{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T23:29:38Z","timestamp":1777937378998,"version":"3.51.4"},"reference-count":11,"publisher":"SAGE Publications","issue":"4","license":[{"start":{"date-parts":[[2017,5,12]],"date-time":"2017-05-12T00:00:00Z","timestamp":1494547200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Pediatr Dev Pathol"],"published-print":{"date-parts":[[2018,7]]},"abstract":"<jats:p>The fatal infantile neuromuscular type is the most severe form of glycogen storage disease type IV (GSD IV). We report a case of a 22-day-old female neonate born at 34 weeks gestation with polyhyramnios, fetal hydrops, and severe hypotonia. Placental examination revealed numerous periodic acid schiff-positive diastase-resistant polyglucosan bodies in the cytoplasm of extravillous trophoblast predominantly in the placental basal plate. Muscle biopsy and autopsy findings supported a diagnosis of neuromuscular-type glycogen storage disease type IV with extensive involvement of skeletal muscle, heart, and liver. The diagnosis was confirmed by molecular genetic testing. We could only find 1 prior report in the English literature that describes placental pathological changes. Our findings suggest that placental examination can be a useful adjunct for early diagnosis, as placentas are often received for pathological examination shortly after birth and usually before a diagnostic muscle biopsy can be performed. Pathologists need to be aware of characteristic placental features.<\/jats:p>","DOI":"10.1177\/1093526617707852","type":"journal-article","created":{"date-parts":[[2017,12,31]],"date-time":"2017-12-31T04:29:34Z","timestamp":1514694574000},"page":"423-427","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":5,"title":["Polyglucosan Bodies in Placental Extravillious Trophoblast for the Diagnosis of Fatal Perinatal Neuromuscular-type Glycogen Storage Disease Type IV"],"prefix":"10.1177","volume":"21","author":[{"given":"Weiming","family":"Yu","sequence":"first","affiliation":[{"name":"Departments of Pathology & Laboratory Medicine and Paediatrics, University of Calgary Cumming School of Medicine and Calgary Laboratory Services, Calgary, Alberta, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marie-Anne","family":"Brundler","sequence":"additional","affiliation":[{"name":"Departments of Pathology & Laboratory Medicine and Paediatrics, University of Calgary Cumming School of Medicine and Calgary Laboratory Services, Calgary, Alberta, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"suffix":"Jr.","given":"James R","family":"Wright","sequence":"additional","affiliation":[{"name":"Departments of Pathology & Laboratory Medicine and Paediatrics, University of Calgary Cumming School of Medicine and Calgary Laboratory Services, Calgary, Alberta, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2017,5,12]]},"reference":[{"key":"bibr1-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2011.10.001"},{"key":"bibr2-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2010.07.275"},{"key":"bibr3-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1016\/j.placenta.2008.01.005"},{"key":"bibr4-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9378(85)90551-4"},{"key":"bibr5-1093526617707852","unstructured":"Benirschke K, Kaufmann P, Baergen R. Pathology of the Human Placenta, 5th ed. New York, NY: Springer, 2006, pp. 577\u2013583."},{"key":"bibr6-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-4419-7494-5"},{"key":"bibr7-1093526617707852","unstructured":"Fox H, Sebire NJ. Pathology of the Placenta, 3rd ed. Philadelphia, PA: Saunders Elsevier, 2007, pp. 257\u2013262."},{"key":"bibr8-1093526617707852","volume-title":"Diagnostic Pathology: Placenta","author":"Heerema-McKenney A","year":"2015"},{"key":"bibr9-1093526617707852","unstructured":"Joshi VV. Handbook of Placental Pathology, New York, NY: Igaku-Shoin, 1994, pp. 110."},{"key":"bibr10-1093526617707852","doi-asserted-by":"crossref","unstructured":"Kraus FT, Redline RW, Gersell DJ, Nelson DM, Dicke JM. Placental Pathology. Atlas of Nontumor Pathology, First Series Fascicle 3, Washington, DC: American Registry of Pathology, 2004, pp. 65\u201366.","DOI":"10.55418\/1881041891"},{"key":"bibr11-1093526617707852","doi-asserted-by":"publisher","DOI":"10.1016\/S0143-4004(05)80215-2"}],"container-title":["Pediatric and Developmental Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1093526617707852","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/1093526617707852","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1093526617707852","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,2]],"date-time":"2026-05-02T04:04:07Z","timestamp":1777694647000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/1093526617707852"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,5,12]]},"references-count":11,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2018,7]]}},"alternative-id":["10.1177\/1093526617707852"],"URL":"https:\/\/doi.org\/10.1177\/1093526617707852","relation":{},"ISSN":["1093-5266","1615-5742"],"issn-type":[{"value":"1093-5266","type":"print"},{"value":"1615-5742","type":"electronic"}],"subject":[],"published":{"date-parts":[[2017,5,12]]}}}