{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T12:51:50Z","timestamp":1756385510966},"reference-count":0,"publisher":"Georg Thieme Verlag KG","issue":"04","license":[{"start":{"date-parts":[[2017,12,29]],"date-time":"2017-12-29T00:00:00Z","timestamp":1514505600000},"content-version":"vor","delay-in-days":89,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["AJP Rep"],"published-print":{"date-parts":[[2017,10]]},"abstract":"<jats:p>\n            Background\u2003Congenital diaphragmatic hernia (CDH) is fatal in severe cases of pulmonary hypoplasia. We experienced a fatal case of pulmonary hypoplasia due to CDH, thoracic myelomeningocele (MMC), and thoracic dysplasia. This constellation of anomalies has not been previously reported.<\/jats:p><jats:p>\n            Case Report\u2003A male infant with a prenatal diagnosis of thoracic MMC with severe hydrocephalus and scoliosis was born at 36 weeks of gestation. CDH was found after birth and the patient died of respiratory failure due to pulmonary hypoplasia and persistent pulmonary hypertension of the newborn at 30 hours of age despite neonatal intensive care. An autopsy revealed a left CDH without herniation of the liver or stomach into the thoracic cavity, severe hydrocephalus, Chiari malformation type II, MMC with spina bifida from Th4 to Th12, hemivertebrae, fused ribs, deformities of the thoracic cage and legs, short trunk, and agenesis of the left kidney.<\/jats:p><jats:p>\n            Conclusion\u2003We speculate that two factors may be associated with the severe pulmonary hypoplasia: decreased thoracic space due to the herniation of visceral organs caused by CDH and thoracic dysplasia due to skeletal deformity and severe scoliosis.<\/jats:p>","DOI":"10.1055\/s-0037-1615791","type":"journal-article","created":{"date-parts":[[2017,12,29]],"date-time":"2017-12-29T23:40:47Z","timestamp":1514590847000},"page":"e234-e237","source":"Crossref","is-referenced-by-count":2,"title":["A Case of Fatal Pulmonary Hypoplasia with Congenital Diaphragmatic Hernia, Thoracic Myelomeningocele, and Thoracic Dysplasia"],"prefix":"10.1055","volume":"07","author":[{"given":"Ai","family":"Ito","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"},{"name":"Department of Postgraduate Education and Training, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hideshi","family":"Fujinaga","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"},{"name":"Division of Neonatology, Department of Pediatrics, Nishisaitama-chuo National Hospital, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sachiko","family":"Matsui","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kumiko","family":"Tago","sequence":"additional","affiliation":[{"name":"Department of Pathology, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuka","family":"Iwasaki","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shuhei","family":"Fujino","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Junko","family":"Nagasawa","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shoichiro","family":"Amari","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masao","family":"Kaneshige","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuka","family":"Wada","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shigehiro","family":"Takahashi","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiko","family":"Tsukamoto","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Osamu","family":"Miyazaki","sequence":"additional","affiliation":[{"name":"Department of Radiology, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takako","family":"Yoshioka","sequence":"additional","affiliation":[{"name":"Department of Pathology, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akira","family":"Ishiguro","sequence":"additional","affiliation":[{"name":"Department of Postgraduate Education and Training, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yushi","family":"Ito","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"194","published-online":{"date-parts":[[2017,12,29]]},"container-title":["American Journal of Perinatology Reports"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.thieme-connect.de\/products\/ejournals\/pdf\/10.1055\/s-0037-1615791.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2019,5,6]],"date-time":"2019-05-06T19:35:57Z","timestamp":1557171357000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.thieme-connect.de\/DOI\/DOI?10.1055\/s-0037-1615791"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,10]]},"references-count":0,"journal-issue":{"issue":"04","published-online":{"date-parts":[[2017,11,13]]},"published-print":{"date-parts":[[2017,10]]}},"URL":"https:\/\/doi.org\/10.1055\/s-0037-1615791","relation":{},"ISSN":["2157-6998","2157-7005"],"issn-type":[{"value":"2157-6998","type":"print"},{"value":"2157-7005","type":"electronic"}],"subject":[],"published":{"date-parts":[[2017,10]]}}}