A case of safe airway management by fiber-optic nasotracheal intubation in general anesthesia in a pediatric patient with Hajdu-Cheney syndrome: a case report.

Airway management Difficult airway Hajdu-Cheney syndrome Nasotracheal intubation

Journal

JA clinical reports
ISSN: 2363-9024
Titre abrégé: JA Clin Rep
Pays: Germany
ID NLM: 101682121

Informations de publication

Date de publication:
12 Jun 2023
Historique:
received: 06 04 2023
accepted: 02 06 2023
revised: 22 05 2023
medline: 12 6 2023
pubmed: 12 6 2023
entrez: 12 6 2023
Statut: epublish

Résumé

Hajdu-Cheney syndrome (HCS) is an extremely rare disorder characterized by progressive acro-osteolysis. A unique facial structure and deformity of the cervical spine are associated with a difficult airway. Although several reports describe general anesthesia with orotracheal intubation for patients with HCS, there have been no reports of nasotracheal intubation with a risk of skull base fracture. We describe nasotracheal intubation for oral surgery in a patient with HCS. A 13-year-old girl with HCS was scheduled for dental surgery. Preoperative computed tomography revealed no abnormalities including fractures in the skull base or cervical spine. After confirming a lack of vocal cord paralysis by bronchofiberscopic inspection from the nose, general anesthesia was induced with sevoflurane, remifentanil, and rocuronium. Fiber-optic nasotracheal intubation was successfully performed without complications such as depletion of oxygen saturation and massive epistaxis, and the surgery was completed uneventfully. She was discharged the day after surgery with no anesthesia-related complications. We were able to safely manage the airway of a patient with HCS by nasotracheal intubation under general anesthesia.

Sections du résumé

BACKGROUND BACKGROUND
Hajdu-Cheney syndrome (HCS) is an extremely rare disorder characterized by progressive acro-osteolysis. A unique facial structure and deformity of the cervical spine are associated with a difficult airway. Although several reports describe general anesthesia with orotracheal intubation for patients with HCS, there have been no reports of nasotracheal intubation with a risk of skull base fracture. We describe nasotracheal intubation for oral surgery in a patient with HCS.
CASE PRESENTATION METHODS
A 13-year-old girl with HCS was scheduled for dental surgery. Preoperative computed tomography revealed no abnormalities including fractures in the skull base or cervical spine. After confirming a lack of vocal cord paralysis by bronchofiberscopic inspection from the nose, general anesthesia was induced with sevoflurane, remifentanil, and rocuronium. Fiber-optic nasotracheal intubation was successfully performed without complications such as depletion of oxygen saturation and massive epistaxis, and the surgery was completed uneventfully. She was discharged the day after surgery with no anesthesia-related complications.
CONCLUSIONS CONCLUSIONS
We were able to safely manage the airway of a patient with HCS by nasotracheal intubation under general anesthesia.

Identifiants

pubmed: 37306808
doi: 10.1186/s40981-023-00627-7
pii: 10.1186/s40981-023-00627-7
pmc: PMC10260714
doi:

Types de publication

Journal Article

Langues

eng

Pagination

33

Informations de copyright

© 2023. The Author(s).

Références

J Clin Epidemiol. 2017 Sep;89:218-235
pubmed: 28529185
Anesth Analg. 2000 Oct;91(4):837-55
pubmed: 11004035
Am J Med Genet A. 2023 Jan;191(1):271-274
pubmed: 36301051
Paediatr Anaesth. 2015 Jul;25(7):727-37
pubmed: 25736220
Clin Orthop Relat Res. 1990 Jun;(255):111-6
pubmed: 2347142
Am J Med Genet. 1997 Jun 13;70(3):261-6
pubmed: 9188663
J Anesth. 2013 Dec;27(6):949-50
pubmed: 23728363
Am J Med Genet. 2001 May 15;100(4):292-310
pubmed: 11343321
Indian J Med Res. 2016 May;143(5):663-4
pubmed: 27488012
Paediatr Anaesth. 2009 Jun;19(6):649-50
pubmed: 19646007
J Med Case Rep. 2013 Sep 10;7:223
pubmed: 24228906
Br J Anaesth. 1998 Feb;80(2):140-6
pubmed: 9602574
Clin Genet. 1997 Apr;51(4):271-4
pubmed: 9184252

Auteurs

Atsushi Kokita (A)

Department of Anesthesiology, Sapporo Medical University School of Medicine, South 1, West 16, Chuo-Ku, Sapporo, Hokkaido, 060-8543, Japan. a.kokita0119@gmail.com.

Tomohiro Chaki (T)

Department of Anesthesiology, Sapporo Medical University School of Medicine, South 1, West 16, Chuo-Ku, Sapporo, Hokkaido, 060-8543, Japan.

Michiaki Yamakage (M)

Department of Anesthesiology, Sapporo Medical University School of Medicine, South 1, West 16, Chuo-Ku, Sapporo, Hokkaido, 060-8543, Japan.

Classifications MeSH