Literature DB >> 29987841

Report of second case and clinical and molecular characterization of Eiken syndrome.

A Moirangthem1, D L Narayanan2, P Jacob1, G Nishimura3, G Mortier4, K M Girisha1.   

Abstract

We report a boy with Eiken syndrome caused by a homozygous missense variant in Parathyroid hormone 1 receptor (PTH1R) c.103G > A [p.(Glu35Lys)]. Eiken syndrome is a very rare skeletal dysplasia due to bi-allelic variants in PTH1R. Only one affected family has been known to-date. The hallmarks include delayed ossification of bone including the epiphyses, pubic symphysis, and primary ossification centers of the short tubular bones, coarse bone trabeculae, and modeling abnormalities. The phenotype being described here recapitulates the delayed ossification and modeling abnormalities of Eiken syndrome. In addition, supernumerary epiphyses of the tubular bones of the hands and primary failure of eruption of teeth were observed in our proband. This report characterizes Eiken syndrome and confirms that bi-allelic hypomorphic variants in PTH1R are probably to cause this condition.
© 2018 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Eiken syndrome; PTH1R; bone remodeling; delayed ossification; pseudoepiphysis; skeletal dysplasia; tooth eruption failure

Mesh:

Substances:

Year:  2018        PMID: 29987841     DOI: 10.1111/cge.13413

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  4 in total

1.  Functional Properties of Two Distinct PTH1R Mutants Associated With Either Skeletal Defects or Pseudohypoparathyroidism.

Authors:  Ignacio Portales-Castillo; Thomas Dean; Ashok Khatri; Harald Jüppner; Thomas J Gardella
Journal:  JBMR Plus       Date:  2022-04-14

2.  A novel nonsense PTH1R variant shows incomplete penetrance of primary failure of eruption: a case report.

Authors:  Cristina Grippaudo; Concetta Cafiero; Isabella D'Apolito; Agnese Re; Maurizio Genuardi; Pietro Chiurazzi; Sylvia A Frazier-Bowers
Journal:  BMC Oral Health       Date:  2019-11-15       Impact factor: 2.757

3.  Case Report: Inactivating PTH/PTHrP Signaling Disorder Type 1 Presenting With PTH Resistance.

Authors:  Tanguy Demaret; René Wintjens; Gwenaelle Sana; Joachim Docquir; Frederic Bertin; Christophe Ide; Olivier Monestier; Deniz Karadurmus; Valerie Benoit; Isabelle Maystadt
Journal:  Front Endocrinol (Lausanne)       Date:  2022-06-30       Impact factor: 6.055

Review 4.  Primary failure of tooth eruption: Etiology and management.

Authors:  Tetsutaro Yamaguchi; Kazuyoshi Hosomichi; Tatsuo Shirota; Yoichi Miyamoto; Wanida Ono; Noriaki Ono
Journal:  Jpn Dent Sci Rev       Date:  2022-09-15
  4 in total

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