{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T08:34:18Z","timestamp":1782462858368,"version":"3.54.5"},"reference-count":97,"publisher":"The Company of Biologists","issue":"23","license":[{"start":{"date-parts":[[2022,11,30]],"date-time":"2022-11-30T00:00:00Z","timestamp":1669766400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","award":["1R01HD100985"],"award-info":[{"award-number":["1R01HD100985"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,12,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Undescended testis (UDT) affects 6% of male births. Despite surgical correction, some men with unilateral UDT may experience infertility with the contralateral descended testis (CDT) showing no A-dark spermatogonia. To improve our understanding of the etiology of infertility in UDT, we generated a novel murine model of left unilateral UDT. Gubernaculum-specific Wnt4 knockout (KO) mice (Wnt4-cKO) were generated using retinoic acid receptor \u03b22-cre mice and were found to have a smaller left-unilateral UDT. Wnt4-cKO mice with abdominal UDT had an increase in serum follicle-stimulating hormone and luteinizing hormone and an absence of germ cells in the undescended testicle. Wnt4-cKO mice with inguinal UDT had normal hormonal profiles, and 50% of these mice had no sperm in the left epididymis. Wnt4-cKO mice had fertility defects and produced 52% fewer litters and 78% fewer pups than control mice. Wnt4-cKO testes demonstrated increased expression of estrogen receptor \u03b1 and SOX9, upregulation of female gonadal genes, and a decrease in male gonadal genes in both CDT and UDT. Several WNT4 variants were identified in boys with UDT. The presence of UDT and fertility defects in Wnt4-cKO mice highlights the crucial role of WNT4 in testicular development.<\/jats:p>","DOI":"10.1242\/dev.201093","type":"journal-article","created":{"date-parts":[[2022,11,30]],"date-time":"2022-11-30T09:42:49Z","timestamp":1669801369000},"source":"Crossref","is-referenced-by-count":10,"title":["Loss of WNT4 in the gubernaculum causes unilateral cryptorchidism and fertility defects"],"prefix":"10.1242","volume":"149","author":[{"given":"Abhishek","family":"Seth","sequence":"first","affiliation":[{"name":"Baylor College of Medicine 1 Scott Department of Urology , , Houston, TX 77030 , USA"},{"name":"Center for Reproductive Medicine, Baylor College of Medicine 2 , Houston, TX 77030 , USA"},{"name":"Department of Surgery, Nemours Children's Health 3 , Orlando, FL 32827 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juan C.","family":"Bournat","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 Scott Department of Urology , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olga","family":"Medina-Martinez","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 Scott Department of Urology , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Armando","family":"Rivera","sequence":"additional","affiliation":[{"name":"Center for Reproductive Medicine, Baylor College of Medicine 2 , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Moore","sequence":"additional","affiliation":[{"name":"Center for Reproductive Medicine, Baylor College of Medicine 2 , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hunter","family":"Flores","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 Scott Department of Urology , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jill A.","family":"Rosenfeld","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 4 Department of Molecular and Human Genetics , , Houston, TX 77030 , USA"},{"name":"Baylor College of Medicine 5 Baylor Genetics Laboratories , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liya","family":"Hu","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 6 Department of Biochemistry and Molecular Biology , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9203-6331","authenticated-orcid":false,"given":"Carolina J.","family":"Jorgez","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 Scott Department of Urology , , Houston, TX 77030 , USA"},{"name":"Center for Reproductive Medicine, Baylor College of Medicine 2 , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2022,11,30]]},"reference":[{"key":"2022121615301885800_DEV201093C1","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1038\/nmeth0410-248","article-title":"A method and server for predicting damaging missense mutations","volume":"7","author":"Adzhubei","year":"2010","journal-title":"Nat. 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