{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T03:02:33Z","timestamp":1779850953438,"version":"3.53.1"},"reference-count":10,"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>Myhre syndrome (MS) is a rare genetic connective tissue disorder characterized by intellectual disability, growth deficiency, muscular pseudohypertrophy, hearing loss, restricted joint mobility, laryngotracheal stenosis, choanal stenosis, and facial deformities. We encountered a case of difficult endotracheal intubation in a patient with MS. A 17-year-old girl with trismus, macroglossia, and difficulty with neck flexion was scheduled to undergo general anesthesia for dental treatment. Because of these characteristic clinical manifestations, endotracheal intubation was performed with a flexible fiber-optic bronchoscope. When providing general anesthesia for patients with MS, preoperative evaluation of the airway and preparation for potentially difficult tracheal intubation are required.<\/jats:p>","DOI":"10.2344\/24-0037","type":"journal-article","created":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T11:10:10Z","timestamp":1765365010000},"page":"249-250","source":"Crossref","is-referenced-by-count":0,"title":["Anesthetic Management of a Patient With Myhre Syndrome"],"prefix":"10.2344","volume":"72","author":[{"given":"Yayoi","family":"Morita","sequence":"first","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chiho","family":"Kudo","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hikaru","family":"Nakagawa","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ken","family":"Mihara","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toshihiro","family":"Watanabe","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hitoshi","family":"Niwa","sequence":"additional","affiliation":[{"name":"Department of Dental Anesthesiology, Osaka University Graduate School of Dentistry, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1191","reference":[{"key":"i1878-7177-72-4-249-b01","series-title":"Clin Genet","first-page":"1-5.2","article-title":"A new growth deficiency syndrome","volume":"20","author":"Myhre SA,","year":"1981","unstructured":"Myhre SA,\nRuvalcaba RHA,\nGraham CB. A new growth deficiency syndrome. Clin Genet. 1981;20:1-5.2."},{"issue":"1","key":"i1878-7177-72-4-249-b02","series-title":"Nat Genet","first-page":"85","article-title":"Mutations at a single codon in Mad homology 2 domain of SMAD4 cause Myhre syndrome","volume":"44","author":"Le Goff C,","year":"2011","unstructured":"Le Goff C,\nMahaut C,\nAbhyankar A, et al. Mutations at a single codon in Mad homology 2 domain of SMAD4 cause Myhre syndrome. Nat Genet. 2011;44(1):85\u201388."},{"issue":"12","key":"i1878-7177-72-4-249-b03","series-title":"Am J Med Genet A","first-page":"2893","article-title":"Myhre syndrome: clinical features and restrictive cardiopulmonary complications","volume":"167A","author":"Starr LJ,","year":"2015","unstructured":"Starr LJ,\nGrange DK,\nDelaney JW, et al. Myhre syndrome: clinical features and restrictive cardiopulmonary complications. Am J Med Genet A. 2015;167A(12):2893\u20132901."},{"issue":"6","key":"i1878-7177-72-4-249-b04","series-title":"Am J Respir Crit Care Med","first-page":"731","article-title":"Life-threatening multilevel airway stenosis due to Myhre syndrome","volume":"201","author":"Alape D,","year":"2020","unstructured":"Alape D,\nSingh R,\nFolch E,\nFernandez Bussy S,\nAgnew A,\nMajid A. Life-threatening multilevel airway stenosis due to Myhre syndrome. Am J Respir Crit Care Med. 2020;201(6):731\u2013732."},{"issue":"5","key":"i1878-7177-72-4-249-b05","series-title":"Am J Otolaryngol","first-page":"636","article-title":"Myhre-LAPs syndrome and intubation related airway stenosis: keys to diagnosis and critical therapeutic interventions","volume":"36","author":"Oldenburg MS,","year":"2015","unstructured":"Oldenburg MS,\nFrisch CD,\nLindor NM,\nEdell ES,\nKasperbauer JL,\nO\u2019Brien EK. Myhre-LAPs syndrome and intubation related airway stenosis: keys to diagnosis and critical therapeutic interventions. Am J Otolaryngol. 2015;36(5):636\u2013641."},{"key":"i1878-7177-72-4-249-b01","series-title":"Clin Genet","first-page":"1-5.2","article-title":"A new growth deficiency syndrome","volume":"20","author":"Myhre SA,","year":"1981","unstructured":"Myhre SA,\nRuvalcaba RHA,\nGraham CB. A new growth deficiency syndrome. Clin Genet. 1981;20:1-5.2."},{"issue":"1","key":"i1878-7177-72-4-249-b02","series-title":"Nat Genet","first-page":"85","article-title":"Mutations at a single codon in Mad homology 2 domain of SMAD4 cause Myhre syndrome","volume":"44","author":"Le Goff C,","year":"2011","unstructured":"Le Goff C,\nMahaut C,\nAbhyankar A, et al. Mutations at a single codon in Mad homology 2 domain of SMAD4 cause Myhre syndrome. Nat Genet. 2011;44(1):85\u201388."},{"issue":"12","key":"i1878-7177-72-4-249-b03","series-title":"Am J Med Genet A","first-page":"2893","article-title":"Myhre syndrome: clinical features and restrictive cardiopulmonary complications","volume":"167A","author":"Starr LJ,","year":"2015","unstructured":"Starr LJ,\nGrange DK,\nDelaney JW, et al. Myhre syndrome: clinical features and restrictive cardiopulmonary complications. Am J Med Genet A. 2015;167A(12):2893\u20132901."},{"issue":"6","key":"i1878-7177-72-4-249-b04","series-title":"Am J Respir Crit Care Med","first-page":"731","article-title":"Life-threatening multilevel airway stenosis due to Myhre syndrome","volume":"201","author":"Alape D,","year":"2020","unstructured":"Alape D,\nSingh R,\nFolch E,\nFernandez Bussy S,\nAgnew A,\nMajid A. Life-threatening multilevel airway stenosis due to Myhre syndrome. Am J Respir Crit Care Med. 2020;201(6):731\u2013732."},{"issue":"5","key":"i1878-7177-72-4-249-b05","series-title":"Am J Otolaryngol","first-page":"636","article-title":"Myhre-LAPs syndrome and intubation related airway stenosis: keys to diagnosis and critical therapeutic interventions","volume":"36","author":"Oldenburg MS,","year":"2015","unstructured":"Oldenburg MS,\nFrisch CD,\nLindor NM,\nEdell ES,\nKasperbauer JL,\nO\u2019Brien EK. Myhre-LAPs syndrome and intubation related airway stenosis: keys to diagnosis and critical therapeutic interventions. 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