Literature DB >> 29483676

The second point mutation in PREPL: a case report and literature review.

Sebastian Silva1, Noriko Miyake2, Carolina Tapia3, Naomichi Matsumoto4.   

Abstract

Prolyl endopeptidase-like (PREPL) deficiency (MIM# 616224) is a rare autosomal recessive inherited congenital myasthenic syndrome characterized by neonatal hypotonia, feeding problems, mild dysmorphism, and neuromuscular symptoms, followed by hyperphagia and obesity in later childhood. Some patients also exhibit growth deficits, sexual hormone deficiency, and cognitive impairments. This syndrome is caused by biallelic mutations in PREPL. To date, only one nucleotide deletion and seven small microdeletions in PREPL have been reported. Here we report a female patient with a novel homozygous frameshift mutation in PREPL (NM_006036.4, c.342delA:p.Val115Leufs*39). Her clinical features are similar to those of previously reported cases. The mutation is the first homozygous point mutation reported in humans.

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Year:  2018        PMID: 29483676     DOI: 10.1038/s10038-018-0426-y

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  16 in total

1.  Discovery and statistical genotyping of copy-number variation from whole-exome sequencing depth.

Authors:  Menachem Fromer; Jennifer L Moran; Kimberly Chambert; Eric Banks; Sarah E Bergen; Douglas M Ruderfer; Robert E Handsaker; Steven A McCarroll; Michael C O'Donovan; Michael J Owen; George Kirov; Patrick F Sullivan; Christina M Hultman; Pamela Sklar; Shaun M Purcell
Journal:  Am J Hum Genet       Date:  2012-10-05       Impact factor: 11.025

2.  Novel KCNB1 mutation associated with non-syndromic intellectual disability.

Authors:  Xénia Latypova; Naomichi Matsumoto; Cécile Vinceslas-Muller; Stéphane Bézieau; Bertrand Isidor; Noriko Miyake
Journal:  J Hum Genet       Date:  2016-12-08       Impact factor: 3.172

3.  Accurate and exact CNV identification from targeted high-throughput sequence data.

Authors:  Alex S Nord; Ming Lee; Mary-Claire King; Tom Walsh
Journal:  BMC Genomics       Date:  2011-04-12       Impact factor: 3.969

4.  Further delineation of genotype-phenotype correlation in homozygous 2p21 deletion syndromes: first description of patients without cystinuria.

Authors:  Deborah Bartholdi; Reza Asadollahi; Beatrice Oneda; Thomas Schmitt-Mechelke; Paolo Tonella; Alessandra Baumer; Anita Rauch
Journal:  Am J Med Genet A       Date:  2013-06-21       Impact factor: 2.802

5.  2p21 Deletions in hypotonia-cystinuria syndrome.

Authors:  Thomas Eggermann; Sabrina Spengler; Andreas Venghaus; Bernd Denecke; Klaus Zerres; Michael Baudis; Regina Ensenauer
Journal:  Eur J Med Genet       Date:  2012-07-17       Impact factor: 2.708

6.  Deletion of PREPL, a gene encoding a putative serine oligopeptidase, in patients with hypotonia-cystinuria syndrome.

Authors:  Jaak Jaeken; Kevin Martens; Inge Francois; Francois Eyskens; Claudine Lecointre; Rita Derua; Sandra Meulemans; Jerry W Slootstra; Etienne Waelkens; Francis de Zegher; John W M Creemers; Gert Matthijs
Journal:  Am J Hum Genet       Date:  2005-11-23       Impact factor: 11.025

7.  Global distribution of the most prevalent deletions causing hypotonia-cystinuria syndrome.

Authors:  Kevin Martens; Inge Heulens; Sandra Meulemans; Marco Zaffanello; David Tilstra; Frederik J Hes; Raoul Rooman; Inge François; Francis de Zegher; Jaak Jaeken; Gert Matthijs; John W M Creemers
Journal:  Eur J Hum Genet       Date:  2007-06-20       Impact factor: 4.246

8.  Two novel deletions in hypotonia-cystinuria syndrome.

Authors:  Luc Régal; Halil Ibrahim Aydin; Anne-Marie Dieltjens; Hilde Van Esch; Inge Francois; Ilyas Okur; Cengiz Zeybek; Sandra Meulemans; Christine Van Mol; Lore Van Bruwaene; Siao-Hann Then; Jaak Jaeken; John Creemers
Journal:  Mol Genet Metab       Date:  2012-06-26       Impact factor: 4.797

9.  Whole exome sequencing of suspected mitochondrial patients in clinical practice.

Authors:  Saskia B Wortmann; David A Koolen; Jan A Smeitink; Lambert van den Heuvel; Richard J Rodenburg
Journal:  J Inherit Metab Dis       Date:  2015-03-04       Impact factor: 4.982

10.  PREPL deficiency: delineation of the phenotype and development of a functional blood assay.

Authors:  Luc Régal; Emma Mårtensson; Isabelle Maystadt; Nicol Voermans; Damien Lederer; Alberto Burlina; María Jesús Juan Fita; A Jeannette M Hoogeboom; Mia Olsson Engman; Tess Hollemans; Meyke Schouten; Sandra Meulemans; Tord Jonson; Inge François; David Gil Ortega; Erik-Jan Kamsteeg; John W M Creemers
Journal:  Genet Med       Date:  2017-07-20       Impact factor: 8.822

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  2 in total

1.  A Family Case of Congenital Myasthenic Syndrome-22 Induced by Different Combinations of Molecular Causes in Siblings.

Authors:  Olga Shchagina; Ludmila Bessonova; Igor Bychkov; Tatiana Beskorovainaya; Aleksander Poliakov
Journal:  Genes (Basel)       Date:  2020-07-19       Impact factor: 4.096

2.  First maternal uniparental disomy for chromosome 2 with PREPL novel frameshift mutation of congenital myasthenic syndrome 22 in an infant.

Authors:  Ping Zhang; Bingbing Wu; Yulan Lu; Qi Ni; Renchao Liu; Wenhao Zhou; Huijun Wang
Journal:  Mol Genet Genomic Med       Date:  2020-01-27       Impact factor: 2.183

  2 in total

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