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PEDIATRIC ANESTHESIA

A Case of Difficult Airway Due to Lingual Tonsillar Hypertrophy in a Patient with Down’s Syndrome

Department of Anaesthesiology & Critical Care Medicine, Tokyo Medical & Dental University School of Medicine, Tokyo, Japan

Address correspondence and reprint requests to K. Nakazawa, MD, Department of Anaesthesiology & Critical Care Medicine, Tokyo Medical & Dental University School of Medicine, 1-5-45 Yushima, Bunkyo-ku, Tokyo 1138519, Japan. Address e-mail to nakazawa.mane{at}tmd.ac.jp

	Abstract

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In this report, we describe airway management of symptomatic lingual tonsillar hypertrophy in a pediatric patient with Down’s syndrome. Besides obstructive sleep apnea, the history included a small atrial septal defect with mild aortic regurgitation and Moyamoya disease. Anesthesia was induced with IV administration of 1 mg/kg of propofol, followed by inhalation of sevoflurane in 100% oxygen. Muscle relaxants were not used on induction. Rigid laryngoscopy could not visualize the epiglottis because of hypertrophied tonsillar tissue, and mask ventilation became difficult when spontaneous breathing stopped. We avoided using a laryngeal mask airway because of a slight bleeding tendency presumably caused by preoperative antiplatelet therapy. Fiberoptic bronchoscopy through the nasal cavity in combination with jet ventilation successfully identified the glottis and allowed nasotracheal intubation to be accomplished. After lingual tonsillectomy, the patient was extubated on the seventh postoperative day, after supraglottic edema had resolved. Fiberoptic nasotracheal intubation under inhaled anesthesia may therefore be preferable in pediatric or uncooperative patients with symptomatic lingual tonsillar hypertrophy.

IMPLICATIONS: We report a case of airway management of symptomatic lingual tonsillar hypertrophy (LTH) in a patient with Down’s syndrome. Fiberoptic nasotracheal intubation while maintaining spontaneous breathing under inhaled anesthesia may be preferable in pediatric or uncooperative patients with LTH.

	Introduction

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Children with Down’s syndrome frequently demonstrate obstructive sleep apnea (OSA) syndrome (1), of which there are many causes, including lymphoid hyperplasia, macroglossia, laryngomalacia, congenital subglottic stenosis, and tracheal stenosis (1,2). Lingual tonsillar hypertrophy (LTH) is a relatively rare form of lymphoid hyperplasia, but it may cause life-threatening airway obstruction (3,4). Generally, LTH may also be detected by the presence of an unanticipated airway difficulty during anesthesia (5–11). Airway management of symptomatic LTH in patients with Down’s syndrome may be challenging because of the patient’s lack of cooperation with airway management and underlying complications. We report the case of a patient scheduled for tonsillectomy in whom airway management was difficult because of lingual hypertrophy.

	Case Report

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An 11-yr-old girl with Down’s syndrome was scheduled for elective lingual tonsillectomy. The patient had a history of OSA and had undergone adenoidectomy at 3 and 9 yr of age. No history of difficult intubation was evident during these operations. After the second adenoidectomy, the patient presented with dyspnea every time she acquired an upper respiratory tract infection and with gradually deteriorating OSA, to the point where she was unable to sleep in the supine position. The patient was referred to our hospital after being diagnosed with LTH. Besides Down’s syndrome and OSA, the medical history included Moyamoya disease and a small atrial septal defect with moderate aortic regurgitation, which did not limit everyday activity. Magnetic resonance imaging, which was performed previously, revealed a small infarction in the left parietal lobe; however, no paralytic symptoms were evident. Transient cerebral ischemic attacks were controlled by aspirin and dipyridamole.

Preoperative fiberoptic laryngoscopy performed by an otolaryngologist demonstrated multiple lobular hypertrophied lingual tonsils at the base of tongue. The epiglottis was edematous, and the glottis could not be identified. On physical examination, the tongue was large, and a Mallampati score of III was determined. Head and neck movement were not limited, and there was no suggestion of atlantoaxial instability on radiograph. Although aspirin and dipyridamole were discontinued 7 days before surgery, bleeding time was slightly prolonged (5 min). Awake fiberoptic intubation was not considered to be an appropriate choice because of the patient’s developmental delay and inability to cooperate. Furthermore, the parents wished to avoid elective tracheostomy unless it was required in the case of a life-threatening emergency. In addition, the otolaryngologist requested nasotracheal intubation to facilitate the operative procedure.

At the induction of anesthesia, a laryngeal mask airway (LMA), laryngeal tube, fiberoptic bronchoscope, endoscopy mask, percutaneous cricothyrotomy kit, jet ventilator, and surgical tracheostomy were prepared in case of failed intubation. Anesthesia was induced with IV propofol 1 mg/kg, followed by 3%–4% sevoflurane in 100% oxygen so as not to induce apnea. Mask-assisted ventilation was possible during spontaneous respiration but was difficult when spontaneous breathing stopped.

After topical anesthesia of the lingual surface with 8% lidocaine spray, rigid direct laryngoscopy was attempted. The epiglottis could not be exposed because of hypertrophied tonsillar tissues, and we avoided advancing the laryngoscope blade by force so that tonsillar bleeding would be prevented. Insertion of an LMA was considered inappropriate because of periglottic obstruction and bleeding tendency. After topical application of phenylephrine to the nasal mucosa, we therefore performed fiberoptic bronchoscopy through the nasal cavity under spontaneous breathing in combination with jet ventilation through the oropharynx. The fiberscopic view was also poor and the epiglottis was not visible because of excessive tonsillar tissues. However, after elevation of the mandible, the vocal cord was identified, and a cuffed, reinforced 5.5-mm-inner-diameter tracheal tube was successfully placed through the fiberoptic bronchoscope. Oxygenation was adequate throughout the laryngoscopy and intubation. Anesthesia was maintained with sevoflurane in 50% oxygen with air, and the patient was paralyzed with vecuronium bromide as necessary. End-tidal carbon dioxide tension was kept at 40 mm Hg during the operative procedure.

After surgery, the patient was kept tracheally intubated because postoperative supraglottic edema was anticipated. Supraglottic edema was observed from the first to the sixth postoperative days, and the patient was extubated in the operating room on the seventh day. Symptoms of OSA gradually subsided, and the patient was discharged from the hospital uneventfully.

	Discussion

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Because the presence of LTH may be overlooked, the condition may be unanticipated and manifest on the induction of anesthesia (5–11). Indeed, a case report has described cerebral anoxia resulting in patient death because of the "cannot intubate-cannot ventilate" situation (11). Therefore, the first-choice approach to the airway in symptomatic LTH may be tracheostomy (12). However, the decision to perform a preliminary tracheostomy is not easy, especially in a pediatric patient. In cases of LTH, the decision to secure the airway with tracheostomy or naso-/orotracheal intubation depends on the individual background of the surgeon, the anesthesiologist, and patient issues.

Use of LMA is controversial in patients with LTH. Although there are several reports of successful use of LMA in patients with upper airway obstruction secondary to a lingual tonsil (6,8), Fundingsland and Benumof (7) reported a case in which LMA was of only limited efficacy as a ventilatory conduit. Furthermore, Asai et al. (10) cautioned that insertion of LMA and subsequent blind intubation could potentially damage periglottic tissues. Davies et al. (8) also reported that repeated insertions of LMA between intubation attempts can be traumatic and cause edema of the airway. In this case, we avoided using LMA despite difficult mask ventilation because of the potential bleeding tendency of the tonsillar tissues. We believe that the major reasons for successful fiberoptic intubation in this case were to avoid damaging tonsillar tissue by inserting an oral airway, to refrain from tracheal tube insertion under poor laryngeal visualization on a rigid blade, and to maintain spontaneous breathing.

Although the preoperative conditions in this case other than airway conditions were diverse, Moyamoya disease and cardiac anomalies did not significantly affect the course of anesthesia other than making the administration of antiplatelet therapy necessary.

It is not certain whether LTH is associated with Down’s syndrome. A compensatory mechanism for hypertrophy after adenotonsillectomy (3,13,14) and repeated upper respiratory tract infections was considerable in this case. Because LTH is apt to be not recognized, careful preoperative airway screening is crucial in patients with Down’s syndrome who have a history of adenotonsillectomy. Fiberoptic nasotracheal intubation while maintaining spontaneous breathing under inhaled anesthesia is one of the recommended methods of securing the airway in uncooperative pediatric patients with LTH.

	References

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9. Ovassapian A, Glassenberg R, Randel GI, et al. The unexpected difficult airway and lingual tonsil hyperplasia: a case series and a review of the literature. Anesthesiology 2002; 97: 124–32.[ISI][Medline]
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11. Jones DH, Cohle SD. Unanticipated difficult airway secondary to lingual tonsillar hyperplasia. Anesth Analg 1993; 77: 1285–8.[Free Full Text]
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Anesthesia & Analgesia® is published for the International Anesthesia Research Society® by Lippincott Williams & Wilkins with the assistance of Stanford University Libraries' HighWire Press®. Copyright 2006 by the International Anesthesia Research Society. Online ISSN: 1526-7598   Print ISSN: 0003-2999