{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T14:51:43Z","timestamp":1791384703903,"version":"4.3.3"},"reference-count":41,"publisher":"SAGE Publications","issue":"4","license":[{"start":{"date-parts":[[2023,1,22]],"date-time":"2023-01-22T00:00:00Z","timestamp":1674345600000},"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":["The Cleft Palate Craniofacial Journal"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:p>\n                    <jats:bold>Objective:<\/jats:bold>\n                    This review was conducted to define the natural history of unoperated Beckwith-Wiedemann syndrome (BWS) macroglossia and the effect of tongue reduction surgery upon breathing, eating, speaking and dentoskeletal development in individuals having BWS.\n                    <jats:bold>Design:<\/jats:bold>\n                    This is a retrospective study of medical records. Setting: All patients were evaluated and treated in one of two Children's Hospitals by an ACPA approved Craniofacial Team. Patients\/Participants: Medical records were reviewed of 526 individuals having a diagnosis of BWS and evaluated in-person by a single craniofacial surgeon between 1986 and 2014 in conjunction with a series of multi-disciplinary craniofacial team colleagues. 28 individuals were excluded having had multiple tongue reductions elsewhere. 498 individuals comprise the \u201cpre tongue-reduction group\u201d. The \u201cpost tongue-reduction group\u201d consists of 391 individuals who underwent surgical tongue reduction by one surgeon using one technique between 1986 and 2014. Main Outcome Measures: The primary outcome measure was change in anterior dental occlusion following tongue reduction surgery. Tongue reduction surgery was performed on the assumption that it would improve dentoskeletal relationships. Secondary outcome measures were: breathing, feeding\/swallowing, and speech.\n                    <jats:bold>Results:<\/jats:bold>\n                    A significant difference (p&lt;0.001) over time between the two groups was found with less anterior occlusal abnormality in the tongue reduction group. Tongue reduction surgery had no mortality and minimal morbidity for breathing, feeding\/swallowing, and speech and can ameliorate obstructive sleep apnea.\n                    <jats:bold>Conclusions:<\/jats:bold>\n                    Surgical tongue reduction for BWS macroglossia is recommended for the infant or child in primary dentition with a grossly abnormal anterior tooth\/jaw relationship and\/or obstructive sleep apnea.\n                  <\/jats:p>","DOI":"10.1177\/10556656221148900","type":"journal-article","created":{"date-parts":[[2023,1,23]],"date-time":"2023-01-23T02:51:45Z","timestamp":1674442305000},"page":"599-609","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":10,"title":["Beckwith-Widemann Macroglossia: The Role of Surgical Tongue Reduction"],"prefix":"10.1177","volume":"61","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7766-2131","authenticated-orcid":false,"given":"Jeffrey L","family":"Marsh","sequence":"first","affiliation":[{"name":"4623 Pershing Place, Saint Louis, MO, 63108, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chad A","family":"Perlyn","sequence":"additional","affiliation":[{"name":"Plastic Surgery, Nicklaus Children's Hospital, 3100 SW 62nd Avenue Miami, Miami, FL, 33155-3009, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2023,1,22]]},"reference":[{"key":"e_1_3_3_2_2","unstructured":"Beckwith JB. Extreme cytomegaly of the adrenal fetal cortex omphalocele hyperplasia of the kidneys and pancreas and Leydig-cell hyperplasia another syndrome? Western Society for Pediatric Research Los Angeles CA. 1963."},{"key":"e_1_3_3_3_2","first-page":"188","article-title":"Macroglossia, omphalocele, adrenal cytomegaly, gigantism, and hyperplastic visceromegaly","author":"Beckwith JB","year":"1969","unstructured":"Beckwith JB. Macroglossia, omphalocele, adrenal cytomegaly, gigantism, and hyperplastic visceromegaly. Birth Defects Orig Artic Ser 2. 1969:188-196.","journal-title":"Birth Defects Orig Artic Ser 2"},{"key":"e_1_3_3_4_2","first-page":"223","article-title":"Complexe malformatif familial avec hernie ombilicale et macroglossie - un \"syndrome nouveua\"?","volume":"13","author":"Wiedemann HR","year":"1964","unstructured":"Wiedemann HR. Complexe malformatif familial avec hernie ombilicale et macroglossie - un \"syndrome nouveua\"? 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