Zhao Qin,Jiang Liping,Yu Jie,Xiao Jianwen,Zhao Xiaodong.Genotype and phenotype correlation of Wiskott-Aldrich syndrome: a report based on 24 Chinese patients[J].J Amry Med Univ (J Third Mil Med Univ),2011,33(13):1404-1407.
Zhao Qin,Jiang Liping,Yu Jie,Xiao Jianwen,Zhao Xiaodong.Genotype and phenotype correlation of Wiskott-Aldrich syndrome: a report based on 24 Chinese patients[J].J Amry Med Univ (J Third Mil Med Univ),2011,33(13):1404-1407.DOI:
Genotype and phenotype correlation of Wiskott-Aldrich syndrome: a report based on 24 Chinese patients
Objective To study genotype-phenotype correlation preliminarily in 24 Chinese Wiskott-Aldrich syndrome (WAS) patients. Methods Clinical data of 24 Chinese male WAS patients from 21 unrelated families were collected
then patients’ phenotypes were scored according to international scoring standards. Flow cytometry was used to analyze WAS protein (WASP) expression in peripheral blood mononuclear cells (PBMCs). WASP gene was directly sequenced
then the genotype-phenotype correlation was analyzed. Results Among the 24 patients
2 of them were X-linked thrombocytopenia (XLT)with scores 2
and the others were classic WAS with scores 3 to 5. WASP of 1 patient was of normal expression
that of 2 was of reduced expression
and that of the others was of negative expression. We had found 7 missenses
4 nonsenses
6 deletions
and 7 splice site mutations. Among 21 detected unique mutations
there were 3 novel mutations
that is
180 CT (A49V)
342-343del CA (S103fsX121) and IVS7+2 TC. There were 6 hot spot mutations were reported which were 291 GA(R86H)
291GT(R86L)
1035del G(G334fsX444)
665 CT(R211X)
IVS6+5 GA and IVS8+1 GA. Two of all missense mutation patients were XLT
the others were classic WAS. All nonsense
deletion and splice site mutations patients were classic WAS
some of whose phenotype were severe. Conclusion Mutations in downstream of WASP gene
nonsense
deletion and splice site mutations probably lead to negative expression of WASP
unstable or trancated WASP production
with typical clinical manifestations of WAS. What’s more