Literature DB >> 30088852

A comprehensive clinical and genetic study in 127 patients with ID in Kinshasa, DR Congo.

Aimé Lumaka1,2,3,4, Valerie Race5, Hilde Peeters5, Anniek Corveleyn5, Zeynep Coban-Akdemir6, Shalini N Jhangiani6, Xiaofei Song6, Gerrye Mubungu1,3,4,5, Jennifer Posey7, James R Lupski6,7,8,9, Joris R Vermeesch5, Prosper Lukusa1,2,3,5, Koenraad Devriendt5.   

Abstract

Pathogenic variants account for 4 to 41% of patients with intellectual disability (ID) or developmental delay (DD). In Sub-Saharan Africa, the prevalence of ID is thought to be higher, but data in Central Africa are limited to some case reports. In addition, clinical descriptions of some syndromes are not available for this population. This study aimed at providing an estimate for the fraction of ID/DD for which an underlying etiological genetic cause may be elucidated and provide insights into their clinical presentation in special institutions in a Central African country. A total of 127 patients (33 females and 94 males, mean age 10.03 ± 4.68 years), were recruited from six institutions across Kinshasa. A clinical diagnosis was achieved in 44 but molecular confirmation was achieved in 21 of the 22 patients with expected genetic defect (95% clinical sensitivity). Identified diseases included Down syndrome (15%), submicroscopic copy number variants (9%), aminoacylase deficiency (0.8%), Partington syndrome in one patient (0.8%) and his similarly affected brother, X-linked syndromic Mental Retardation type 33 (0.8%), and two conditions without clear underlying molecular genetic etiologies (Oculo-Auriculo-Vertebral and Amniotic Bands Sequence). We have shown that genetic etiologies, similar to those reported in Caucasian subjects, are a common etiologic cause of ID in African patients from Africa. We have confirmed the diagnostic utility of clinical characterization prior to genetic testing. Finally, our clinical descriptions provide insights into the presentation of these genetic diseases in African patients.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  Africa; Down syndrome; NGS in Africa; Partington syndrome; intellectual disability; specialized schools; striped micro-array

Mesh:

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Year:  2018        PMID: 30088852      PMCID: PMC6325645          DOI: 10.1002/ajmg.a.40382

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  70 in total

1.  Screening of ARX in mental retardation families: Consequences for the strategy of molecular diagnosis.

Authors:  K Poirier; D Lacombe; B Gilbert-Dussardier; M Raynaud; V Desportes; A P M de Brouwer; C Moraine; J P Fryns; H H Ropers; C Beldjord; J Chelly; T Bienvenu
Journal:  Neurogenetics       Date:  2005-10-19       Impact factor: 2.660

2.  Diagnostic yield of various genetic approaches in patients with unexplained developmental delay or mental retardation.

Authors:  Anita Rauch; Juliane Hoyer; Sabine Guth; Christiane Zweier; Cornelia Kraus; Christian Becker; Martin Zenker; Ulrike Hüffmeier; Christian Thiel; Franz Rüschendorf; Peter Nürnberg; André Reis; Udo Trautmann
Journal:  Am J Med Genet A       Date:  2006-10-01       Impact factor: 2.802

3.  ARX polyalanine expansions are highly implicated in familial cases of mental retardation with infantile epilepsy and/or hand dystonia.

Authors:  Mireille Cossée; Laurence Faivre; Christophe Philippe; Heifa Hichri; Anne de Saint-Martin; Vincent Laugel; Nadia Bahi-Buisson; Jean-François Lemaitre; Bruno Leheup; Bruno Delobel; Bénédicte Demeer; Karine Poirier; Valérie Biancalana; Jean-Michel Pinoit; Sophie Julia; Jamel Chelly; Didier Devys; Jean-Louis Mandel
Journal:  Am J Med Genet A       Date:  2011-01       Impact factor: 2.802

4.  The black American nose.

Authors:  F A Ofodile; F J Bokhari; C Ellis
Journal:  Ann Plast Surg       Date:  1993-09       Impact factor: 1.539

5.  The 22q11.2 deletion in African-American patients: an underdiagnosed population?

Authors:  Donna M McDonald-McGinn; Nancy Minugh-Purvis; Richard E Kirschner; Abbas Jawad; Melissa K Tonnesen; Jason R Catanzaro; Elizabeth Goldmuntz; Deborah Driscoll; Don Larossa; Beverly S Emanuel; Elaine H Zackai
Journal:  Am J Med Genet A       Date:  2005-04-30       Impact factor: 2.802

6.  Poverty and people with intellectual disabilities.

Authors:  Eric Emerson
Journal:  Ment Retard Dev Disabil Res Rev       Date:  2007

7.  NAHR-mediated copy-number variants in a clinical population: mechanistic insights into both genomic disorders and Mendelizing traits.

Authors:  Piotr Dittwald; Tomasz Gambin; Przemyslaw Szafranski; Jian Li; Stephen Amato; Michael Y Divon; Lisa Ximena Rodríguez Rojas; Lindsay E Elton; Daryl A Scott; Christian P Schaaf; Wilfredo Torres-Martinez; Abby K Stevens; Jill A Rosenfeld; Satish Agadi; David Francis; Sung-Hae L Kang; Amy Breman; Seema R Lalani; Carlos A Bacino; Weimin Bi; Aleksandar Milosavljevic; Arthur L Beaudet; Ankita Patel; Chad A Shaw; James R Lupski; Anna Gambin; Sau Wai Cheung; Pawel Stankiewicz
Journal:  Genome Res       Date:  2013-05-08       Impact factor: 9.043

8.  Homozygous and hemizygous CNV detection from exome sequencing data in a Mendelian disease cohort.

Authors:  Tomasz Gambin; Zeynep C Akdemir; Bo Yuan; Shen Gu; Theodore Chiang; Claudia M B Carvalho; Chad Shaw; Shalini Jhangiani; Philip M Boone; Mohammad K Eldomery; Ender Karaca; Yavuz Bayram; Asbjørg Stray-Pedersen; Donna Muzny; Wu-Lin Charng; Vahid Bahrambeigi; John W Belmont; Eric Boerwinkle; Arthur L Beaudet; Richard A Gibbs; James R Lupski
Journal:  Nucleic Acids Res       Date:  2017-02-28       Impact factor: 16.971

9.  An experimental loop design for the detection of constitutional chromosomal aberrations by array CGH.

Authors:  Joke Allemeersch; Steven Van Vooren; Femke Hannes; Bart De Moor; Joris Robert Vermeesch; Yves Moreau
Journal:  BMC Bioinformatics       Date:  2009-11-19       Impact factor: 3.169

10.  MRX87 family with Aristaless X dup24bp mutation and implication for polyAlanine expansions.

Authors:  Carmela Laperuta; Letizia Spizzichino; Pio D'Adamo; Jlenia Monfregola; Antonio Maiorino; Angela D'Eustacchio; Valerio Ventruto; Giovanni Neri; Michele D'Urso; Pietro Chiurazzi; Matilde Valeria Ursini; Maria Giuseppina Miano
Journal:  BMC Med Genet       Date:  2007-05-04       Impact factor: 2.103

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  4 in total

1.  Increasing African genomic data generation and sharing to resolve rare and undiagnosed diseases in Africa: a call-to-action by the H3Africa rare diseases working group.

Authors:  Aimé Lumaka; Nadia Carstens; Koenraad Devriendt; Amanda Krause; Benard Kulohoma; Judit Kumuthini; Gerrye Mubungu; John Mukisa; Melissa Nel; Timothy O Olanrewaju; Zané Lombard; Guida Landouré
Journal:  Orphanet J Rare Dis       Date:  2022-06-16       Impact factor: 4.303

Review 2.  Centers for Mendelian Genomics: A decade of facilitating gene discovery.

Authors:  Samantha M Baxter; Jennifer E Posey; Nicole J Lake; Nara Sobreira; Jessica X Chong; Steven Buyske; Elizabeth E Blue; Lisa H Chadwick; Zeynep H Coban-Akdemir; Kimberly F Doheny; Colleen P Davis; Monkol Lek; Christopher Wellington; Shalini N Jhangiani; Mark Gerstein; Richard A Gibbs; Richard P Lifton; Daniel G MacArthur; Tara C Matise; James R Lupski; David Valle; Michael J Bamshad; Ada Hamosh; Shrikant Mane; Deborah A Nickerson; Heidi L Rehm; Anne O'Donnell-Luria
Journal:  Genet Med       Date:  2022-02-09       Impact factor: 8.864

3.  Diagnosis of genetic diseases in seriously ill children by rapid whole-genome sequencing and automated phenotyping and interpretation.

Authors:  Michelle M Clark; Amber Hildreth; Sergey Batalov; Yan Ding; Shimul Chowdhury; Kelly Watkins; Katarzyna Ellsworth; Brandon Camp; Cyrielle I Kint; Calum Yacoubian; Lauge Farnaes; Matthew N Bainbridge; Curtis Beebe; Joshua J A Braun; Margaret Bray; Jeanne Carroll; Julie A Cakici; Sara A Caylor; Christina Clarke; Mitchell P Creed; Jennifer Friedman; Alison Frith; Richard Gain; Mary Gaughran; Shauna George; Sheldon Gilmer; Joseph Gleeson; Jeremy Gore; Haiying Grunenwald; Raymond L Hovey; Marie L Janes; Kejia Lin; Paul D McDonagh; Kyle McBride; Patrick Mulrooney; Shareef Nahas; Daeheon Oh; Albert Oriol; Laura Puckett; Zia Rady; Martin G Reese; Julie Ryu; Lisa Salz; Erica Sanford; Lawrence Stewart; Nathaly Sweeney; Mari Tokita; Luca Van Der Kraan; Sarah White; Kristen Wigby; Brett Williams; Terence Wong; Meredith S Wright; Catherine Yamada; Peter Schols; John Reynders; Kevin Hall; David Dimmock; Narayanan Veeraraghavan; Thomas Defay; Stephen F Kingsmore
Journal:  Sci Transl Med       Date:  2019-04-24       Impact factor: 19.319

Review 4.  Clinical presentation and evolution of Xia-Gibbs syndrome due to p.Gly375ArgfsTer3 variant in a patient from DR Congo (Central Africa).

Authors:  Gerrye Mubungu; Prince Makay; Bouchra Boujemla; Stephane Yanda; Jennifer E Posey; James R Lupski; Vincent Bours; Prosper Lukusa; Koenraad Devriendt; Aimé Lumaka
Journal:  Am J Med Genet A       Date:  2020-12-29       Impact factor: 2.578

  4 in total

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