Literature DB >> 9382145

Skeletal anomalies and deformities in patients with deletions of 22q11.

J E Ming1, D M McDonald-McGinn, T E Megerian, D A Driscoll, E R Elias, B M Russell, M Irons, B S Emanuel, R I Markowitz, E H Zackai.   

Abstract

Skeletal anomalies in patients with a 22q11.2 deletion are reported infrequently. We report the skeletal findings in 108 patients with a 22q11.2 deletion, of whom 37 (36%) had a skeletal anomaly. Twenty-two patients (20%) had anomalies of the limbs, 7 of the upper limb, including preaxial or postaxial polydactyly. An anomaly of the lower limb was found in 16 patients, including postaxial polydactyly, clubfoot, severely overfolded toes, and 2-3 toe cutaneous syndactyly. Chest films of 63 patients were examined; 30% of them had abnormal findings, most commonly supernumerary ribs (17%) or a "butterfly" vertebral body (11%). Hypoplastic vertebrae, hemivertebrae, and vertebral coronal clefts were also noted. Thus, skeletal anomalies are not uncommon in patients with a 22q11.2 deletion and may occur more frequently than recognized previously.

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Mesh:

Year:  1997        PMID: 9382145     DOI: 10.1002/(sici)1096-8628(19971017)72:2<210::aid-ajmg16>3.0.co;2-q

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  21 in total

1.  Velocardiofacial syndrome presenting as distal arthrogryposis.

Authors:  Koen Devriendt; Ann Swillen; Marc Gewillig; Jean-Pierre Fryns; Pierre Moens; Luc De Smet
Journal:  Eur J Pediatr       Date:  2004-06       Impact factor: 3.183

Review 2.  Syndromes of the first and second branchial arches, part 2: syndromes.

Authors:  J M Johnson; G Moonis; G E Green; R Carmody; H N Burbank
Journal:  AJNR Am J Neuroradiol       Date:  2010-04-01       Impact factor: 3.825

3.  Qualitative MRI findings in adults with 22q11 deletion syndrome and schizophrenia.

Authors:  E W Chow; D J Mikulis; R B Zipursky; L E Scutt; R Weksberg; A S Bassett
Journal:  Biol Psychiatry       Date:  1999-11-15       Impact factor: 13.382

Review 4.  A review of supernumerary and absent limbs and digits of the upper limb.

Authors:  Zachary Klaassen; Monica Choi; Ruth Musselman; Deborah Eapen; R Shane Tubbs; Marios Loukas
Journal:  Surg Radiol Anat       Date:  2011-11-09       Impact factor: 1.246

5.  Expanding the fetal phenotype: Prenatal sonographic findings and perinatal outcomes in a cohort of patients with a confirmed 22q11.2 deletion syndrome.

Authors:  Erica Schindewolf; Nahla Khalek; Mark P Johnson; Juliana Gebb; Beverly Coleman; Terrence Blaine Crowley; Elaine H Zackai; Donna M McDonald-McGinn; Julie S Moldenhauer
Journal:  Am J Med Genet A       Date:  2018-07-28       Impact factor: 2.802

Review 6.  Modeling a model: Mouse genetics, 22q11.2 Deletion Syndrome, and disorders of cortical circuit development.

Authors:  Daniel W Meechan; Thomas M Maynard; Eric S Tucker; Alejandra Fernandez; Beverly A Karpinski; Lawrence A Rothblat; Anthony-S LaMantia
Journal:  Prog Neurobiol       Date:  2015-04-09       Impact factor: 11.685

7.  Copy number analysis of 413 isolated talipes equinovarus patients suggests role for transcriptional regulators of early limb development.

Authors:  David M Alvarado; Jillian G Buchan; Steven L Frick; John E Herzenberg; Matthew B Dobbs; Christina A Gurnett
Journal:  Eur J Hum Genet       Date:  2012-08-15       Impact factor: 4.246

8.  Consequences of JAG1 mutations.

Authors:  B M Kamath; L Bason; D A Piccoli; I D Krantz; N B Spinner
Journal:  J Med Genet       Date:  2003-12       Impact factor: 6.318

Review 9.  22q11.2 deletion syndrome.

Authors:  Donna M McDonald-McGinn; Kathleen E Sullivan; Bruno Marino; Nicole Philip; Ann Swillen; Jacob A S Vorstman; Elaine H Zackai; Beverly S Emanuel; Joris R Vermeesch; Bernice E Morrow; Peter J Scambler; Anne S Bassett
Journal:  Nat Rev Dis Primers       Date:  2015-11-19       Impact factor: 52.329

10.  Three patients with oculo-auriculo-vertebral spectrum and microdeletion 22q11.2.

Authors:  M Cristina Digilio; Donna M McDonald-McGinn; Carrie Heike; Charles Catania; Bruno Dallapiccola; Bruno Marino; Elaine H Zackai
Journal:  Am J Med Genet A       Date:  2009-12       Impact factor: 2.802

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