{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,6]],"date-time":"2026-04-06T15:04:06Z","timestamp":1775487846512,"version":"3.50.1"},"reference-count":9,"publisher":"BMJ","issue":"7","license":[{"start":{"date-parts":[[2025,7,8]],"date-time":"2025-07-08T00:00:00Z","timestamp":1751932800000},"content-version":"unspecified","delay-in-days":7,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,6,15]]},"published-print":{"date-parts":[[2025,7]]},"abstract":"<jats:p>Worster-Drought syndrome (WDS), or congenital suprabulbar paresis, is characterised by congenital dysarthria, dysphagia and other pseudobulbar paresis without structural abnormalities around the Sylvian fissure on imaging. This rare syndrome is challenging to diagnose, particularly in preterm infants. This report describes a low-birth-weight female infant with WDS who had no sucking reflex from birth, airway obstruction due to saliva retention and muscle rigidity, who was diagnosed with the syndrome at a postconceptional age (PCA) of 1\u2009month. She was discharged with only home oxygen therapy as respiratory support at a PCA of 3 months after gradual improvement in her clinical symptoms. Diagnosis of WDS is difficult in the early postnatal period in preterm cases owing to prematurity but should be suspected when bulbar palsy, including absence of the sucking reflex, persistent dysphagia and obstructed breathing, persists beyond a PCA of 40 weeks and when muscle stiffness is present.<\/jats:p>","DOI":"10.1136\/bcr-2024-263524","type":"journal-article","created":{"date-parts":[[2025,8,11]],"date-time":"2025-08-11T04:00:22Z","timestamp":1754884822000},"page":"e263524","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Worster-Drought syndrome with progressive symptomatic improvement in early infancy"],"prefix":"10.1136","volume":"18","author":[{"given":"Kota","family":"Inoue","sequence":"first","affiliation":[{"name":"Pediatrics, Kagawa University Faculty of Medicine Graduate School of Medicine, Kita-gun, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shinji","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Pediatrics, Kagawa University Faculty of Medicine Graduate School of Medicine, Kita-gun, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kosuke","family":"Koyano","sequence":"additional","affiliation":[{"name":"Maternal Perinatal Center, Kagawa University Hospital, Kita-gun, Kagawa, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashi","family":"Kusaka","sequence":"additional","affiliation":[{"name":"Pediatrics, Kagawa University Faculty of Medicine Graduate School of Medicine, Kita-gun, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2025,7,8]]},"reference":[{"key":"2025082712051536000_18.7.e263524.1","doi-asserted-by":"crossref","first-page":"453","DOI":"10.1017\/S0022215100053184","article-title":"Congenital suprabulbar paresis","volume":"70","author":"Worster-Drought","year":"1956","journal-title":"J Laryngol Otol"},{"key":"2025082712051536000_18.7.e263524.2","first-page":"1","article-title":"Suprabulbar Paresis. Congenital suprabulbar paresis and its differential diagnosis, with special reference to acquired suprabulbar paresis","volume":"30","author":"Worster-Drought","year":"1974","journal-title":"Dev Med Child Neurol Suppl"},{"key":"2025082712051536000_18.7.e263524.3","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2009.03475.x"},{"key":"2025082712051536000_18.7.e263524.4","doi-asserted-by":"publisher","DOI":"10.1016\/S0887-8994(03)00006-7"},{"key":"2025082712051536000_18.7.e263524.5","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/123.10.2160"},{"key":"2025082712051536000_18.7.e263524.6","first-page":"389","article-title":"Foix-Chavany-Marie syndrome due to unilateral anterior opercular infarction with leukoaraiosis","volume":"34","author":"Rivas","year":"2021","journal-title":"Proc (Bayl Univ Med Cent)"},{"key":"2025082712051536000_18.7.e263524.7","doi-asserted-by":"crossref","first-page":"279","DOI":"10.1111\/ncn3.12302","article-title":"A case of Foix\u2013Chavany\u2013Marie syndrome due to bilateral corona radiata infarcts","volume":"7","author":"Rossi","year":"2019","journal-title":"Neurol Clin Neurosci"},{"key":"2025082712051536000_18.7.e263524.8","doi-asserted-by":"crossref","first-page":"17","DOI":"10.4103\/jpn.JPN_122_20","article-title":"Profile of Worster Drought Syndrome (WDS): unrecognized subtype of cerebral palsy\u2014from tertiary care center in South India","volume":"17","author":"Patil","year":"2022","journal-title":"J Pediatr Neurosci"},{"key":"2025082712051536000_18.7.e263524.9","doi-asserted-by":"crossref","unstructured":"McMillan HJ , Holahan A-L , Richer J . Worster-Drought Syndrome Associated With LINS Mutations. Child Neurol Open 2018;5. doi:10.1177\/2329048X18791083","DOI":"10.1177\/2329048X18791083"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2024-263524","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T19:05:22Z","timestamp":1756321522000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2024-263524"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,7]]},"references-count":9,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2025,7,8]]},"published-print":{"date-parts":[[2025,7]]}},"alternative-id":["10.1136\/bcr-2024-263524"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2024-263524","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,7]]}}}