{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T14:09:12Z","timestamp":1773756552415,"version":"3.50.1"},"reference-count":32,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2018,10,30]],"date-time":"2018-10-30T00:00:00Z","timestamp":1540857600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Endocrino Diabet &amp; Metabol"],"published-print":{"date-parts":[[2019,1]]},"abstract":"<jats:title>Summary<\/jats:title><jats:sec><jats:title>Context<\/jats:title><jats:p>Cryptorchidism is common in Prader\u2010Willi syndrome (PWS) males, but the testicular histology in childhood remains uncertain. The association between testicular histology and long\u2010term gonadal function in PWS males is also unknown.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objectives<\/jats:title><jats:p>To evaluate the relationship between testicular histology in childhood and long\u2010term gonadal function in PWS males.<\/jats:p><\/jats:sec><jats:sec><jats:title>Patients and Methods<\/jats:title><jats:p>Forty men with PWS were assessed longitudinally at our institute over the past 24\u00a0years. Clinical examinations and blood tests for LH, FSH and testosterone levels were compared with normal reference values. Tissue specimens were collected during orchiopexy and analyzed based on Nistal categories.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of nine testes available for pathological assessments, two showed favourable histology (Nistal I) and seven showed unfavourable histology (Nistal II or III). Of five postpubertal males with histology available, four reached puberty spontaneously, but only one reached Tanner stage 5. In a male with favourable histology, LH and FSH were high, but testosterone was normal, though below the average of the reference range. In three males with unfavourable histology, LH was normal, but FSH was highly elevated, and testosterone was at the lower limit of normal. One patient took hCG treatment to induce puberty; this patient showed favourable histology, but LH, FSH and testosterone were not elevated in adolescence.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Testicular histology of PWS men in childhood varies from normal to Sertoli Cell\u2010Only Syndrome. Regardless of the testicular histology in childhood, hypogonadism in PWS adults arises as a consequence of primary testicular dysfunction with highly elevated FSH and insufficient testosterone levels.<\/jats:p><\/jats:sec>","DOI":"10.1002\/edm2.49","type":"journal-article","created":{"date-parts":[[2018,10,31]],"date-time":"2018-10-31T03:19:11Z","timestamp":1540955951000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Gonadal function and testicular histology in males with Prader\u2010Willi syndrome"],"prefix":"10.1002","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2460-0313","authenticated-orcid":false,"given":"Satoko","family":"Matsuyama","sequence":"first","affiliation":[{"name":"Department of Urology Osaka Women\u2019s and Children\u2019s Hospital Osaka Japan"},{"name":"Department of Integrative Cancer Therapy and Urology Kanazawa University Graduate School of Medical Science Kanazawa 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