عنوان

A CRISPR-based screen for Hedgehog signaling provides insights into ciliary function and ciliopathies.

شماره

LA7pz039q5

عنوان اصلي

A CRISPR-based screen for Hedgehog signaling provides insights into ciliary function and ciliopathies.

نام عام مواد

[Article]

نام نخستين پديدآور

Breslow, David K; Hoogendoorn, Sascha; Kopp, Adam R; Morgens, David W; Vu, Brandon K; Kennedy, Margaret C; Han, Kyuho; Li, Amy; Hess, Gaelen T; Bassik, Michael C; Chen, James K; Nachury, Maxence V

متن يادداشت

Primary cilia organize Hedgehog signaling and shape embryonic development, and their dysregulation is the unifying cause of ciliopathies. We conducted a functional genomic screen for Hedgehog signaling by engineering antibiotic-based selection of Hedgehog-responsive cells and applying genome-wide CRISPR-mediated gene disruption. The screen can robustly identify factors required for ciliary signaling with few false positives or false negatives. Characterization of hit genes uncovered novel components of several ciliary structures, including a protein complex that contains δ-tubulin and ε-tubulin and is required for centriole maintenance. The screen also provides an unbiased tool for classifying ciliopathies and showed that many congenital heart disorders are caused by loss of ciliary signaling. Collectively, our study enables a systematic analysis of ciliary function and of ciliopathies, and also defines a versatile platform for dissecting signaling pathways through CRISPR-based screening.

تاريخ نشر

2018

عنوان

UCSF

نام الکترونيکي

نوع ماده

[Article]

کد کاربرگه

275578

سطح دسترسي

a

تكميل شده

Y