title: Zebrafish as a model system to study heritable skin diseases.
abstract: Heritable skin diseases represent a broad spectrum of clinical manifestations due to mutations in ∼500 different genes. A number of model systems have been developed to advance our understanding of the pathomechanisms of genodermatoses. Zebrafish (Danio rerio), a freshwater vertebrate, has a well-characterized genome, the expression of which can be easily manipulated. The larvae develop rapidly, with all major organs having largely developed by 5-6 days post-fertilization, including the skin which consists at that stage of the epidermis comprising two cell layers and separated from the dermal collagenous matrix by a basement membrane zone. Here, we describe the use of morpholino-based antisense oligonucleotides to knockdown the expression of specific genes in zebrafish and to examine the consequent knockdown efficiency and skin phenotypes. Zebrafish can provide a useful model system to study heritable skin diseases.
authors: Department of Dermatology and Cutaneous Biology, Jefferson Medical College, Philadelphia, PA, USA. Qiaoli.Li@Jefferson.edu
authors: Qiaoli
authors: Q
authors: Li
authors: Jouni
authors: J
authors: Uitto
mesh_terms: D000818:Animals; D004195:Disease Models, Animal; D055808:Drug Discovery / Q000379:methods; D055785:Gene Knockdown Techniques / Q000379:methods*; D015698:Genomic Library; D008845:Microinjections / Q000379:methods; D060172:Morpholinos / Q000008:administration & dosage* / Q000235:genetics; D016376:Oligonucleotides, Antisense / Q000008:administration & dosage* / Q000235:genetics; D010641:Phenotype; D012867:Skin / Q000378:metabolism / Q000473:pathology*; D012871:Skin Diseases / Q000235:genetics* / Q000473:pathology; D015027:Zebrafish / Q000196:embryology / Q000235:genetics*; D029961:Zebrafish Proteins / Q000235:genetics
chemical_list: D060172:Morpholinos; D016376:Oligonucleotides, Antisense; D029961:Zebrafish Proteins