{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,18]],"date-time":"2026-06-18T00:55:15Z","timestamp":1781744115823,"version":"3.54.5"},"reference-count":44,"publisher":"American Dental Society of Anesthesiology (ADSA)","issue":"2","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,6]]},"abstract":"<jats:p>\n                    Cornelia de Lange syndrome (CdLS) is a rare genetic disorder with a wide variation of presentations affecting several organ systems. Most patients with CdLS have a mutation of the\n                    <jats:italic>NIPBL<\/jats:italic>\n                    (Nipped-B-like protein) gene responsible for making delangin, a protein important in human development. Patients with CdLS typically present with intellectual disability, growth delay, cleft palate, and extremely limited mouth opening. These findings can complicate induction of general anesthesia, airway management, and intubation. Treatment of CdLS is based on clinical presentation and includes pharmacologic, therapeutic, and surgical intervention. Cleft palate repair is common in these patients which may impact the anesthetic plan. Additionally, the anesthesiologist should be aware of associated cardiovascular anomalies, ranging from innocent murmurs to persistent structural defects and leading to increased morbidity and mortality in these patients. This case report discusses CdLS and its implications on the general anesthetic management of a 37-year-old woman undergoing treatment for dental caries in an ambulatory care setting. Although CdLS is a rare genetic disorder, anesthesiologists treating such patients with special needs should be familiar with its presentation and anesthetic considerations.\n                  <\/jats:p>","DOI":"10.2344\/25-0002","type":"journal-article","created":{"date-parts":[[2026,6,18]],"date-time":"2026-06-18T00:30:49Z","timestamp":1781742649000},"page":"90-96","source":"Crossref","is-referenced-by-count":0,"title":["Cornelia de Lange Syndrome and General Anesthesia for Dental Treatment: A Case Report"],"prefix":"10.2344","volume":"73","author":[{"given":"Kira","family":"Douglas","sequence":"first","affiliation":[{"name":"Clinical Assistant Professor, University of Colorado School of Dental Medicine, Denver, CO"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Schwartz","sequence":"additional","affiliation":[{"name":"Dentist Anesthesiologist and Oral and Maxillofacial Surgeon, Pittsburgh, PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Craig","family":"McKenzie","sequence":"additional","affiliation":[{"name":"Assistant Professor of Dental Anesthesiology and Director of the Center for Patients with Special Needs, University of Pittsburgh School of Dental Medicine, Pittsburgh, PA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1191","reference":[{"key":"i1878-7177-73-2-90-b01","article-title":"Cornelia de Lange Syndrome - NORD (National Organization for Rare Disorders)","author":"NORD (National Organization for Rare Disorders).","year":"2021","unstructured":"NORD (National Organization for Rare Disorders). 2021. Cornelia de Lange Syndrome - NORD (National Organization for Rare Disorders) [online]. Accessed October 19, 2021. https:\/\/rarediseases.org\/rare-diseases\/cornelia-de-lange-syndrome\/"},{"key":"i1878-7177-73-2-90-b02","article-title":"Cornelia de Lange syndrome","author":"Rarediseases.info.nih.gov","year":"2021","unstructured":"Rarediseases.info.nih.gov. 2021. Cornelia de Lange syndrome [online]. Accessed October 19, 2021. https:\/\/rarediseases.info.nih.gov\/diseases\/10109\/cornelia-de-lange-syndrome"},{"key":"i1878-7177-73-2-90-b03","series-title":"Acta Paediatr Scand","first-page":"765","article-title":"Mortality, pathological findings and causes of death in the de Lange syndrome","volume":"74","author":"Beck","year":"1985","unstructured":"Beck B,\nFenger K. Mortality, pathological findings and causes of death in the de Lange syndrome. Acta Paediatr Scand. 1985;74:765\u2013769."},{"issue":"3","key":"i1878-7177-73-2-90-b04","series-title":"J Anaesthes Clin Pharmacol","first-page":"419","article-title":"Anesthetic considerations in a patient with Cornelia de-Lange syndrome","volume":"31","author":"Vestergaard","year":"2015","unstructured":"Vestergaard L,\nDey N,\nWinding R. Anesthetic considerations in a patient with Cornelia de-Lange syndrome. J Anaesthes Clin Pharmacol. 2015;31(3):419."},{"key":"i1878-7177-73-2-90-b05","series-title":"Middle East J Anesthesiol","first-page":"773","article-title":"Anesthetic management in a patient with Cornelia de Lange syndrome","volume":"20","author":"Washington","year":"2010","unstructured":"Washington V,\nKaye AD. Anesthetic management in a patient with Cornelia de Lange syndrome. Middle East J Anesthesiol. 2010;20:773\u2013778."},{"key":"i1878-7177-73-2-90-b06","series-title":"Anesth Prog","first-page":"63","article-title":"Anesthetic management of a patient with Cornelia De Lange syndrome","volume":"34","author":"Takeshita","year":"1987","unstructured":"Takeshita T,\nAkita S,\nKawahara M. Anesthetic management of a patient with Cornelia De Lange syndrome. Anesth Prog. 1987;34:63\u201365."},{"issue":"1","key":"i1878-7177-73-2-90-b07","series-title":"Bull Tokyo Dent Coll","first-page":"53","article-title":"Parental satisfaction with ambulatory anesthesia during dental treatment for disabled individuals and their preference for same in future","volume":"60","author":"Ohtawa","year":"2019","unstructured":"Ohtawa Y,\nYoshida M,\nFukuda K. Parental satisfaction with ambulatory anesthesia during dental treatment for disabled individuals and their preference for same in future. Bull Tokyo Dent Coll. 2019;60(1):53\u201360."},{"issue":"4","key":"i1878-7177-73-2-90-b08","series-title":"J Anaesthes Clin Pharmacol","first-page":"432","article-title":"Anesthesia for intellectually disabled [online]","volume":"33","author":"Chaudhary","year":"2019","unstructured":"Chaudhary K,\nBagharwal P,\nWadhawan S. Anesthesia for intellectually disabled [online]. J Anaesthes Clin Pharmacol. 2019;33(4):432\u2013440. Accessed November 12, 2021. https:\/\/www.ncbi.nlm.nih.gov\/pmc\/articles\/PMC5791252\/"},{"key":"i1878-7177-73-2-90-b09","series-title":"Med Oral Patol Oral Cir Bucal","article-title":"Premedication with midazolam in intellectually disabled dental patients: intramuscular or oral administration? A retrospective study","author":"Hanamoto","year":"2016","unstructured":"Hanamoto H,\nBoku A,\nSugimura M,\nOyamaguchi A,\nInoue M,\nNiwa H. Premedication with midazolam in intellectually disabled dental patients: intramuscular or oral administration? A retrospective study. Med Oral Patol Oral Cir Bucal. 2016. doi:10.4317\/medoral.21086"},{"issue":"1","key":"i1878-7177-73-2-90-b10","series-title":"J Anesthes","first-page":"126","article-title":"Combination of oral ketamine and midazolam as a premedication for a severely autistic and combative patient","volume":"23","author":"Shah","year":"2009","unstructured":"Shah S,\nShah S,\nApuya J.,\nGopalakrishnan S,\nMartin T. Combination of oral ketamine and midazolam as a premedication for a severely autistic and combative patient. J Anesthes, 2009;23(1):126\u2013128."},{"issue":"1","key":"i1878-7177-73-2-90-b11","series-title":"Anaesthesiol Intens Ther","first-page":"72","article-title":"Complications associated with nasotracheal intubation and proposal of simple countermeasure","volume":"51","author":"Yamamoto","year":"2019","unstructured":"Yamamoto T,\nFlenner M,\nSchindler E. Complications associated with nasotracheal intubation and proposal of simple countermeasure. Anaesthesiol Intens Ther. 2019;51(1):72\u201373."},{"issue":"9","key":"i1878-7177-73-2-90-b12","series-title":"Anaesthesia","first-page":"923","article-title":"Nasal intubation and previous cleft palate repair","volume":"59","author":"Solan","year":"2004","unstructured":"Solan K. Nasal intubation and previous cleft palate repair. Anaesthesia, 2004;59(9):923\u2013924."},{"issue":"2","key":"i1878-7177-73-2-90-b13","series-title":"Am J Med Genet Part C: Semin Med Genet","first-page":"222","article-title":"Sedation and general anesthesia for patients with Cornelia De Lange syndrome: a case series","volume":"172","author":"Moretto","year":"2016","unstructured":"Moretto A,\nScaravilli V,\nCiceri V, et al. Sedation and general anesthesia for patients with Cornelia De Lange syndrome: a case series. Am J Med Genet Part C: Semin Med Genet. 2016;172(2):222\u2013228."},{"issue":"1","key":"i1878-7177-73-2-90-b14","series-title":"Cleft Palate J","first-page":"46","article-title":"The relationship between nasal airway size and nasal-oral breathing in cleft lip and palate","volume":"27","author":"Warren","year":"1990","unstructured":"Warren D,\nHairfield W,\nDalston E. The relationship between nasal airway size and nasal-oral breathing in cleft lip and palate. Cleft Palate J. 1990;27(1):46\u201352."},{"key":"i1878-7177-73-2-90-b15","series-title":"Am J Orthod Dentofacial Orthop","first-page":"390","article-title":"The relationship between nasal airway size and nasal airway resistance","volume":"92","author":"Warren","year":"1987","unstructured":"Warren DW,\nHairfield WM,\nSeaton D, et al. The relationship between nasal airway size and nasal airway resistance. Am J Orthod Dentofacial Orthop. 1987;92:390\u2013395."},{"issue":"7","key":"i1878-7177-73-2-90-b16","series-title":"Canad J Anesthes","first-page":"721","article-title":"Airway management in a patient with a cleft palate after pharyngoplasty: a case report","volume":"50","author":"Hee","year":"2003","unstructured":"Hee H,\nConskunfirat N,\nWong S,\nChen C. Airway management in a patient with a cleft palate after pharyngoplasty: a case report. Canad J Anesthes. 2003;50(7):721\u2013724."},{"key":"i1878-7177-73-2-90-b17","series-title":"J Canad Dent Assoc","first-page":"688","article-title":"Frequency of pharyngoplasty after primary repair of cleft palate","volume":"68","author":"Bicknell","year":"2002","unstructured":"Bicknell S,\nMcFadden LR,\nCurran JB. Frequency of pharyngoplasty after primary repair of cleft palate. J Canad Dent Assoc. 2002; 68: 688\u2013692."},{"issue":"10","key":"i1878-7177-73-2-90-b18","series-title":"Am J Med Genet Part A","first-page":"2499","article-title":"Congenital heart disease in Cornelia de Lange syndrome: phenotype and genotype analysis","volume":"158A","author":"Chatfield","year":"2012","unstructured":"Chatfield K,\nSchrier S,\nLi J. Congenital heart disease in Cornelia de Lange syndrome: phenotype and genotype analysis. Am J Med Genet Part A, 2012;158A(10):2499\u20132505."},{"key":"i1878-7177-73-2-90-b19","series-title":"Am J Med Genet Part A","first-page":"1268","article-title":"Analysis of congenital heart defects in 87 consecutive patients with Brachmann\u2013de Lange syndrome","volume":"149A","author":"Selicorni","year":"2009","unstructured":"Selicorni A,\nColli AM,\nPassarini A, et al. Analysis of congenital heart defects in 87 consecutive patients with Brachmann\u2013de Lange syndrome. Am J Med Genet Part A. 2009;149A:1268\u20131272."},{"key":"i1878-7177-73-2-90-b20","series-title":"Am J Med Genet","first-page":"940","article-title":"de Lange syndrome: a clinical review of 310 individuals","volume":"47","author":"Jackson","year":"1993","unstructured":"Jackson L,\nKline AD,\nBarr MA,\nKoch S. de Lange syndrome: a clinical review of 310 individuals. Am J Med Genet. 1993;47:940\u2013946."},{"key":"i1878-7177-73-2-90-b21","article-title":"CdLS and Congenital Heart Issues. [online","author":"Rockow","year":"2018","unstructured":"Rockow J. 2018. CdLS and Congenital Heart Issues. [online] CdLS Foundation. Accessed 15 November 2021. http:\/\/chromeextension:\/\/efaidnbmnnnibpcajpcglclefindmkaj\/viewer.html?pdfurl=https%3A%2F%2Fwww.cdlsusa.org%2Fwp-content%2Fuploads%2F2018%2F10%2FCONGENITAL-HEART-ISSUES.pdf&clen=346290&chunk=true"},{"key":"i1878-7177-73-2-90-b22","series-title":"Paediatr Anaesthes","first-page":"159","article-title":"Anaesthetic implications of Cornelia de Lange syndrome","volume":"8","author":"Corsini","year":"1998","unstructured":"Corsini LM,\nStefano GD,\nPorras MC,\nGalindo S,\nPalencia J. Anaesthetic implications of Cornelia de Lange syndrome. Paediatr Anaesthes. 1998;8:159\u2013162."},{"key":"i1878-7177-73-2-90-b01","article-title":"Cornelia de Lange Syndrome - NORD (National Organization for Rare Disorders)","author":"NORD (National Organization for Rare Disorders).","year":"2021","unstructured":"NORD (National Organization for Rare Disorders). 2021. Cornelia de Lange Syndrome - NORD (National Organization for Rare Disorders) [online]. Accessed October 19, 2021. https:\/\/rarediseases.org\/rare-diseases\/cornelia-de-lange-syndrome\/"},{"key":"i1878-7177-73-2-90-b02","article-title":"Cornelia de Lange syndrome","author":"Rarediseases.info.nih.gov","year":"2021","unstructured":"Rarediseases.info.nih.gov. 2021. Cornelia de Lange syndrome [online]. Accessed October 19, 2021. https:\/\/rarediseases.info.nih.gov\/diseases\/10109\/cornelia-de-lange-syndrome"},{"key":"i1878-7177-73-2-90-b03","series-title":"Acta Paediatr Scand","first-page":"765","article-title":"Mortality, pathological findings and causes of death in the de Lange syndrome","volume":"74","author":"Beck","year":"1985","unstructured":"Beck B,\nFenger K. Mortality, pathological findings and causes of death in the de Lange syndrome. Acta Paediatr Scand. 1985;74:765\u2013769."},{"issue":"3","key":"i1878-7177-73-2-90-b04","series-title":"J Anaesthes Clin Pharmacol","first-page":"419","article-title":"Anesthetic considerations in a patient with Cornelia de-Lange syndrome","volume":"31","author":"Vestergaard","year":"2015","unstructured":"Vestergaard L,\nDey N,\nWinding R. Anesthetic considerations in a patient with Cornelia de-Lange syndrome. J Anaesthes Clin Pharmacol. 2015;31(3):419."},{"key":"i1878-7177-73-2-90-b05","series-title":"Middle East J Anesthesiol","first-page":"773","article-title":"Anesthetic management in a patient with Cornelia de Lange syndrome","volume":"20","author":"Washington","year":"2010","unstructured":"Washington V,\nKaye AD. Anesthetic management in a patient with Cornelia de Lange syndrome. Middle East J Anesthesiol. 2010;20:773\u2013778."},{"key":"i1878-7177-73-2-90-b06","series-title":"Anesth Prog","first-page":"63","article-title":"Anesthetic management of a patient with Cornelia De Lange syndrome","volume":"34","author":"Takeshita","year":"1987","unstructured":"Takeshita T,\nAkita S,\nKawahara M. Anesthetic management of a patient with Cornelia De Lange syndrome. Anesth Prog. 1987;34:63\u201365."},{"issue":"1","key":"i1878-7177-73-2-90-b07","series-title":"Bull Tokyo Dent Coll","first-page":"53","article-title":"Parental satisfaction with ambulatory anesthesia during dental treatment for disabled individuals and their preference for same in future","volume":"60","author":"Ohtawa","year":"2019","unstructured":"Ohtawa Y,\nYoshida M,\nFukuda K. Parental satisfaction with ambulatory anesthesia during dental treatment for disabled individuals and their preference for same in future. Bull Tokyo Dent Coll. 2019;60(1):53\u201360."},{"issue":"4","key":"i1878-7177-73-2-90-b08","series-title":"J Anaesthes Clin Pharmacol","first-page":"432","article-title":"Anesthesia for intellectually disabled [online]","volume":"33","author":"Chaudhary","year":"2019","unstructured":"Chaudhary K,\nBagharwal P,\nWadhawan S. Anesthesia for intellectually disabled [online]. J Anaesthes Clin Pharmacol. 2019;33(4):432\u2013440. Accessed November 12, 2021. https:\/\/www.ncbi.nlm.nih.gov\/pmc\/articles\/PMC5791252\/"},{"key":"i1878-7177-73-2-90-b09","series-title":"Med Oral Patol Oral Cir Bucal","article-title":"Premedication with midazolam in intellectually disabled dental patients: intramuscular or oral administration? A retrospective study","author":"Hanamoto","year":"2016","unstructured":"Hanamoto H,\nBoku A,\nSugimura M,\nOyamaguchi A,\nInoue M,\nNiwa H. Premedication with midazolam in intellectually disabled dental patients: intramuscular or oral administration? A retrospective study. Med Oral Patol Oral Cir Bucal. 2016. doi:10.4317\/medoral.21086"},{"issue":"1","key":"i1878-7177-73-2-90-b10","series-title":"J Anesthes","first-page":"126","article-title":"Combination of oral ketamine and midazolam as a premedication for a severely autistic and combative patient","volume":"23","author":"Shah","year":"2009","unstructured":"Shah S,\nShah S,\nApuya J.,\nGopalakrishnan S,\nMartin T. Combination of oral ketamine and midazolam as a premedication for a severely autistic and combative patient. J Anesthes, 2009;23(1):126\u2013128."},{"issue":"1","key":"i1878-7177-73-2-90-b11","series-title":"Anaesthesiol Intens Ther","first-page":"72","article-title":"Complications associated with nasotracheal intubation and proposal of simple countermeasure","volume":"51","author":"Yamamoto","year":"2019","unstructured":"Yamamoto T,\nFlenner M,\nSchindler E. Complications associated with nasotracheal intubation and proposal of simple countermeasure. Anaesthesiol Intens Ther. 2019;51(1):72\u201373."},{"issue":"9","key":"i1878-7177-73-2-90-b12","series-title":"Anaesthesia","first-page":"923","article-title":"Nasal intubation and previous cleft palate repair","volume":"59","author":"Solan","year":"2004","unstructured":"Solan K. Nasal intubation and previous cleft palate repair. Anaesthesia, 2004;59(9):923\u2013924."},{"issue":"2","key":"i1878-7177-73-2-90-b13","series-title":"Am J Med Genet Part C: Semin Med Genet","first-page":"222","article-title":"Sedation and general anesthesia for patients with Cornelia De Lange syndrome: a case series","volume":"172","author":"Moretto","year":"2016","unstructured":"Moretto A,\nScaravilli V,\nCiceri V, et al. Sedation and general anesthesia for patients with Cornelia De Lange syndrome: a case series. Am J Med Genet Part C: Semin Med Genet. 2016;172(2):222\u2013228."},{"issue":"1","key":"i1878-7177-73-2-90-b14","series-title":"Cleft Palate J","first-page":"46","article-title":"The relationship between nasal airway size and nasal-oral breathing in cleft lip and palate","volume":"27","author":"Warren","year":"1990","unstructured":"Warren D,\nHairfield W,\nDalston E. The relationship between nasal airway size and nasal-oral breathing in cleft lip and palate. Cleft Palate J. 1990;27(1):46\u201352."},{"key":"i1878-7177-73-2-90-b15","series-title":"Am J Orthod Dentofacial Orthop","first-page":"390","article-title":"The relationship between nasal airway size and nasal airway resistance","volume":"92","author":"Warren","year":"1987","unstructured":"Warren DW,\nHairfield WM,\nSeaton D, et al. The relationship between nasal airway size and nasal airway resistance. Am J Orthod Dentofacial Orthop. 1987;92:390\u2013395."},{"issue":"7","key":"i1878-7177-73-2-90-b16","series-title":"Canad J Anesthes","first-page":"721","article-title":"Airway management in a patient with a cleft palate after pharyngoplasty: a case report","volume":"50","author":"Hee","year":"2003","unstructured":"Hee H,\nConskunfirat N,\nWong S,\nChen C. Airway management in a patient with a cleft palate after pharyngoplasty: a case report. Canad J Anesthes. 2003;50(7):721\u2013724."},{"key":"i1878-7177-73-2-90-b17","series-title":"J Canad Dent Assoc","first-page":"688","article-title":"Frequency of pharyngoplasty after primary repair of cleft palate","volume":"68","author":"Bicknell","year":"2002","unstructured":"Bicknell S,\nMcFadden LR,\nCurran JB. Frequency of pharyngoplasty after primary repair of cleft palate. J Canad Dent Assoc. 2002; 68: 688\u2013692."},{"issue":"10","key":"i1878-7177-73-2-90-b18","series-title":"Am J Med Genet Part A","first-page":"2499","article-title":"Congenital heart disease in Cornelia de Lange syndrome: phenotype and genotype analysis","volume":"158A","author":"Chatfield","year":"2012","unstructured":"Chatfield K,\nSchrier S,\nLi J. Congenital heart disease in Cornelia de Lange syndrome: phenotype and genotype analysis. Am J Med Genet Part A, 2012;158A(10):2499\u20132505."},{"key":"i1878-7177-73-2-90-b19","series-title":"Am J Med Genet Part A","first-page":"1268","article-title":"Analysis of congenital heart defects in 87 consecutive patients with Brachmann\u2013de Lange syndrome","volume":"149A","author":"Selicorni","year":"2009","unstructured":"Selicorni A,\nColli AM,\nPassarini A, et al. Analysis of congenital heart defects in 87 consecutive patients with Brachmann\u2013de Lange syndrome. Am J Med Genet Part A. 2009;149A:1268\u20131272."},{"key":"i1878-7177-73-2-90-b20","series-title":"Am J Med Genet","first-page":"940","article-title":"de Lange syndrome: a clinical review of 310 individuals","volume":"47","author":"Jackson","year":"1993","unstructured":"Jackson L,\nKline AD,\nBarr MA,\nKoch S. de Lange syndrome: a clinical review of 310 individuals. Am J Med Genet. 1993;47:940\u2013946."},{"key":"i1878-7177-73-2-90-b21","article-title":"CdLS and Congenital Heart Issues. [online","author":"Rockow","year":"2018","unstructured":"Rockow J. 2018. CdLS and Congenital Heart Issues. [online] CdLS Foundation. Accessed 15 November 2021. http:\/\/chromeextension:\/\/efaidnbmnnnibpcajpcglclefindmkaj\/viewer.html?pdfurl=https%3A%2F%2Fwww.cdlsusa.org%2Fwp-content%2Fuploads%2F2018%2F10%2FCONGENITAL-HEART-ISSUES.pdf&clen=346290&chunk=true"},{"key":"i1878-7177-73-2-90-b22","series-title":"Paediatr Anaesthes","first-page":"159","article-title":"Anaesthetic implications of Cornelia de Lange syndrome","volume":"8","author":"Corsini","year":"1998","unstructured":"Corsini LM,\nStefano GD,\nPorras MC,\nGalindo S,\nPalencia J. Anaesthetic implications of Cornelia de Lange syndrome. 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