{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T19:51:21Z","timestamp":1780343481898,"version":"3.54.1"},"reference-count":26,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2025,12,9]],"date-time":"2025-12-09T00:00:00Z","timestamp":1765238400000},"content-version":"vor","delay-in-days":8,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Case Reports"],"published-print":{"date-parts":[[2025,12]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Congenital syngnathia, the rare fusion of the maxilla and mandible, poses significant feeding and respiratory challenges. Its management is complicated by rarity and potential syndromic associations. We present a 1\u2010week\u2010old female neonate who presented with left jaw deviation, respiratory distress (SpO\n                    <jats:sub>2<\/jats:sub>\n                    69%, tachypnea), and fever. Examination revealed near\u2010complete inability to open the mouth except for a small right paramedical gap. A CT scan confirmed left maxilla\u2010mandibular bony fusion (Laster type 2b, Dawson type 2b), mandibular hypoplasia, semilobar holoprosencephaly, corpus callosal dysgenesis, a dorsal cyst, and microcephaly. Concurrent neonatal sepsis was diagnosed. Management included antibiotics, NG tube feeding, and oxygen support. Surgical intervention was deferred due to critical instability. Despite supportive care, the infant's condition deteriorated, resulting in fatal respiratory failure. This case underscores the lethal potential of congenital syngnathia when compounded by severe CNS malformations. The CNS anomalies induced hypoventilation and aspiration, making surgery nonviable and leading to sepsis and respiratory failure. The absence of antenatal care prevented early detection and tertiary delivery planning. Prompt neuroimaging is crucial, and prognosis is heavily dictated by CNS comorbidities, necessitating consideration of palliative prioritization in resource\u2010limited settings.\n                  <\/jats:p>","DOI":"10.1002\/ccr3.71659","type":"journal-article","created":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T03:57:59Z","timestamp":1765339079000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Congenital Syngnathia With Holoprosencephaly: A Case Report of a Fatal Presentation in a Resource\u2010Limited Setting"],"prefix":"10.1002","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-2750-2121","authenticated-orcid":false,"given":"Asteway M.","family":"Haile","sequence":"first","affiliation":[{"name":"School of Medicine, College of Health Sciences Addis Ababa University  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-4647-111X","authenticated-orcid":false,"given":"Biruk T.","family":"Mengistie","sequence":"additional","affiliation":[{"name":"School of Medicine, College of Health Sciences Addis Ababa University  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tinsae Y.","family":"Tamiru","sequence":"additional","affiliation":[{"name":"School of Medicine, College of Health Sciences Addis Ababa University  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-0061-6458","authenticated-orcid":false,"given":"Chernet T.","family":"Mengistie","sequence":"additional","affiliation":[{"name":"School of Medicine, College of Health Sciences Addis Ababa University  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dawit G.","family":"Abebe","sequence":"additional","affiliation":[{"name":"Pediatrics Department ALERT Comprehensive Specialized Hospital  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bezawit M.","family":"Haile","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Child Health, College of Health Sciences Addis Ababa University  Addis Ababa Ethiopia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,12,9]]},"reference":[{"key":"e_1_2_14_2_1","first-page":"201","article-title":"Congenital Deformities of the Face","volume":"61","author":"Davis W. 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