TY - JOUR
T1 - Mutation spectrum of and founder effects affecting the PTS gene in East Asian populations
AU - Chiu, Yen Hui
AU - Chang, Ying Chen
AU - Chang, Yu Hsin
AU - Niu, Dau Ming
AU - Yang, Yan Ling
AU - Ye, Jun
AU - Jiang, Jianhui
AU - Okano, Yoshiyuki
AU - Lee, Dong Hwan
AU - Pangkanon, Suthipong
AU - Kuptanon, Chulaluck
AU - Hock, Ngu Lock
AU - Chiong, Mary Anne
AU - Cavan, Barbra V.
AU - Hsiao, Kwang Jen
AU - Liu, Tze Tze
PY - 2012/2
Y1 - 2012/2
N2 - The enzyme 6-pyruvoyl-tetrahydropterin synthase (PTPS, gene symbol: PTS) is involved in the second step of the de novo biosynthesis of tetrahydrobiopterin (BH4), which is a vital cofactor of nitric oxide synthases and three types of aromatic amino acid hydroxylases; the latter are important enzymes in the production of neurotransmitters. We conducted a study of PTS mutations in East Asia, including Taiwan, Mainland China, Japan, South Korea, the Philippines, Thailand and Malaysia. A total of 43 mutations were identified, comprising 22 previously reported mutations and 21 new discovered mutations. Among these, the c.155A>G, c.259C>T, c. 272A>G, c.286G>A and c.84-291A>G mutations were the most common PTS mutations in East Asia, while the c.58T>C and c.243G>A mutations were, respectively, specific to Filipinos and Japanese originating from Okinawa. Further studies demonstrated that each of the mutations listed above was in linkage disequilibrium to a specific allele of polymorphic microsatellite marker, D11S1347. These results suggest the presence of founder effects that have affected these frequent mutations in East Asia populations. In this context, D11S1347 should become one of the most reliable polymorphic markers for use in prenatal diagnosis among PTPS deficient families, especially where mutations are yet to be identified.
AB - The enzyme 6-pyruvoyl-tetrahydropterin synthase (PTPS, gene symbol: PTS) is involved in the second step of the de novo biosynthesis of tetrahydrobiopterin (BH4), which is a vital cofactor of nitric oxide synthases and three types of aromatic amino acid hydroxylases; the latter are important enzymes in the production of neurotransmitters. We conducted a study of PTS mutations in East Asia, including Taiwan, Mainland China, Japan, South Korea, the Philippines, Thailand and Malaysia. A total of 43 mutations were identified, comprising 22 previously reported mutations and 21 new discovered mutations. Among these, the c.155A>G, c.259C>T, c. 272A>G, c.286G>A and c.84-291A>G mutations were the most common PTS mutations in East Asia, while the c.58T>C and c.243G>A mutations were, respectively, specific to Filipinos and Japanese originating from Okinawa. Further studies demonstrated that each of the mutations listed above was in linkage disequilibrium to a specific allele of polymorphic microsatellite marker, D11S1347. These results suggest the presence of founder effects that have affected these frequent mutations in East Asia populations. In this context, D11S1347 should become one of the most reliable polymorphic markers for use in prenatal diagnosis among PTPS deficient families, especially where mutations are yet to be identified.
KW - HPA
KW - PTPS deficiency
KW - founder effect
KW - microsatellite marker
KW - mutation
UR - http://www.scopus.com/inward/record.url?scp=84863229713&partnerID=8YFLogxK
U2 - 10.1038/jhg.2011.146
DO - 10.1038/jhg.2011.146
M3 - Article
C2 - 22237589
AN - SCOPUS:84863229713
SN - 1434-5161
VL - 57
SP - 145
EP - 152
JO - Journal of Human Genetics
JF - Journal of Human Genetics
IS - 2
ER -