Literature DB >> 32256301

Robinow Syndrome and Brachydactyly: An Interplay of High-Throughput Sequencing and Deep Phenotyping in a Kindred.

Ranjana Mishra1, Vibha Jain1, Deepti Gupta1, Renu Saxena1, Samarth Kulshreshtha1, Vedam L Ramprasad2, Ishwar C Verma1, Ratna Dua Puri1.   

Abstract

We report a family with a spectrum of short stature, craniofacial dysmorphism, and digital anomalies in a father and 2 daughters, with the youngest (proband) displaying a severe phenotype. Clinically, autosomal dominant Robinow syndrome (ADRS) was diagnosed. Whole-exome sequencing identified a heterozygous pathogenic BMP2 variant in the father and his daughters. The phenotype of short stature, facial dysmorphism, and skeletal anomalies with or without cardiac anomalies related to BMP2 haploinsufficiency has some facial and digital resemblance to ADRS. Although this variant segregated in the affected members, it failed to explain the severe phenotype of the proband. A reanalysis of the girl's raw data confirmed 2 disorders: a de novo likely pathogenic DVL1 variant implicated in ADRS and the familial BMP2 variant. A close interplay of high-throughput sequencing and deep phenotyping unraveled the complexities of the blended phenotype in the proband.
Copyright © 2020 by S. Karger AG, Basel.

Entities:  

Keywords:  BMP2; Blended phenotype; DVL1; Reanalysis; Whole-exome sequencing

Year:  2020        PMID: 32256301      PMCID: PMC7109436          DOI: 10.1159/000505506

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  15 in total

1.  Mutations in DVL1 cause an osteosclerotic form of Robinow syndrome.

Authors:  Kieran J Bunn; Phil Daniel; Heleen S Rösken; Adam C O'Neill; Sophia R Cameron-Christie; Tim Morgan; Han G Brunner; Angeline Lai; Henricus P M Kunst; David M Markie; Stephen P Robertson
Journal:  Am J Hum Genet       Date:  2015-03-26       Impact factor: 11.025

2.  DVL1 frameshift mutations clustering in the penultimate exon cause autosomal-dominant Robinow syndrome.

Authors:  Janson White; Juliana F Mazzeu; Alexander Hoischen; Shalini N Jhangiani; Tomasz Gambin; Michele Calijorne Alcino; Samantha Penney; Jorge M Saraiva; Hanne Hove; Flemming Skovby; Hülya Kayserili; Elicia Estrella; Anneke T Vulto-van Silfhout; Marloes Steehouwer; Donna M Muzny; V Reid Sutton; Richard A Gibbs; James R Lupski; Han G Brunner; Bregje W M van Bon; Claudia M B Carvalho
Journal:  Am J Hum Genet       Date:  2015-03-26       Impact factor: 11.025

3.  WNT Signaling Perturbations Underlie the Genetic Heterogeneity of Robinow Syndrome.

Authors:  Janson J White; Juliana F Mazzeu; Zeynep Coban-Akdemir; Yavuz Bayram; Vahid Bahrambeigi; Alexander Hoischen; Bregje W M van Bon; Alper Gezdirici; Elif Yilmaz Gulec; Francis Ramond; Renaud Touraine; Julien Thevenon; Marwan Shinawi; Erin Beaver; Jennifer Heeley; Julie Hoover-Fong; Ceren D Durmaz; Halil Gurhan Karabulut; Ebru Marzioglu-Ozdemir; Atilla Cayir; Mehmet B Duz; Mehmet Seven; Susan Price; Barbara Merfort Ferreira; Angela M Vianna-Morgante; Sian Ellard; Andrew Parrish; Karen Stals; Josue Flores-Daboub; Shalini N Jhangiani; Richard A Gibbs; Han G Brunner; V Reid Sutton; James R Lupski; Claudia M B Carvalho
Journal:  Am J Hum Genet       Date:  2017-12-21       Impact factor: 11.025

Review 4.  Integration of BMP, Wnt, and notch signaling pathways in osteoblast differentiation.

Authors:  Grace L Lin; Kurt D Hankenson
Journal:  J Cell Biochem       Date:  2011-12       Impact factor: 4.429

5.  The Sequence Alignment/Map format and SAMtools.

Authors:  Heng Li; Bob Handsaker; Alec Wysoker; Tim Fennell; Jue Ruan; Nils Homer; Gabor Marth; Goncalo Abecasis; Richard Durbin
Journal:  Bioinformatics       Date:  2009-06-08       Impact factor: 6.937

6.  Monoallelic BMP2 Variants Predicted to Result in Haploinsufficiency Cause Craniofacial, Skeletal, and Cardiac Features Overlapping Those of 20p12 Deletions.

Authors:  Tiong Yang Tan; Claudia Gonzaga-Jauregui; Elizabeth J Bhoj; Kevin A Strauss; Karlla Brigatti; Erik Puffenberger; Dong Li; LiQin Xie; Nanditha Das; Ioanna Skubas; Ron A Deckelbaum; Virginia Hughes; Susannah Brydges; Sarah Hatsell; Chia-Jen Siao; Melissa G Dominguez; Aris Economides; John D Overton; Valerie Mayne; Peter J Simm; Bryn O Jones; Stefanie Eggers; Gwenaël Le Guyader; Fanny Pelluard; Tobias B Haack; Marc Sturm; Angelika Riess; Stephan Waldmueller; Michael Hofbeck; Katharina Steindl; Pascal Joset; Anita Rauch; Hakon Hakonarson; Naomi L Baker; Peter G Farlie
Journal:  Am J Hum Genet       Date:  2017-11-30       Impact factor: 11.025

7.  Resolution of Disease Phenotypes Resulting from Multilocus Genomic Variation.

Authors:  Jennifer E Posey; Tamar Harel; Pengfei Liu; Jill A Rosenfeld; Regis A James; Zeynep H Coban Akdemir; Magdalena Walkiewicz; Weimin Bi; Rui Xiao; Yan Ding; Fan Xia; Arthur L Beaudet; Donna M Muzny; Richard A Gibbs; Eric Boerwinkle; Christine M Eng; V Reid Sutton; Chad A Shaw; Sharon E Plon; Yaping Yang; James R Lupski
Journal:  N Engl J Med       Date:  2016-12-07       Impact factor: 91.245

8.  Deriving the consequences of genomic variants with the Ensembl API and SNP Effect Predictor.

Authors:  William McLaren; Bethan Pritchard; Daniel Rios; Yuan Chen; Paul Flicek; Fiona Cunningham
Journal:  Bioinformatics       Date:  2010-06-18       Impact factor: 6.937

9.  Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology.

Authors:  Sue Richards; Nazneen Aziz; Sherri Bale; David Bick; Soma Das; Julie Gastier-Foster; Wayne W Grody; Madhuri Hegde; Elaine Lyon; Elaine Spector; Karl Voelkerding; Heidi L Rehm
Journal:  Genet Med       Date:  2015-03-05       Impact factor: 8.822

10.  Fast and accurate long-read alignment with Burrows-Wheeler transform.

Authors:  Heng Li; Richard Durbin
Journal:  Bioinformatics       Date:  2010-01-15       Impact factor: 6.937

View more
  2 in total

Review 1.  A novel frameshift mutation of DVL1-induced Robinow syndrome: A case report and literature review.

Authors:  Ruolan Hu; Yu Qiu; Yifei Li; Jinrong Li
Journal:  Mol Genet Genomic Med       Date:  2022-02-09       Impact factor: 2.183

2.  Novel pathogenic variants and quantitative phenotypic analyses of Robinow syndrome: WNT signaling perturbation and phenotypic variability.

Authors:  Chaofan Zhang; Angad Jolly; Brian J Shayota; Juliana F Mazzeu; Haowei Du; Moez Dawood; Patricia Celestino Soper; Ariadne Ramalho de Lima; Bárbara Merfort Ferreira; Zeynep Coban-Akdemir; Janson White; Deborah Shears; Fraser Robert Thomson; Sarah Louise Douglas; Andrew Wainwright; Kathryn Bailey; Paul Wordsworth; Mike Oldridge; Tracy Lester; Alistair D Calder; Katja Dumic; Siddharth Banka; Dian Donnai; Shalini N Jhangiani; Lorraine Potocki; Wendy K Chung; Sara Mora; Hope Northrup; Myla Ashfaq; Jill A Rosenfeld; Kati Mason; Lynda C Pollack; Allyn McConkie-Rosell; Wei Kelly; Marie McDonald; Natalie S Hauser; Peter Leahy; Cynthia M Powell; Raquel Boy; Rachel Sayuri Honjo; Fernando Kok; Lucia R Martelli; Vicente Odone Filho; Donna M Muzny; Richard A Gibbs; Jennifer E Posey; Pengfei Liu; James R Lupski; V Reid Sutton; Claudia M B Carvalho
Journal:  HGG Adv       Date:  2021-12-03
  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.