Literature DB >> 31437444

Nicastrin Deficiency Induces Tyrosinase-Dependent Depigmentation and Skin Inflammation.

Chia-Hao Hsu1, Gunn-Guang Liou2, Yun-Jin Jiang3.   

Abstract

Skin depigmentation diseases, such as vitiligo, are pigmentation disorders that often destroy melanocytes. However, their pathological mechanisms remain unclear, and therefore, promising treatments or prevention has been lacking. Here, we demonstrate that a zebrafish insertional mutant showing a significant reduction of nicastrin transcript possesses melanosome maturation defect, Tyrosinase-dependent mitochondrial swelling, and melanophore cell death. The depigmentation phenotypes are proven to be a result of γ-secretase inactivation. Furthermore, live imaging demonstrates that macrophages are recruited to and can phagocytose melanophore debris. Thus, we characterize a potential zebrafish depigmentation disease model, a nicastrinhi1384 mutant, which can be used for further treatment or drug development of diseases related to skin depigmentation and/or inflammation.
Copyright © 2019 The Authors. Published by Elsevier Inc. All rights reserved.

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Year:  2019        PMID: 31437444     DOI: 10.1016/j.jid.2019.07.702

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  1 in total

1.  Novel nicastrin mutation in hidradenitis suppurativa-Dowling-Degos disease clinical phenotype: more than just clinical overlap?

Authors:  S Garcovich; P M Tricarico; C Nait-Meddour; G Giovanardi; K Peris; S Crovella; M Boniotto
Journal:  Br J Dermatol       Date:  2020-05-26       Impact factor: 9.302

  1 in total

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