{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T16:10:23Z","timestamp":1792167023948,"version":"4.4.0"},"reference-count":24,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2005,6,2]],"date-time":"2005-06-02T00:00:00Z","timestamp":1117670400000},"content-version":"vor","delay-in-days":4993,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Teratology"],"published-print":{"date-parts":[[1991,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The so\u2010called \u201cprune belly\u201d syndrome (PBS) consists of megalocystis, cryptorchidism, and marked abdominal distension; in addition to these findings, many other abnormalities are commonly present, rarely including severe anomalies of the leg. We report two such individuals, in whom PBS coexisted with abnormal development of the lower extremity. The first, a stillborn male fetus, was delivered at 21 weeks gestation; generalized hypoplasia of both legs was present, more marked distally than proximally, and more severe on the left. The second case, a liveborn male infant, was the second of dichorionic twins, delivered at 33 weeks gestation; this infant died at two hours from respiratory insufficiency due to pulmonary hypoplasia. There was severe hypoplasia of the right leg, with gangrenous necrosis of all tissues distal to the knee. Additional findings included a single right umbilical artery, and a small congenital cystic adenomatoid malformation of the right lung. The findings in these cases are compared to other similar cases in the literature, and possible mechanisms for the etiology and pathogenesis of maldevelopment of the leg in PBS are discussed.<\/jats:p>","DOI":"10.1002\/tera.1420440402","type":"journal-article","created":{"date-parts":[[2005,6,11]],"date-time":"2005-06-11T15:46:23Z","timestamp":1118504783000},"page":"365-371","source":"Crossref","is-referenced-by-count":10,"title":["Complexities of limb anomalies: The lower extremity in the \u201cprune belly\u201d phenotype"],"prefix":"10.1002","volume":"44","author":[{"given":"David R.","family":"Genest","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shirley G.","family":"Driscoll","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederick R.","family":"Bieber","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2005,6,2]]},"reference":[{"key":"e_1_2_1_2_1","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1982.03970370025005"},{"key":"e_1_2_1_3_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.54.8.614"},{"key":"e_1_2_1_4_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.1.5176.841"},{"key":"e_1_2_1_5_1","first-page":"19","article-title":"Lower limb deficiency and the urethral obstruction sequence","volume":"3","author":"Carey J. C.","year":"1982","journal-title":"Birth Defects: Original Article Series, Vol 18"},{"key":"e_1_2_1_6_1","doi-asserted-by":"crossref","first-page":"721","DOI":"10.1542\/peds.6.5.721","article-title":"Congenital deficiency of abdominal musculature with associated genito\u2010urinary abnormalities: A syndrome. Report of nine cases","volume":"6","author":"Eagle J. F.","year":"1950","journal-title":"Pediatrics"},{"key":"e_1_2_1_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0095-5108(18)30811-X"},{"key":"e_1_2_1_8_1","doi-asserted-by":"crossref","first-page":"707","DOI":"10.1016\/S0022-5347(17)41793-9","article-title":"The prune belly syndrome: A review of its etiology, defects, treatments, and prognosis","volume":"140","author":"Greskovich S. J.","year":"1988","journal-title":"J. Urol."},{"key":"e_1_2_1_9_1","first-page":"795","article-title":"Absent abdominal musculature, genitourinary anomalies and deficiency in the pelvic autonomic nervous system","volume":"86","author":"Henley W. L.","year":"1958","journal-title":"Am. J. Dis. Child."},{"key":"e_1_2_1_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(82)80343-0"},{"key":"e_1_2_1_11_1","first-page":"562","volume-title":"Smith's Recognizable Patterns of Human Malformation","author":"Jones J. 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L.","year":"1975"},{"key":"e_1_2_1_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/path.1711040104"},{"key":"e_1_2_1_18_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.78.3.451"},{"key":"e_1_2_1_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/ki.1981.198"},{"key":"e_1_2_1_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(74)80753-5"},{"key":"e_1_2_1_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9378(65)90588-0"},{"key":"e_1_2_1_22_1","doi-asserted-by":"crossref","first-page":"109","DOI":"10.2106\/00004623-197860010-00016","article-title":"Prune\u2010belly syndrome. A report of twelve cases and a review of the literature","volume":"60","author":"Tuch B. A.","year":"1978","journal-title":"J. Bone Joint Surg."},{"key":"e_1_2_1_23_1","first-page":"944","volume-title":"Congenital Malformations: Notes and Comments","author":"Warkany J.","year":"1977"},{"key":"e_1_2_1_24_1","first-page":"17","volume-title":"Pathology Annual","author":"Wigger H. 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M.","year":"1988"}],"container-title":["Teratology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Ftera.1420440402","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/tera.1420440402","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,23]],"date-time":"2023-10-23T00:16:49Z","timestamp":1698020209000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/tera.1420440402"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[1991,10]]},"references-count":24,"journal-issue":{"issue":"4","published-print":{"date-parts":[[1991,10]]}},"alternative-id":["10.1002\/tera.1420440402"],"URL":"https:\/\/doi.org\/10.1002\/tera.1420440402","archive":["Portico"],"relation":{},"ISSN":["0040-3709","1096-9926"],"issn-type":[{"value":"0040-3709","type":"print"},{"value":"1096-9926","type":"electronic"}],"subject":[],"published":{"date-parts":[[1991,10]]}}}