TY - JOUR
T1 - The SCA17 phenotype can include features of MSA-C, PSP and cognitive impairment
AU - Lin, I. Sheng
AU - Wu, Ruey Meei
AU - Lee-Chen, Guey Jen
AU - Shan, Din E.
AU - Gwinn-Hardy, Katrina
PY - 2007/5
Y1 - 2007/5
N2 - Spinocerebellar ataxia (SCA) 17 is a dominant neurodegenerative disorder characterized by ataxia, cognitive decline, dystonia, and parkinsonism. The disease is caused by unstable cytosine-adenine-guanine (CAG) trinucleotide expansion mutation coding for polyglutamine tracts in the TATA box-binding protein (TBP), a general transcription initiation factor. Herein, we report a SCA17 case with a phenotype not previously reported, which consisted of progressive ataxia, autonomic dysfunction, parkinsonism, supranuclear palsy and cognitive impairment. Cerebrospinal fluid study and 18F-dopa PET scanning demonstrated dopamine deficiency and nigrostrital degeneration. This case expands the current phenotype associated with SCA17. SCA17 should be considered in the differential diagnosis of cases resembling multiple system atrophy, especially those with atypical features.
AB - Spinocerebellar ataxia (SCA) 17 is a dominant neurodegenerative disorder characterized by ataxia, cognitive decline, dystonia, and parkinsonism. The disease is caused by unstable cytosine-adenine-guanine (CAG) trinucleotide expansion mutation coding for polyglutamine tracts in the TATA box-binding protein (TBP), a general transcription initiation factor. Herein, we report a SCA17 case with a phenotype not previously reported, which consisted of progressive ataxia, autonomic dysfunction, parkinsonism, supranuclear palsy and cognitive impairment. Cerebrospinal fluid study and 18F-dopa PET scanning demonstrated dopamine deficiency and nigrostrital degeneration. This case expands the current phenotype associated with SCA17. SCA17 should be considered in the differential diagnosis of cases resembling multiple system atrophy, especially those with atypical features.
KW - MSA
KW - PSP
KW - SCA17
KW - TATA box-binding protein (TBP)
KW - Taiwanese
UR - http://www.scopus.com/inward/record.url?scp=34247117930&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=34247117930&partnerID=8YFLogxK
U2 - 10.1016/j.parkreldis.2006.04.009
DO - 10.1016/j.parkreldis.2006.04.009
M3 - Article
C2 - 16793320
AN - SCOPUS:34247117930
SN - 1353-8020
VL - 13
SP - 246
EP - 249
JO - Parkinsonism and Related Disorders
JF - Parkinsonism and Related Disorders
IS - 4
ER -