Literature DB >> 10467913

X-linked adrenoleukodystrophy: spinocerebellar variant.

E K Tan1, S H Lim, L L Chan, M C Wong, K P Tan.   

Abstract

The phenotypic variability in X-linked adrenoleukodystrophy (X-ALD) can be wide and varied. Rarely, it can present with clinical signs of spinocerebellar degeneration. There are very few reported cases of selective predominant white matter disease of the cerebellum in these patients. We report a patient with a rare variant of adult onset ALD who was previously diagnosed as spinocerebellar ataxia. He was a 24-year-old male who had delayed developmental milestones, developed signs of spinocerebellar degeneration (SCD) after 10 years of Addison's disease. Serial Magnetic Resonance Imaging (MRI), revealed cerebellar and pontine white matter disease but sparing the cerebral cortex and supratentorial white matter. His diagnosis of X-ALD was subsequently confirmed by the elevated serum very long chain fatty acids. This patient illustrates the unusual clinical presentation and imaging features of X-ALD and the importance of considering X-ALD in the clinical context of spinocerebellar degeneration. Early recognition of this rare variant would allow proper genetic counselling and institution of dietary therapy and/or bone marrow transplantation.

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Year:  1999        PMID: 10467913     DOI: 10.1016/s0303-8467(99)00028-1

Source DB:  PubMed          Journal:  Clin Neurol Neurosurg        ISSN: 0303-8467            Impact factor:   1.876


  5 in total

1.  Clinical and genetic aspects in twelve Korean patients with adrenomyeloneuropathy.

Authors:  Hyung Jun Park; Ha Young Shin; Hoon-Chul Kang; Byung-Ok Choi; Bum Chun Suh; Ho Jin Kim; Young-Chul Choi; Phil Hyu Lee; Seung Min Kim
Journal:  Yonsei Med J       Date:  2014-04-01       Impact factor: 2.759

2.  Adult-onset cerebello-brainstem dominant form of X-linked adrenoleukodystrophy presenting as multiple system atrophy: case report and literature review.

Authors:  Kotaro Ogaki; Shunsuke Koga; Naoya Aoki; Wenlang Lin; Kinuko Suzuki; Owen A Ross; Dennis W Dickson
Journal:  Neuropathology       Date:  2015-07-31       Impact factor: 1.906

3.  Isolated cerebellar variant of adrenoleukodystrophy with a de novo adenosine triphosphate-binding cassette D1 (ABCD1) gene mutation.

Authors:  Joon Won Kang; Sang Mi Lee; Kyo Yeon Koo; Young-Mock Lee; Hyo Suk Nam; Zhejiu Quan; Hoon-Chul Kang
Journal:  Yonsei Med J       Date:  2014-07       Impact factor: 2.759

4.  A case of adrenoleukodystrophy presenting as progressive cerebellar dysfunction.

Authors:  Seunguk Jung; Jong Won Chung; Ji Young Yun; Han-Joon Kim; Beom Seok Jeon
Journal:  J Mov Disord       Date:  2009-10-30

5.  Unmasking adrenoleukodystrophy in a cohort of cerebellar ataxia.

Authors:  Ying-Hao Chen; Yi-Chung Lee; Yu-Shuen Tsai; Yuh-Cherng Guo; Cheng-Tsung Hsiao; Pei-Chien Tsai; Jin-An Huang; Yi-Chu Liao; Bing-Wen Soong
Journal:  PLoS One       Date:  2017-05-08       Impact factor: 3.240

  5 in total

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