cattle channelopathy craniofacial dysmorphism hydrosyringomyelia neuromuscular disorder paradoxical myotonia congenita potassium voltage-gated channel precision medicine skeletal muscle

Journal

Genes
ISSN: 2073-4425
Titre abrégé: Genes (Basel)
Pays: Switzerland
ID NLM: 101551097

Informations de publication

Date de publication:
12 11 2021
Historique:
received: 18 10 2021
revised: 29 10 2021
accepted: 10 11 2021
entrez: 27 11 2021
pubmed: 28 11 2021
medline: 10 2 2022
Statut: epublish

Résumé

Inherited channelopathies are a clinically and heritably heterogeneous group of disorders that result from ion channel dysfunction. The aim of this study was to characterize the clinicopathologic features of a Belgian Blue x Holstein crossbred calf with paradoxical myotonia congenita, craniofacial dysmorphism, and myelodysplasia, and to identify the most likely genetic etiology. The calf displayed episodes of exercise-induced generalized myotonic muscle stiffness accompanied by increase in serum potassium. It also showed slight flattening of the splanchnocranium with deviation to the right side. On gross pathology, myelodysplasia (hydrosyringomielia and segmental hypoplasia) in the lumbosacral intumescence region was noticed. Histopathology of the muscle profile revealed loss of the main shape in 5.3% of muscle fibers. Whole-genome sequencing revealed a heterozygous missense variant in

Identifiants

pubmed: 34828398
pii: genes12111792
doi: 10.3390/genes12111792
pmc: PMC8618021
pii:
doi:

Substances chimiques

Potassium Channels, Voltage-Gated 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

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Auteurs

Joana G P Jacinto (JGP)

Department of Veterinary Medical Sciences, University of Bologna, 40064 Ozzano Emilia, Italy.
Institute of Genetics, Vetsuisse Faculty, University of Bern, 3012 Bern, Switzerland.

Irene M Häfliger (IM)

Institute of Genetics, Vetsuisse Faculty, University of Bern, 3012 Bern, Switzerland.

Eylem Emek Akyürek (EE)

Department of Comparative Biomedicine and Food Science, University of Padova, 35020 Legnaro, Italy.

Roberta Sacchetto (R)

Department of Comparative Biomedicine and Food Science, University of Padova, 35020 Legnaro, Italy.

Cinzia Benazzi (C)

Department of Veterinary Medical Sciences, University of Bologna, 40064 Ozzano Emilia, Italy.

Arcangelo Gentile (A)

Department of Veterinary Medical Sciences, University of Bologna, 40064 Ozzano Emilia, Italy.

Cord Drögemüller (C)

Institute of Genetics, Vetsuisse Faculty, University of Bern, 3012 Bern, Switzerland.

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Classifications MeSH