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Somatic Mutations of lats2 Cause Peripheral Nerve Sheath Tumors in Zebrafish. Cells 2019 Aug 25;8(9)



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Pubmed Central ID




Scopus ID

2-s2.0-85084889096 (requires institutional sign-in at Scopus site)   9 Citations


The cellular signaling pathways underlying peripheral nerve sheath tumor (PNST) formation are poorly understood. Hippo signaling has been recently implicated in the biology of various cancers, and is thought to function downstream of mutations in the known PNST driver, NF2. Utilizing CRISPR-Cas9 gene editing, we targeted the canonical Hippo signaling kinase Lats2. We show that, while germline deletion leads to early lethality, targeted somatic mutations of zebrafish lats2 leads to peripheral nerve sheath tumor formation. These peripheral nerve sheath tumors exhibit high levels of Hippo effectors Yap and Taz, suggesting that dysregulation of these transcriptional co-factors drives PNST formation in this model. These data indicate that somatic lats2 deletion in zebrafish can serve as a powerful experimental platform to probe the mechanisms of PNST formation and progression.

Author List

Brandt ZJ, North PN, Link BA


Brian A. Link PhD Professor in the Cell Biology, Neurobiology and Anatomy department at Medical College of Wisconsin
Paula E. North MD, PhD Professor in the Pathology department at Medical College of Wisconsin

MESH terms used to index this publication - Major topics in bold

CRISPR-Cas Systems
Cell Proliferation
Gene Editing
Neoplasms, Experimental
Nerve Sheath Neoplasms
Signal Transduction
Zebrafish Proteins