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An autopsy case report of adult-onset Krabbe disease: Comparison with an infantile-onset case.
Sasaki, Miu; Ebata, Miori; Tanei, Zen-Ichi; Oda, Yoshitaka; Hamauchi, Akiko; Tanikawa, Satoshi; Sugino, Hirokazu; Ishida, Yusuke; Abe, Takenori; Arai, Nobutaka; Sako, Kazuya; Tanaka, Shinya.
Afiliação
  • Sasaki M; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Ebata M; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Tanei ZI; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Oda Y; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Hamauchi A; Department of Neurology, Nakamura Memorial Hospital, Sapporo, Japan.
  • Tanikawa S; Department of Internal Medicine & Neurology, Takeuchi Clinic, Doushoukai Medical Corporation, Setouchi, Japan.
  • Sugino H; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Ishida Y; Institute for Chemical Reaction Design and Discovery (WPI-ICReDD), Hokkaido University, Sapporo, Japan.
  • Abe T; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Arai N; Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.
  • Sako K; Department of Neurology, Nakamura Memorial Hospital, Sapporo, Japan.
  • Tanaka S; Laboratory of Neuropathology, Tokyo Metropolitan Institute of Medical Science, Tokyo, Japan.
Pathol Int ; 72(11): 558-565, 2022 Nov.
Article em En | MEDLINE | ID: mdl-36200664
Krabbe disease is a lysosomal storage disease caused by a deficiency of the galactocerebrosidase (GALC) enzyme, which leads to demyelination of the central and peripheral nervous systems. Almost all patients with Krabbe disease are infants, and this is the first report of adult-onset cases that describe pathological findings. Here, we present two autopsy cases: a 73-year-old female and a 2-year-old male. The adult-onset case developed symptoms in her late thirties and was diagnosed by the identification of GALC D528N and L634S mutations and by T2-weighted magnetic resonance imaging; she had increased signal in the white matter along the pyramidal tract to the bilateral precentral gyrus, as well as from the triangular part to the posterior horn of the lateral ventricle. Microscopically, Klüver-Barrera staining was pale in the white matter of the precentral gyrus and occipito-thalamic radiation, and a few globoid cells were observed. The GALC mutations that were identified in the present adult-onset case do not completely inactivate GALC enzyme activity, resulting in focal demyelination of the brain.
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Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Leucodistrofia de Células Globoides Tipo de estudo: Diagnostic_studies / Prognostic_studies Limite: Adult / Aged / Child, preschool / Female / Humans / Infant / Male Idioma: En Revista: Pathol Int Assunto da revista: PATOLOGIA Ano de publicação: 2022 Tipo de documento: Article País de afiliação: Japão

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Leucodistrofia de Células Globoides Tipo de estudo: Diagnostic_studies / Prognostic_studies Limite: Adult / Aged / Child, preschool / Female / Humans / Infant / Male Idioma: En Revista: Pathol Int Assunto da revista: PATOLOGIA Ano de publicação: 2022 Tipo de documento: Article País de afiliação: Japão