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Brief Report

Somatic Mutations of lats2 Cause Peripheral Nerve Sheath Tumors in Zebrafish

1
Department of Cell Biology, Neurobiology and Anatomy, Medical College of Wisconsin, Milwaukee, WI 53226, USA
2
Department of Pediatrics, Medical College of Wisconsin, Milwaukee, WI 53226, USA
*
Author to whom correspondence should be addressed.
Cells 2019, 8(9), 972; https://doi.org/10.3390/cells8090972
Received: 25 June 2019 / Revised: 15 August 2019 / Accepted: 22 August 2019 / Published: 25 August 2019
(This article belongs to the Special Issue Disease and the Hippo Pathway: Cellular and Molecular Mechanisms)
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. View Full-Text
Keywords: Lats2; Hippo; Yap; Taz; peripheral nerve sheath tumor; schwannoma; zebrafish Lats2; Hippo; Yap; Taz; peripheral nerve sheath tumor; schwannoma; zebrafish
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MDPI and ACS Style

Brandt, Z.J.; North, P.N.; Link, B.A. Somatic Mutations of lats2 Cause Peripheral Nerve Sheath Tumors in Zebrafish. Cells 2019, 8, 972. https://doi.org/10.3390/cells8090972

AMA Style

Brandt ZJ, North PN, Link BA. Somatic Mutations of lats2 Cause Peripheral Nerve Sheath Tumors in Zebrafish. Cells. 2019; 8(9):972. https://doi.org/10.3390/cells8090972

Chicago/Turabian Style

Brandt, Zachary J., Paula N. North, and Brian A. Link. 2019. "Somatic Mutations of lats2 Cause Peripheral Nerve Sheath Tumors in Zebrafish" Cells 8, no. 9: 972. https://doi.org/10.3390/cells8090972

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