{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T06:02:00Z","timestamp":1788242520293,"version":"build-2803163510"},"reference-count":22,"publisher":"American Dental Society of Anesthesiology (ADSA)","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,12]]},"abstract":"<jats:p>Williams syndrome (WS) is a genetic disorder marked by intellectual disability, distinctive craniofacial features, dental abnormalities, and congenital heart defects, particularly supravalvular aortic stenosis\u2014a narrowing above the aortic valve. Patients with WS are at increased risk of perioperative sudden cardiac death, necessitating thorough cardiovascular assessment. Additionally, craniofacial anomalies such as micrognathia and retrognathia may lead to difficult airway management and often require awake intubation. However, awake intubation and hospitalization can be distressing for patients with WS due to their developmental challenges. These facts indicate the importance of identifying during the preoperative assessment for ambulatory general anesthesia those at low risk for cardiovascular complications and airway difficulties. This case report details the successful anesthetic management of a 9-year-old boy with WS undergoing ambulatory general anesthesia for dental treatment by maintaining a balance between myocardial oxygen supply and demand to minimize cardiovascular fluctuations and by paying careful attention to managing his airway. Our case highlights the feasibility and safety of ambulatory general anesthesia for patients with WS, minimizing hospitalization stress and ensuring patient safety.<\/jats:p>","DOI":"10.2344\/24-0032","type":"journal-article","created":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T11:10:04Z","timestamp":1765365004000},"page":"241-245","source":"Crossref","is-referenced-by-count":1,"title":["Ambulatory General Anesthesia for Dental Treatment in a Patient With Williams Syndrome and Supravalvular Aortic Restenosis: A Case Report"],"prefix":"10.2344","volume":"72","author":[{"given":"Yukako","family":"Tsutsui","sequence":"first","affiliation":[{"name":"Department of Dental Anesthesiology, The Nippon Dental University School of Life Dentistry at Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hitomi","family":"Izumikawa","sequence":"additional","affiliation":[{"name":"Dental Department, Kashiwa City Municipal Medical Treatment Center, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miyuki","family":"Itakura","sequence":"additional","affiliation":[{"name":"Dental Department, Kashiwa City Municipal Medical Treatment Center, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katsuhisa","family":"Sunada","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, The Nippon Dental University School of Life Dentistry at Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1191","reference":[{"key":"i1878-7177-72-4-241-b01","series-title":"Circulation","first-page":"1311","article-title":"Supravalvular aortic stenosis","volume":"24","author":"Williams JCP,","year":"1961","unstructured":"Williams JCP,\nBarratt-Boyes BG,\nLowe JB. Supravalvular aortic stenosis. Circulation. 1961;24:1311\u20131318."},{"key":"i1878-7177-72-4-241-b02","series-title":"PLoS Genetics","first-page":"e1002479","article-title":"The Williams-Beuren Syndrome-a window into genetic variants leading to the development of cardiovascular disease","volume":"8","author":"Adams GN,","year":"2012","unstructured":"Adams GN,\nSchmaier AH. The Williams-Beuren Syndrome-a window into genetic variants leading to the development of cardiovascular disease. PLoS Genetics. 2012;8:e1002479."},{"key":"i1878-7177-72-4-241-b03","series-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod","first-page":"573","article-title":"Medical considerations in dental treatment of children with Williams syndrome","volume":"99","author":"Moskovitz M,","year":"2005","unstructured":"Moskovitz M,\nBrener D,\nFaibis S,\nPeretz B. Medical considerations in dental treatment of children with Williams syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2005;99:573\u2013580."},{"key":"i1878-7177-72-4-241-b04","series-title":"Can J Anaesth","first-page":"1203","article-title":"Anaesthetic management of a patient with Williams syndrome undergoing aortoplasty for supravalvular aortic stenosis","volume":"45","author":"Kawahito S,","year":"1998","unstructured":"Kawahito S,\nKitahata H,\nKimura H, et al. Anaesthetic management of a patient with Williams syndrome undergoing aortoplasty for supravalvular aortic stenosis. Can J Anaesth. 1998;45:1203\u20131206."},{"key":"i1878-7177-72-4-241-b05","series-title":"Med Oral Pathol Oral Radiol Endod","first-page":"e17","article-title":"General anesthesia for dental treatment in a Williams syndrome patient with severe aortic and pulmonary valve stenosis: suspected episode of postoperatively malignant hyperthermia","volume":"104","author":"Kohase H,","year":"2007","unstructured":"Kohase H,\nWakita R,\nDoi S,\nUmino M. General anesthesia for dental treatment in a Williams syndrome patient with severe aortic and pulmonary valve stenosis: suspected episode of postoperatively malignant hyperthermia. Med Oral Pathol Oral Radiol Endod. 2007;104:e17\u2013e20."},{"key":"i1878-7177-72-4-241-b06","series-title":"Pediatr Anesth","first-page":"483","article-title":"Williams syndrome","volume":"29","author":"Twite MD,","year":"2019","unstructured":"Twite MD,\nStenquist S,\nIng RJ. Williams syndrome. Pediatr Anesth. 2019;29:483\u2013490."},{"key":"i1878-7177-72-4-241-b07","series-title":"Am J Med Genet","first-page":"234","article-title":"Risk of sudden death in the Williams-Beuren Syndrome","volume":"127A","author":"Wessel A,","year":"2004","unstructured":"Wessel A,\nGravenhorst V,\nBuchhorn R,\nGosch A,\nPartsch CJ,\nPankau R. Risk of sudden death in the Williams-Beuren Syndrome. Am J Med Genet. 2004;127A:234\u2013237."},{"key":"i1878-7177-72-4-241-b08","series-title":"Anesth Analg","first-page":"125","volume":"101","author":"Albertin A,","year":"2005","unstructured":"Albertin A,\nCasati A,\nFederica L, et al. Anesth Analg. 2005;101:125\u2013130."},{"key":"i1878-7177-72-4-241-b09","series-title":"Hum Genet","first-page":"317","article-title":"Anesthesiologic problems in Williams syndrome: the CACNL2A locus is not involved","volume":"98","author":"Mammi I,","year":"1996","unstructured":"Mammi I,\nIles DE,\nSmeets D,\nClementi M,\nTenconi R. Anesthesiologic problems in Williams syndrome: the CACNL2A locus is not involved. Hum Genet. 1996;98:317\u2013320."},{"key":"i1878-7177-72-4-241-b10","series-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod","first-page":"e1","article-title":"Malignant hyperthermia in the oral and maxillofacial surgery patient: an update","volume":"112","author":"Patil PM.","year":"2011","unstructured":"Patil PM. Malignant hyperthermia in the oral and maxillofacial surgery patient: an update. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;112:e1\u2013e7."},{"key":"i1878-7177-72-4-241-b11","series-title":"Orphanet J Rare Dis","first-page":"93","article-title":"Malignant hyperthermia: a review","volume":"10","author":"Rosenberg H,","year":"2015","unstructured":"Rosenberg H,\nPollock N,\nSchiemann A,\nBulger T,\nStowell K. Malignant hyperthermia: a review. Orphanet J Rare Dis. 2015;10:93."},{"key":"i1878-7177-72-4-241-b01","series-title":"Circulation","first-page":"1311","article-title":"Supravalvular aortic stenosis","volume":"24","author":"Williams JCP,","year":"1961","unstructured":"Williams JCP,\nBarratt-Boyes BG,\nLowe JB. Supravalvular aortic stenosis. Circulation. 1961;24:1311\u20131318."},{"key":"i1878-7177-72-4-241-b02","series-title":"PLoS Genetics","first-page":"e1002479","article-title":"The Williams-Beuren Syndrome-a window into genetic variants leading to the development of cardiovascular disease","volume":"8","author":"Adams GN,","year":"2012","unstructured":"Adams GN,\nSchmaier AH. The Williams-Beuren Syndrome-a window into genetic variants leading to the development of cardiovascular disease. PLoS Genetics. 2012;8:e1002479."},{"key":"i1878-7177-72-4-241-b03","series-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod","first-page":"573","article-title":"Medical considerations in dental treatment of children with Williams syndrome","volume":"99","author":"Moskovitz M,","year":"2005","unstructured":"Moskovitz M,\nBrener D,\nFaibis S,\nPeretz B. Medical considerations in dental treatment of children with Williams syndrome. 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General anesthesia for dental treatment in a Williams syndrome patient with severe aortic and pulmonary valve stenosis: suspected episode of postoperatively malignant hyperthermia. Med Oral Pathol Oral Radiol Endod. 2007;104:e17\u2013e20."},{"key":"i1878-7177-72-4-241-b06","series-title":"Pediatr Anesth","first-page":"483","article-title":"Williams syndrome","volume":"29","author":"Twite MD,","year":"2019","unstructured":"Twite MD,\nStenquist S,\nIng RJ. Williams syndrome. Pediatr Anesth. 2019;29:483\u2013490."},{"key":"i1878-7177-72-4-241-b07","series-title":"Am J Med Genet","first-page":"234","article-title":"Risk of sudden death in the Williams-Beuren Syndrome","volume":"127A","author":"Wessel A,","year":"2004","unstructured":"Wessel A,\nGravenhorst V,\nBuchhorn R,\nGosch A,\nPartsch CJ,\nPankau R. Risk of sudden death in the Williams-Beuren Syndrome. 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Malignant hyperthermia in the oral and maxillofacial surgery patient: an update. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;112:e1\u2013e7."},{"key":"i1878-7177-72-4-241-b11","series-title":"Orphanet J Rare Dis","first-page":"93","article-title":"Malignant hyperthermia: a review","volume":"10","author":"Rosenberg H,","year":"2015","unstructured":"Rosenberg H,\nPollock N,\nSchiemann A,\nBulger T,\nStowell K. Malignant hyperthermia: a review. 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