{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T13:06:59Z","timestamp":1787663219850,"version":"build-2736575974"},"reference-count":61,"publisher":"The Company of Biologists","issue":"4","license":[{"start":{"date-parts":[[2024,2,13]],"date-time":"2024-02-13T00:00:00Z","timestamp":1707782400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["K99\/R00"],"award-info":[{"award-number":["K99\/R00"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"crossref","award":["1K99 GM145224-01"],"award-info":[{"award-number":["1K99 GM145224-01"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100007698","name":"University of Florida","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007698","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,2,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>The mechanosensitive PIEZO channel family has been linked to over 26 disorders and diseases. Although progress has been made in understanding these channels at the structural and functional levels, the underlying mechanisms of PIEZO-associated diseases remain elusive. In this study, we engineered four PIEZO-based disease models using CRISPR\/Cas9 gene editing. We performed an unbiased chemical mutagen-based genetic suppressor screen to identify putative suppressors of a conserved gain-of-function variant pezo-1[R2405P] that in human PIEZO2 causes distal arthrogryposis type 5 (DA5; p. R2718P). Electrophysiological analyses indicate that pezo-1(R2405P) is a gain-of-function allele. Using genomic mapping and whole-genome sequencing approaches, we identified a candidate suppressor allele in the C. elegans gene gex-3. This gene is an ortholog of human NCKAP1 (NCK-associated protein 1), a subunit of the Wiskott-Aldrich syndrome protein (WASP)-verprolin homologous protein (WAVE\/SCAR) complex, which regulates F-actin polymerization. Depletion of gex-3 by RNAi, or with the suppressor allele gex-3(av259[L353F]), significantly increased brood size and ovulation rate, as well as alleviating the crushed oocyte phenotype of the pezo-1(R2405P) mutant. Expression of GEX-3 in the soma is required to rescue the brood size defects in pezo-1(R2405P) animals. Actin organization and orientation were disrupted and distorted in the pezo-1 mutants. Mutation of gex-3(L353F) partially alleviated these defects. The identification of gex-3 as a suppressor of the pathogenic variant pezo-1(R2405P) suggests that the PIEZO coordinates with the cytoskeleton regulator to maintain the F-actin network and provides insight into the molecular mechanisms of DA5 and other PIEZO-associated diseases.<\/jats:p>","DOI":"10.1242\/dev.202214","type":"journal-article","created":{"date-parts":[[2024,2,13]],"date-time":"2024-02-13T11:27:50Z","timestamp":1707823670000},"source":"Crossref","is-referenced-by-count":7,"title":["A mutation in F-actin polymerization factor suppresses the distal arthrogryposis type 5 PIEZO2 pathogenic variant in\n                    <i>Caenorhabditis elegans<\/i>"],"prefix":"10.1242","volume":"151","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8179-8162","authenticated-orcid":false,"given":"Xiaofei","family":"Bai","sequence":"first","affiliation":[{"name":"University of Florida 1 Department of Biology , , Gainesville, FL 32610 , USA"},{"name":"Genetics Institute, University of Florida 2 , Gainesville, FL 32610 , USA"},{"name":"National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health 3 , Bethesda, MD 20892 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3517-5973","authenticated-orcid":false,"given":"Harold E.","family":"Smith","sequence":"additional","affiliation":[{"name":"National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health 3 , Bethesda, MD 20892 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9855-7592","authenticated-orcid":false,"given":"Luis O.","family":"Romero","sequence":"additional","affiliation":[{"name":"College of Medicine, University of Tennessee Health Science Center 4 Department of Physiology , , Memphis, TN 38103 , USA"},{"name":"College of Graduate Health Sciences 5 Integrated Biomedical Sciences Graduate Program , , Memphis, TN 38163 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9797-6402","authenticated-orcid":false,"given":"Briar","family":"Bell","sequence":"additional","affiliation":[{"name":"College of Medicine, University of Tennessee Health Science Center 4 Department of Physiology , , Memphis, TN 38103 , USA"},{"name":"College of Graduate Health Sciences 5 Integrated Biomedical Sciences Graduate Program , , Memphis, TN 38163 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8494-1534","authenticated-orcid":false,"given":"Valeria","family":"V\u00e1squez","sequence":"additional","affiliation":[{"name":"College of Medicine, University of Tennessee Health Science Center 4 Department of Physiology , , Memphis, TN 38103 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8599-2031","authenticated-orcid":false,"given":"Andy","family":"Golden","sequence":"additional","affiliation":[{"name":"National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health 3 , Bethesda, MD 20892 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,2,13]]},"reference":[{"key":"2024021316254995400_DEV202214C1","doi-asserted-by":"publisher","first-page":"1884","DOI":"10.1038\/ncomms2899","article-title":"Dehydrated hereditary stomatocytosis linked to gain-of-function mutations in mechanically activated PIEZO1 ion channels","volume":"4","author":"Albuisson","year":"2013","journal-title":"Nat. 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