Triple mosaic variants of PURA in a patient with multiple congenital anomalies

被引:0
|
作者
Fujita, Atsushi [1 ]
Suenaga, Yuta [2 ]
Takeshita, Eri [2 ]
Takahashi, Yuji [3 ]
Suzuki, Yuichi [4 ]
Ohori, Sachiko [1 ]
Tsuchida, Naomi [1 ,5 ]
Uchiyama, Yuri [1 ,5 ]
Koshimizu, Eriko [1 ]
Miyatake, Satoko [1 ,6 ]
Mizuguchi, Takeshi [1 ]
Matsumoto, Naomichi [1 ,5 ,6 ]
机构
[1] Yokohama City Univ, Grad Sch Med, Dept Human Genet, Yokohama, Kanagawa, Japan
[2] Natl Ctr Hosp, Natl Ctr Neurol & Psychiat, Dept Child Neurol, Kodaira, Tokyo, Japan
[3] Natl Ctr Hosp, Natl Ctr Neurol & Psychiat, Dept Neurol, Kodaira, Tokyo, Japan
[4] Fukushima Med Univ, Sch Med, Dept Pediat, Fukushima, Japan
[5] Yokohama City Univ Med, Dept Rare Dis Genom, Yokohama, Kanagawa, Japan
[6] Yokohama City Univ Med, Dept Clin Genet, Yokohama, Kanagawa, Japan
基金
日本学术振兴会;
关键词
MUTATIONS; ALPHA;
D O I
10.1038/s10038-024-01315-9
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
In monogenic diseases, double mosaic variants of the same gene have rarely been identified. Here, we report the case of triple mosaic variants in PURA, a gene responsible for a neurodevelopmental syndrome (OMIM# 616158). Whole-exome sequencing identified three somatic PURA variants in our case with a similar neurodevelopmental syndrome: NM_005859.5: c.222C>A p.(Tyr74*), c.224T>A p.(Leu75Gln), and c.233A>G p.(Lys78Arg). The two missense variants were on the same sequence read, but the nonsense variant was not. To determine the origin of the alleles, we performed long-read sequencing because of the absence of informative SNPs near the somatic variants. Long-read sequencing revealed that these three somatic variants are derived from the same chromosome. The exact mechanism behind their occurrence is unclear, but the nonsense variant could have occurred de novo as a germline event and incomplete post-zygotic rescue for the germline variant could have led to the two missense variants.
引用
收藏
页码:227 / 230
页数:4
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