Literature DB >> 31833208

Amniotic band sequence in paternal half-siblings with vascular Ehlers-Danlos syndrome.

Mary B Callaghan1, Rob Hadden2, Jon S King3, Katherine Lachlan4, Fleur S van Dijk5, Peter D Turnpenny1.   

Abstract

Familial amniotic band sequence (ABS) is rare but has been reported in the offspring of mothers with connective tissue disorders. We present a family of two half-siblings with ABS who share the same biological father. Following a serious vascular event a de novo pathogenic variant in COL3A1 was detected in the father, confirming a diagnosis of vascular Ehlers-Danlos syndrome (vEDS). The same variant was found in both his ABS-affected children but not in his unaffected child. The amniotic membrane is derived from fetal tissue, type III collagen being a component. As the affected children are paternal half-siblings, ABS was less likely due to maternal factors. Rather, the amniotic bands may have resulted from decreased type III collagen production as seen in people with vEDS, causing fragility of the amniotic membrane. Consequently, it is important to consider vEDS in patients with ABS.
© 2019 Wiley Periodicals, Inc.

Entities:  

Keywords:  zzm321990COL3A1 defects; amniotic band sequence; vascular Ehlers-Danlos syndrome

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Year:  2019        PMID: 31833208     DOI: 10.1002/ajmg.a.61449

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


  1 in total

1.  Inherited cause of in utero digital malformations.

Authors:  Catherine Gooch; Caitlin Wright; Katherine Nelson; Nathaniel Robin
Journal:  BMJ Case Rep       Date:  2020-03-24
  1 in total

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