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
相关论文
共 50 条
  • [1] Pathogenic mosaic variants in congenital hypogonadotropic hypogonadism
    Acierno, James S.
    Xu, Cheng
    Papadakis, Georgios E.
    Niederlander, Nicolas J.
    Rademaker, Jesse D.
    Meylan, Jenny
    Messina, Andrea
    Kolesinska, Zofia
    Quinton, Richard
    Lang-Muritano, Mariarosaria
    Bartholdi, Deborah
    Halperin, Irene
    De Geyter, Christian
    Bouligand, Jerome
    Bartoloni, Lucia
    Young, Jacques
    Santoni, Federico A.
    Pitteloud, Nelly
    GENETICS IN MEDICINE, 2020, 22 (11) : 1759 - 1767
  • [2] Identification of de novo variants in KCTD10 as a proposed cause for multiple congenital anomalies
    Morrow, Michelle M.
    Torti, Erin
    McGivern, Bobbi
    Gates, Ryan
    Bekheirnia, Mir Reza
    Bekheirnia, Nasim
    Folk, Leandra
    Holtrop, Shannon
    Palculict, Timothy Blake
    Redlich, Olivia L.
    Reich, Adi
    Sacoto, Maria J. Guillen
    Shi, Lisong
    Wentzensen, Ingrid M.
    Mcwalter, Kirsty
    HUMAN GENETICS AND GENOMICS ADVANCES, 2025, 6 (03):
  • [3] Robust identification of mosaic variants in congenital heart disease
    Manheimer, Kathryn B.
    Richter, Felix
    Edelmann, Lisa J.
    D'Souza, Sunita L.
    Shi, Lisong
    Shen, Yufeng
    Homsy, Jason
    Boskovski, Marko T.
    Tai, Angela C.
    Gorham, Joshua
    Yasso, Christopher
    Goldmuntz, Elizabeth
    Brueckner, Martina
    Lifton, Richard P.
    Chung, Wendy K.
    Seidman, Christine E.
    Seidman, J. G.
    Gelb, Bruce D.
    HUMAN GENETICS, 2018, 137 (02) : 183 - 193
  • [4] New Syndrome of Congenital Circumferential Skin Folds Associated with Multiple Congenital Anomalies
    Basel-Vanagaite, Lina
    Sprecher, Eli
    Gat, Andrea
    Merlob, Paul
    Albin-Kaplanski, Adi
    Konen, Osnat
    Solomon, Benjamin D.
    Muenke, Maximilian
    Grzeschik, Karl H.
    Sirota, Lea
    PEDIATRIC DERMATOLOGY, 2012, 29 (01) : 89 - 95
  • [5] The challenges in the interpretation of genetic variants detected by genomics techniques in patients with congenital anomalies
    Vaseghi, Hajar
    Akrami, Seyed Mohammad
    Rashidi-Nezhad, Ali
    JOURNAL OF CLINICAL LABORATORY ANALYSIS, 2023, 37 (17-18)
  • [6] Two novel nebulin variants in an adult patient with congenital nemaline myopathy
    Guettsches, Anne K.
    Dekomien, Gabriele
    Claeys, Kristl G.
    von der Hagen, Maja
    Huebner, Angela
    Kley, Rudolf A.
    Kirschner, Janbernd
    Vorgerd, Matthias
    NEUROMUSCULAR DISORDERS, 2015, 25 (05) : 392 - 396
  • [7] Genotype-Phenotype Correlation of Congenital Anomalies in Multiple Congenital Anomalies Hypotonia Seizures Syndrome (MCAHS1)/PIGN-Related Epilepsy
    Fleming, Leah
    Lemmon, Monica
    Beck, Natalie
    Johnson, Maria
    Mu, Weiyi
    Murdock, David
    Bodurtha, Joann
    Hoover-Fong, Julie
    Cohn, Ronald
    Bosemani, Thangamadhan
    Baranano, Kristin
    Hamosh, Ada
    AMERICAN JOURNAL OF MEDICAL GENETICS PART A, 2016, 170 (01) : 77 - 86
  • [8] Multiple congenital anomalies in two fetuses with glutathione-synthetase deficit (GSS)
    Jury, Jeanne
    Benoist, Jean-Francois
    Joubert, Madeleine
    Quelin, Chloe
    Besnard, Thomas
    Conrad, Solene
    Le Vaillant, Claudine
    Bezieau, Stephane
    Isidor, Bertrand
    Attie-Bitach, Tania
    Cogne, Benjamin
    Vincent, Marie
    CLINICAL GENETICS, 2024, 106 (06) : 776 - 781
  • [9] Whole exome sequencing identifies multiple diagnoses in congenital glaucoma with systemic anomalies
    Reis, L. M.
    Tyler, R. C.
    Weh, E.
    Hendee, K. E.
    Schilter, K. F.
    Phillips, J. A., III
    Sequeira, S.
    Schinzel, A.
    Semina, E. V.
    CLINICAL GENETICS, 2016, 90 (04) : 378 - 382
  • [10] A Tunisian Patient with Two Rare Syndromes: Triple A Syndrome and Congenital Hypogonadotropic Hypogonadism
    Ben Abdallah, Lamia Cherif
    Lakhoua, Youssef
    Nagara, Majdi
    Khiari, Karima
    Elouej, Sahar
    Messaoud, Olfa
    Bouyacoub, Yosra
    Romdhane, Lilia
    Turki, Zinet
    Abdelhak, Sonia
    Ben Abdallah, Nejib
    HORMONE RESEARCH IN PAEDIATRICS, 2014, 82 (05): : 338 - 343