Identification of a novel PLCD1 mutation in Chinese Han pedigree with hereditary leukonychia and koilonychia.


Journal

Journal of cosmetic dermatology
ISSN: 1473-2165
Titre abrégé: J Cosmet Dermatol
Pays: England
ID NLM: 101130964

Informations de publication

Date de publication:
Jun 2019
Historique:
received: 22 02 2018
accepted: 31 05 2018
pubmed: 14 7 2018
medline: 18 12 2019
entrez: 14 7 2018
Statut: ppublish

Résumé

Hereditary leukonychia is a rare nail dystrophy characterized by distinctive whitening of the nail plate. Mutations in the PLCD1 gene have been identified as a major causative factor in hereditary leukonychia (HL). However, few reports have analyzed the relationship between genotype and phenotype, especially in Chinese HL patients. Our study aims to explore the typical clinical features of hereditary leukonychia cases in Chinese Han pedigree and the correlations with PLCD1 gene mutation. In this study, two Chinese patients presented with leukonychia and koilonychia. Whole-exome sequencing (WES) was performed to screen for the mutations in PLCD1 gene and other candidate genes for hereditary leukonychia. Parents with PLCD1 mutation were selected for Sanger sequencing. A novel heterozygote missense mutation in exon 9 of PLCD1 gene was identified in the proband and his mother. Whole-exome sequencing revealed both, the proband (III.5) and his mother (II.4) carrying c.1451A>G mutation, while other family members had a normal sequence of the PLCD1 gene. For the first time, a hereditary leukonychia case with PLCD1 mutation has been described in Chinese Han pedigree. This finding suggests the PLCD1 mutation maybe involved in hereditary leukonychia.

Sections du résumé

BACKGROUND BACKGROUND
Hereditary leukonychia is a rare nail dystrophy characterized by distinctive whitening of the nail plate. Mutations in the PLCD1 gene have been identified as a major causative factor in hereditary leukonychia (HL). However, few reports have analyzed the relationship between genotype and phenotype, especially in Chinese HL patients. Our study aims to explore the typical clinical features of hereditary leukonychia cases in Chinese Han pedigree and the correlations with PLCD1 gene mutation.
PATIENTS AND METHODS METHODS
In this study, two Chinese patients presented with leukonychia and koilonychia. Whole-exome sequencing (WES) was performed to screen for the mutations in PLCD1 gene and other candidate genes for hereditary leukonychia. Parents with PLCD1 mutation were selected for Sanger sequencing.
RESULTS RESULTS
A novel heterozygote missense mutation in exon 9 of PLCD1 gene was identified in the proband and his mother. Whole-exome sequencing revealed both, the proband (III.5) and his mother (II.4) carrying c.1451A>G mutation, while other family members had a normal sequence of the PLCD1 gene.
CONCLUSION CONCLUSIONS
For the first time, a hereditary leukonychia case with PLCD1 mutation has been described in Chinese Han pedigree. This finding suggests the PLCD1 mutation maybe involved in hereditary leukonychia.

Identifiants

pubmed: 30003652
doi: 10.1111/jocd.12707
doi:

Substances chimiques

PLCD1 protein, human EC 3.1.4.11
Phospholipase C delta EC 3.1.4.11

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

912-915

Subventions

Organisme : National Key Basic Research Program of China
ID : 2014CB541901
Organisme : National Nature Science Foundation of China
ID : 30500440

Informations de copyright

© 2018 Wiley Periodicals, Inc.

Auteurs

Ke Xue (K)

Department of Dermatology, China-Japan Friendship Hospital, Beijing, China.
Graduate School of Peking Union Medical College, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.

Yajie Zheng (Y)

Institute of Dermatology and Department of Dermatology, The First Affiliated Hospital, Anhui Medical University, Hefei, China.

Changbing Shen (C)

Department of Dermatology, China-Japan Friendship Hospital, Beijing, China.
Graduate School of Peking Union Medical College, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.

Yong Cui (Y)

Department of Dermatology, China-Japan Friendship Hospital, Beijing, China.
Graduate School of Peking Union Medical College, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.
Institute of Dermatology and Department of Dermatology, The First Affiliated Hospital, Anhui Medical University, Hefei, China.

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