Literature DB >> 28670783

A de novo germline mutation of DLX3 in a Brown Swiss calf with tricho-dento-osseus-like syndrome.

Sonja Hofstetter1, Monika Welle2,3, Daniela Gorgas4, Pierre Balmer3,5, Petra Roosje3,5, Thomas Mock6, Mireille Meylan6, Vidhya Jagannathan1, Cord Drögemüller1.   

Abstract

OBJECTIVE: A novel congenital disorder affecting a calf was observed, and its phenotype and genetic mutation identified. ANIMAL: A six-month-old female Brown Swiss calf.
METHODS: Diagnostic investigation and whole genome sequencing of a case parent trio was performed.
RESULTS: The calf had a dull kinky coat with mild hypotrichosis, and teeth with brown staining and enamel defects. Histological examination of skin biopsies was compatible with a follicular dysplasia. Radiography and computed tomography revealed thickening of the skull bones and large pulp cavities with a marked thinning of enamel affecting all teeth. A de novo germline mutation affecting the distal-less homeobox gene (DLX3) was identified. The 10 bp frameshift mutation in exon 3 of the bovine DLX3 gene is predicted to replace the second C-terminal transactivation domain of the wild-type protein by a recoded peptide of 99 amino acids without any sequence similarity. CONCLUSION AND CLINICAL IMPORTANCE: A causative mutation for a sporadic phenotype resembling human tricho-dento-osseous syndrome was identified after detection of a de novo germline mutation in the DLX3 gene.
© 2017 ESVD and ACVD.

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Year:  2017        PMID: 28670783     DOI: 10.1111/vde.12462

Source DB:  PubMed          Journal:  Vet Dermatol        ISSN: 0959-4493            Impact factor:   1.589


  1 in total

1.  Mutant dlx3b disturbs normal tooth mineralization and bone formation in zebrafish.

Authors:  Liping Pang; Zhichun Zhang; Yan Shen; Zhenchao Cheng; Xuejun Gao; Bo Zhang; Xiaoyan Wang; Hua Tian
Journal:  PeerJ       Date:  2020-02-19       Impact factor: 2.984

  1 in total

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