Literature DB >> 25168863

Prenatal findings in carpenter syndrome and a novel mutation in RAB23.

Damien Haye1, Corinne Collet, Catherine Sembely-Taveau, Georges Haddad, Christelle Denis, Nathalie Soulé, Annie-Laure Suc, Antoine Listrat, Annick Toutain.   

Abstract

Carpenter syndrome is caused by mutations of the RAB23 gene. To date, 12 distinct mutations have been identified among 34 patients from 26 unrelated families. We report on the prenatal findings in a fetus with Carpenter syndrome with a novel RAB23 mutation. Cystic hygroma, bowed femora, abnormal skull shape and a complex heart defect were seen on ultrasound scan, and Carpenter syndrome was diagnosed at birth. Craniosynostosis and preaxial hexadactyly of the feet were retrospectively detectable on the fetal CT scan. Sequencing of RAB23 identified a homozygous mutation leading to skipping of exon 6 and premature termination codon (c.481G>C; p.Val161Leufs*16). This observation illustrates the difficulty of prenatal ultrasound diagnosis of Carpenter syndrome. To our knowledge, this diagnosis was suggested on ultrasound scan in only one prior patient, although in five other patients abnormal skull shape and variable findings, mainly limb anomalies including bowed femora in one case, were described during the pregnancy. Heart defect and bowed femora are rare postnatal findings. The diagnosis of Carpenter syndrome should therefore be considered on prenatal imaging in cases of bowed femora and/or cardiac defect associated with abnormal skull shape.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  RAB23 gene; acrocephalopolysyndactyly type II; bowed femora; carpenter syndrome; heart defect; prenatal diagnosis

Mesh:

Substances:

Year:  2014        PMID: 25168863     DOI: 10.1002/ajmg.a.36726

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


  11 in total

1.  Ultrasound and MR imaging findings in prenatal diagnosis of craniosynostosis syndromes.

Authors:  Eva I Rubio; Anna Blask; Dorothy I Bulas
Journal:  Pediatr Radiol       Date:  2016-02-25

2.  Drosophila Homolog of the Human Carpenter Syndrome Linked Gene, MEGF8, Is Required for Synapse Development and Function.

Authors:  Shuting Chen; Anand Venkatesan; Yong Qi Lin; Jing Xie; Gregory Neely; Swati Banerjee; Manzoor A Bhat
Journal:  J Neurosci       Date:  2022-08-09       Impact factor: 6.709

Review 3.  Genetic determinants of syndactyly: perspectives on pathogenesis and diagnosis.

Authors:  Afraah Cassim; Dineshani Hettiarachchi; Vajira H W Dissanayake
Journal:  Orphanet J Rare Dis       Date:  2022-05-12       Impact factor: 4.303

4.  Large-scale computational drug repositioning to find treatments for rare diseases.

Authors:  Rajiv Gandhi Govindaraj; Misagh Naderi; Manali Singha; Jeffrey Lemoine; Michal Brylinski
Journal:  NPJ Syst Biol Appl       Date:  2018-03-13

Review 5.  Rab23's genetic structure, function and related diseases: a review.

Authors:  Li-Qiang Zheng; Su-Min Chi; Cheng-Xin Li
Journal:  Biosci Rep       Date:  2017-03-02       Impact factor: 3.840

6.  RNA-sequencing analysis of umbilical cord plasma microRNAs from healthy newborns.

Authors:  Gary P Brennan; Dimitrios M Vitsios; Sophie Casey; Ann-Marie Looney; Boubou Hallberg; David C Henshall; Geraldine B Boylan; Deirdre M Murray; Catherine Mooney
Journal:  PLoS One       Date:  2018-12-03       Impact factor: 3.240

Review 7.  Rab GTPases: Switching to Human Diseases.

Authors:  Noemi Antonella Guadagno; Cinzia Progida
Journal:  Cells       Date:  2019-08-16       Impact factor: 6.600

8.  Multifaceted Functions of Rab23 on Primary Cilium-Mediated and Hedgehog Signaling-Mediated Cerebellar Granule Cell Proliferation.

Authors:  C H H Hor; J C W Lo; A L S Cham; W Y Leong; E L K Goh
Journal:  J Neurosci       Date:  2021-07-01       Impact factor: 6.167

Review 9.  Rab GTPases in cilium formation and function.

Authors:  Oliver E Blacque; Noemie Scheidel; Stefanie Kuhns
Journal:  Small GTPases       Date:  2017-10-26

Review 10.  Ciliary Signalling and Mechanotransduction in the Pathophysiology of Craniosynostosis.

Authors:  Federica Tiberio; Ornella Parolini; Wanda Lattanzi
Journal:  Genes (Basel)       Date:  2021-07-14       Impact factor: 4.096

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