idec, Eisai Inc., Mitsubishi Tanabe Pharma Corporation, Novartis Pharma, Astellas Pharma Inc., Takeda Pharmaceutical Company Limited, Asahi Kasei Medical Co., Daiichi Sankyo, and Nihon Pharmaceutical; serve as an editorial board member of Clinical and Experimental Neuroimmunology (2009-present) and a advisory board member of Sri Lanka journal of Neurology; has received research support from Bayer Schering Pharma, Biogen Idec Japan, Asahi Kasei Medical, The Chemo-SeroTherapeutic Research Institute, Teva Pharmaceutical, Mitsubishi Tanabe Pharma, Teijin Pharma, Chugai Pharmaceutical, Ono Pharmaceutical, Nihon Pharmaceutical, and Genzyme Japan; is funded as the secondary investigator (#22229008, 2010–2015) by the Grants-in-Aid for Scientific Research from the Ministry of Education, Science and Technology of Japan and as the secondary investigator by the Grants-in-Aid for Scientific Research from the Ministry of Health, Welfare and Labor of Japan (2010-present). Prof. Nitrini has no conflicts of interest to disclose.
Publisher Copyright:
© 2015 Oliveira et al.
PY - 2015/9/2
Y1 - 2015/9/2
N2 - Background: Association of leukoencephalopathy and atypical mycobacteriosis has been rarely reported. We present a case that is relevant for its unusual presentation and because it may shed further light on the pathogenic mechanisms underlying reversible encephalopathies. Case report: We report the case of a Hispanic 64-year-old woman with cognitive decline and extensive leukoencephalopathy. Magnetic resonance imaging revealed white-matter lesions with increased water diffusivity, without blood-brain-barrier disruption. Brain biopsy showed tissue rarefaction with vacuolation, mild inflammation, few reactive astrocytes and decreased aquaporin water-channel expression in the lesions. Six months later, she was diagnosed with atypical mycobacterial pulmonary infection. Brain lesions resolved after antimycobacterial treatment. Conclusion: We hypothesize leukoencephalopathic changes and vasogenic edema were associated with decreased aquaporin expression. Further studies should clarify if reversible leukoencephalopathy has a causal relationship with decreased aquaporin expression and atypical mycobacterial infection, and mechanisms underlying leukoencephalopathy resolution after antimycobacterial treatment. This article may contribute to the understanding of pathogenic mechanisms underlying magnetic resonance imaging subcortical lesions and edema, which remain incompletely understood.
AB - Background: Association of leukoencephalopathy and atypical mycobacteriosis has been rarely reported. We present a case that is relevant for its unusual presentation and because it may shed further light on the pathogenic mechanisms underlying reversible encephalopathies. Case report: We report the case of a Hispanic 64-year-old woman with cognitive decline and extensive leukoencephalopathy. Magnetic resonance imaging revealed white-matter lesions with increased water diffusivity, without blood-brain-barrier disruption. Brain biopsy showed tissue rarefaction with vacuolation, mild inflammation, few reactive astrocytes and decreased aquaporin water-channel expression in the lesions. Six months later, she was diagnosed with atypical mycobacterial pulmonary infection. Brain lesions resolved after antimycobacterial treatment. Conclusion: We hypothesize leukoencephalopathic changes and vasogenic edema were associated with decreased aquaporin expression. Further studies should clarify if reversible leukoencephalopathy has a causal relationship with decreased aquaporin expression and atypical mycobacterial infection, and mechanisms underlying leukoencephalopathy resolution after antimycobacterial treatment. This article may contribute to the understanding of pathogenic mechanisms underlying magnetic resonance imaging subcortical lesions and edema, which remain incompletely understood.
KW - Aquaporin water-channel
KW - Leukoencephalopathy
KW - Mycobacteriosis
KW - PRES
KW - Reversible encephalopathy syndrome
KW - Vasogenic edema
UR - http://www.scopus.com/inward/record.url?scp=84940499708&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84940499708&partnerID=8YFLogxK
U2 - 10.1186/s12883-015-0415-0
DO - 10.1186/s12883-015-0415-0
M3 - Article
C2 - 26329680
AN - SCOPUS:84940499708
VL - 15
JO - BMC Neurology
JF - BMC Neurology
SN - 1471-2377
IS - 1
M1 - 159
ER -