Awake Tracheal Intubation in an 8-Year-Old Girl with McCune-Albright Syndrome J. Kyle Bohman, MD, Leal Segura, MD, and Kendra Grim, MD An 8-year-old girl with McCune-Albright syndrome presented for resection of a very large fibrous dysplasia mass of the face with significant distortion of the airway anatomy. She had significant obstructive sleep apnea with daytime somnolence and hemoglobin oxygen desaturations while breathing room air preoperatively. We were able to successfully manage her airway by providing IV sedation, topical anesthesia of the airway, and oral fiberoptic intubation in close collaboration with our otorhinolaryngology colleagues. (A&A Case Reports 2013;1:23–5.)
M
anagement of a difficult airway in the pediatric population is very challenging, especially due to the high likelihood of poor patient cooperation. Careful titration of analgesics and sedation while maintaining spontaneous ventilation and cooperation can be very difficult. In the case described here, we were able to provide a reasonable amount of patient comfort and cooperation without sacrificing safety. Written informed consent was obtained from the patient’s mother for use of the patient’s medical records including images for academic and educational purposes.
CASE REPORT
A 50.5 kg 8-year-old girl with McCune-Albright syndrome presented for resection of a large facial mass due to fibrous dysplasia involving the right facial bones and skull base (Figs. 1 and 2). The mass created significant anatomical shifts of the nasal and oral cavities resulting in severe obstructive airway symptoms and right eye blindness due to compression. She had daytime somnolence, frequent snoring, drooling, and dyspnea with stridor while supine. Further imaging studies, including head computed tomography, demonstrated tumor involvement of the right maxilla, right mandible, right sphenoid, and right temporal bones with near-complete effacement of the right orbit (Fig. 3). Due to significant airway obstruction, surgical resection of the tumor was recommended. Pediatric anesthesia and otorhinolaryngology collaborated on safe airway management for the procedure. Due to the oral and nasal cavity distortion and sleep apnea, mask ventilation in the setting of absent spontaneous breathing would likely have been impossible. We concluded the best initial approach to her airway management would be awake fiberoptic intubation followed by elective tracheostomy to facilitate surgical resection of the tumor. From the Department of Anesthesiology, Mayo Clinic, Rochester, Minnesota. Accepted for publication March 6, 2013. Funding: None. The authors declare no conflicts of interest. This report was previously presented, in part, at the Society of Pediatric Anesthesiology, 2013. Reprints will not be available from the authors. Address correspondence to J. Kyle Bohman, MD, Department of Anesthesiology, Mayo Clinic, 200 First St. SW, Rochester, MN 55905. Address e-mail to [email protected]. Copyright © 2013 International Anesthesia Research Society DOI: 10.1097/ACC.0b013e318291d47e
October 1, 2013 • Volume 1 • Number 1
Due to her history of dyspnea and snoring while supine, we positioned her in a nearly upright sitting position. Sedation included pretreatment with IV glycopyrrolate, followed by dexmedetomidine infusion (2 μg/kg/h without a bolus) and IV ketamine boluses (10–20 mg) as needed for signs of discomfort. Topical anesthesia was performed. Initially, 2 sprays of an aerosolized combination of 14% benzocaine and 2% tetracaine were delivered to the base of the tongue, followed by 5 mL of aerosolized 2% lidocaine directed at the glottis. Due to her poor mouth opening and large tonsils, it was difficult to effectively reach her glottis with the aerosolized lidocaine. Using a fiberoptic bronchoscope loaded with a 5.0 cuffed endotracheal tube, the first 3 attempts were unsuccessful due to large tonsils obstructing passage of the fiberoptic bronchoscope. The patient continued to receive incremental 10 mg boluses of IV ketamine for comfort and cooperation and “blow-by” oxygen. After the initial intubation attempts, topical airway anesthesia was no longer adequate, and the patient received an additional 4 mL of 4% atomized lidocaine directed at the glottis. After 4 more attempts, an adequate view of the glottis was obtained and the bronchoscope was successfully passed into the trachea. The primary reason for multiple attempts was difficulty in maneuvering the bronchoscope through the narrow oropharynx and past the massive tonsils. The tracheal tube was advanced over the bronchoscope easily and tracheal intubation confirmed with bronchoscopy, auscultation, and end-tidal capnography after which general anesthesia was induced with propofol.
DISCUSSION
Our approach to this patient’s difficult airway included multiple concerns. First, mask ventilation was likely impossible due to her contorted nasal, oral anatomy, and severe sleep apnea. Therefore, we had to ensure adequate spontaneous ventilation by minimizing respiratory depression and secretions and by proper positioning. We believe our selection of dexmedetomidine offered several advantages including: analgesia, minimal respiratory depression, maintenance of patient ability to cooperate, and lack of the sialagogue effects of ketamine.1 We also ensured that, if spontaneous ventilation was lost and we could not fiberoptically intubate the trachea, an experienced pediatric otorhinolaryngologist was available and prepared to perform an emergency tracheostomy. Although her neck anatomy appeared normal, we intended cases-anesthesia-analgesia.org
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Awake Intubation in an 8-Year-Old with Difficult Airway
Figure 1. Photograph from the front of the patient demonstrating a large fibrous dysplasia mass involving the right maxilla, mandible, and skull base with near-complete effacement of the right orbit.
Figure 2. Photograph from the left of the patient demonstrating a large fibrous dysplasia mass with severe displacement of the nasal anatomy and obscuring of the oral anatomy.
to avoid a tracheostomy if at all possible. Emergent tracheostomy in children, especially if awake, can be very challenging, if not impossible.2,3 Another concern for the difficult pediatric airway is the limited volume of topical local anesthetic that can be used safely due to patient size. Data for safe dosing of topical lidocaine applied to the oral mucosa are incomplete. In a series of previous prospective studies on children, topical lidocaine applied to the airway up to 4 mg/kg did not result in clinical toxicity. Plasma lidocaine levels measured in the children in these studies suggested a dose of 4 mg/kg yielded occasional serum concentrations larger than the reported toxic threshold of 5 to 10 μg/mL, despite lack of clinical toxicity observed. A topical lidocaine dose up to 2.6 mg/kg yielded a maximum serum lidocaine
concentration of 2.29 μg/mL. Dry oral mucosa resulted in increased serum lidocaine concentrations. Young children, particularly those younger than 2 years of age, had consistently higher serum lidocaine levels after application of topical lidocaine.4–6 In addition, there is a large interindividual variability of systemic absorption of topical lidocaine.7 The literature also stressed the toxic potential of lidocaine’s active metabolite, monoethylglycinexylidide,7 which Walson8 reported is potentially more toxic to the central nervous system than lidocaine itself. In our patient, after the local anesthesia of the airway lost efficacy, we discussed the risk of using more local anesthetic to reanesthetize the airway. The risk-benefit comparison of inadequate airway topicalization versus mild systemic local anesthetic toxicity heavily favored our decision to supplement with an additional dose of topical local anesthetic. In general, due to the rapid uptake of topical local anesthetics secondary to the highly vascularized nature of the airway mucosa, maximum dosing of topical local anesthetics may be considered equivalent to the dosing of IV local anesthetics.9
CONCLUSION
Judiciously applied topical anesthesia combined with dexmedetomidine-based IV sedation facilitated maintenance of spontaneous ventilation while attaining patient comfort and optimal intubating conditions in an 8-year-old child with predicted difficult mask ventilation and tracheal intubation. E
Figure 3. Sagittal computed tomography image illustrating a large fibrous dysplasia mass with extensive compression of the oral cavity.
24 cases-anesthesia-analgesia.org
REFERENCES 1. Tobias JD. Dexmedetomidine: applications in pediatric critical care and pediatric anesthesiology. Pediatr Crit Care Med 2007;8:115–31 2. Navsa N, Tossel G, Boon JM. Dimensions of the neonatal cricothyroid membrane - how feasible is a surgical cricothyroidotomy? Paediatr Anaesth 2005;15:402–6 3. Stacey J, Heard AM, Chapman G, Wallace CJ, Hegarty M, Vijayasekaran S, von Ungern-Sternberg BS. The ‘Can’t Intubate
A & A case reports
Can’t Oxygenate’ scenario in pediatric anesthesia: a comparison of different devices for needle cricothyroidotomy. Paediatr Anaesth 2012;22:1155–8 4. Eyres RL, Bishop W, Oppenheim RC, Brown TC. Plasma lignocaine concentrations following topical laryngeal application. Anaesth Intensive Care 1983;11:23–6 5. Whittet HB, Hayward AW, Battersby E. Plasma lignocaine levels during paediatric endoscopy of the upper respiratory tract. Anaesthesia 1988;43:439–442. 6. Sitbon P, Laffon M, Lesage V, Furet P, Autret E, Mercier C. Lidocaine plasma concentrations in pediatric patients after
October 1, 2013 • Volume 1 • Number 1
providing airway topical anesthesia from a calibrated device. Anesth Analg 1996;82:1003–6 7. Oni G, Brown S, Burrus C, Grant L, Watkins J, Kenkel M, Barton F, Kenkel J. Effect of 4% lidocaine applied to the face on the serum levels of lidocaine and its metabolite, monoethylglycinexylidide. Aesthet Surg J 2010;30:853 8. Walson PD. Toxicity of lidocaine desethyl metabolites [letter]. Clin Pharmacol Therap 1997;62:579 9. Rosenberg PH, Veering BT, Urmey WF. Maximum recommended doses of local anesthetics: a multifactorial concept. Reg Anesth Pain Med 2004;29:564–75
cases-anesthesia-analgesia.org
25
M
anagement of a difficult airway in the pediatric population is very challenging, especially due to the high likelihood of poor patient cooperation. Careful titration of analgesics and sedation while maintaining spontaneous ventilation and cooperation can be very difficult. In the case described here, we were able to provide a reasonable amount of patient comfort and cooperation without sacrificing safety. Written informed consent was obtained from the patient’s mother for use of the patient’s medical records including images for academic and educational purposes.
CASE REPORT
A 50.5 kg 8-year-old girl with McCune-Albright syndrome presented for resection of a large facial mass due to fibrous dysplasia involving the right facial bones and skull base (Figs. 1 and 2). The mass created significant anatomical shifts of the nasal and oral cavities resulting in severe obstructive airway symptoms and right eye blindness due to compression. She had daytime somnolence, frequent snoring, drooling, and dyspnea with stridor while supine. Further imaging studies, including head computed tomography, demonstrated tumor involvement of the right maxilla, right mandible, right sphenoid, and right temporal bones with near-complete effacement of the right orbit (Fig. 3). Due to significant airway obstruction, surgical resection of the tumor was recommended. Pediatric anesthesia and otorhinolaryngology collaborated on safe airway management for the procedure. Due to the oral and nasal cavity distortion and sleep apnea, mask ventilation in the setting of absent spontaneous breathing would likely have been impossible. We concluded the best initial approach to her airway management would be awake fiberoptic intubation followed by elective tracheostomy to facilitate surgical resection of the tumor. From the Department of Anesthesiology, Mayo Clinic, Rochester, Minnesota. Accepted for publication March 6, 2013. Funding: None. The authors declare no conflicts of interest. This report was previously presented, in part, at the Society of Pediatric Anesthesiology, 2013. Reprints will not be available from the authors. Address correspondence to J. Kyle Bohman, MD, Department of Anesthesiology, Mayo Clinic, 200 First St. SW, Rochester, MN 55905. Address e-mail to [email protected]. Copyright © 2013 International Anesthesia Research Society DOI: 10.1097/ACC.0b013e318291d47e
October 1, 2013 • Volume 1 • Number 1
Due to her history of dyspnea and snoring while supine, we positioned her in a nearly upright sitting position. Sedation included pretreatment with IV glycopyrrolate, followed by dexmedetomidine infusion (2 μg/kg/h without a bolus) and IV ketamine boluses (10–20 mg) as needed for signs of discomfort. Topical anesthesia was performed. Initially, 2 sprays of an aerosolized combination of 14% benzocaine and 2% tetracaine were delivered to the base of the tongue, followed by 5 mL of aerosolized 2% lidocaine directed at the glottis. Due to her poor mouth opening and large tonsils, it was difficult to effectively reach her glottis with the aerosolized lidocaine. Using a fiberoptic bronchoscope loaded with a 5.0 cuffed endotracheal tube, the first 3 attempts were unsuccessful due to large tonsils obstructing passage of the fiberoptic bronchoscope. The patient continued to receive incremental 10 mg boluses of IV ketamine for comfort and cooperation and “blow-by” oxygen. After the initial intubation attempts, topical airway anesthesia was no longer adequate, and the patient received an additional 4 mL of 4% atomized lidocaine directed at the glottis. After 4 more attempts, an adequate view of the glottis was obtained and the bronchoscope was successfully passed into the trachea. The primary reason for multiple attempts was difficulty in maneuvering the bronchoscope through the narrow oropharynx and past the massive tonsils. The tracheal tube was advanced over the bronchoscope easily and tracheal intubation confirmed with bronchoscopy, auscultation, and end-tidal capnography after which general anesthesia was induced with propofol.
DISCUSSION
Our approach to this patient’s difficult airway included multiple concerns. First, mask ventilation was likely impossible due to her contorted nasal, oral anatomy, and severe sleep apnea. Therefore, we had to ensure adequate spontaneous ventilation by minimizing respiratory depression and secretions and by proper positioning. We believe our selection of dexmedetomidine offered several advantages including: analgesia, minimal respiratory depression, maintenance of patient ability to cooperate, and lack of the sialagogue effects of ketamine.1 We also ensured that, if spontaneous ventilation was lost and we could not fiberoptically intubate the trachea, an experienced pediatric otorhinolaryngologist was available and prepared to perform an emergency tracheostomy. Although her neck anatomy appeared normal, we intended cases-anesthesia-analgesia.org
23
Awake Intubation in an 8-Year-Old with Difficult Airway
Figure 1. Photograph from the front of the patient demonstrating a large fibrous dysplasia mass involving the right maxilla, mandible, and skull base with near-complete effacement of the right orbit.
Figure 2. Photograph from the left of the patient demonstrating a large fibrous dysplasia mass with severe displacement of the nasal anatomy and obscuring of the oral anatomy.
to avoid a tracheostomy if at all possible. Emergent tracheostomy in children, especially if awake, can be very challenging, if not impossible.2,3 Another concern for the difficult pediatric airway is the limited volume of topical local anesthetic that can be used safely due to patient size. Data for safe dosing of topical lidocaine applied to the oral mucosa are incomplete. In a series of previous prospective studies on children, topical lidocaine applied to the airway up to 4 mg/kg did not result in clinical toxicity. Plasma lidocaine levels measured in the children in these studies suggested a dose of 4 mg/kg yielded occasional serum concentrations larger than the reported toxic threshold of 5 to 10 μg/mL, despite lack of clinical toxicity observed. A topical lidocaine dose up to 2.6 mg/kg yielded a maximum serum lidocaine
concentration of 2.29 μg/mL. Dry oral mucosa resulted in increased serum lidocaine concentrations. Young children, particularly those younger than 2 years of age, had consistently higher serum lidocaine levels after application of topical lidocaine.4–6 In addition, there is a large interindividual variability of systemic absorption of topical lidocaine.7 The literature also stressed the toxic potential of lidocaine’s active metabolite, monoethylglycinexylidide,7 which Walson8 reported is potentially more toxic to the central nervous system than lidocaine itself. In our patient, after the local anesthesia of the airway lost efficacy, we discussed the risk of using more local anesthetic to reanesthetize the airway. The risk-benefit comparison of inadequate airway topicalization versus mild systemic local anesthetic toxicity heavily favored our decision to supplement with an additional dose of topical local anesthetic. In general, due to the rapid uptake of topical local anesthetics secondary to the highly vascularized nature of the airway mucosa, maximum dosing of topical local anesthetics may be considered equivalent to the dosing of IV local anesthetics.9
CONCLUSION
Judiciously applied topical anesthesia combined with dexmedetomidine-based IV sedation facilitated maintenance of spontaneous ventilation while attaining patient comfort and optimal intubating conditions in an 8-year-old child with predicted difficult mask ventilation and tracheal intubation. E
Figure 3. Sagittal computed tomography image illustrating a large fibrous dysplasia mass with extensive compression of the oral cavity.
24 cases-anesthesia-analgesia.org
REFERENCES 1. Tobias JD. Dexmedetomidine: applications in pediatric critical care and pediatric anesthesiology. Pediatr Crit Care Med 2007;8:115–31 2. Navsa N, Tossel G, Boon JM. Dimensions of the neonatal cricothyroid membrane - how feasible is a surgical cricothyroidotomy? Paediatr Anaesth 2005;15:402–6 3. Stacey J, Heard AM, Chapman G, Wallace CJ, Hegarty M, Vijayasekaran S, von Ungern-Sternberg BS. The ‘Can’t Intubate
A & A case reports
Can’t Oxygenate’ scenario in pediatric anesthesia: a comparison of different devices for needle cricothyroidotomy. Paediatr Anaesth 2012;22:1155–8 4. Eyres RL, Bishop W, Oppenheim RC, Brown TC. Plasma lignocaine concentrations following topical laryngeal application. Anaesth Intensive Care 1983;11:23–6 5. Whittet HB, Hayward AW, Battersby E. Plasma lignocaine levels during paediatric endoscopy of the upper respiratory tract. Anaesthesia 1988;43:439–442. 6. Sitbon P, Laffon M, Lesage V, Furet P, Autret E, Mercier C. Lidocaine plasma concentrations in pediatric patients after
October 1, 2013 • Volume 1 • Number 1
providing airway topical anesthesia from a calibrated device. Anesth Analg 1996;82:1003–6 7. Oni G, Brown S, Burrus C, Grant L, Watkins J, Kenkel M, Barton F, Kenkel J. Effect of 4% lidocaine applied to the face on the serum levels of lidocaine and its metabolite, monoethylglycinexylidide. Aesthet Surg J 2010;30:853 8. Walson PD. Toxicity of lidocaine desethyl metabolites [letter]. Clin Pharmacol Therap 1997;62:579 9. Rosenberg PH, Veering BT, Urmey WF. Maximum recommended doses of local anesthetics: a multifactorial concept. Reg Anesth Pain Med 2004;29:564–75
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