INTRODUCTION

A child with diffusely enlarged tongue due to lymphangioma presents a unique challenge to the anesthesiologists. Anesthetic concerns include difficulty in mask ventilation, intubation, bleeding, extrinsic and intrinsic pressure on the airway causing distortion and enlarged upper Anesthetic management is often challenging in such patients because of difficult mask ventilation and airway management. We report a case of lymphangioma of tongue leading to macroglossia in a child. Anesthetic concerns including airway management, alternative airway equipment and possible complications are discussed.

CASE REPORT

A 2 year old male child weighing 10 kg reported to pediatric surgery with generalized swelling of tongue. Parents noticed a pea sized swelling over tongue immediately after birth. They consulted some private physician and took some medicines but no response was observed. Increase in size of the swelling was insidious. Child was delivered at full term in a village hospital. There was no significant antenatal history. Parents gave a history of progressive difficulty in feeding the child but there was no breathing difficulty. Local examination revealed an enlarged tongue protruding and keeping the mouth permanently open (Figures 1 and 2). The oro-dental hygiene was poor with teeth impressions on the surface of tongue. Mouth opening, inter-incisor gap and Mallampatti grading could not be elicited due to the enlarged tongue. On palpation the tongue was non tender and firm in consistency. Patient had a swelling of 2 × 4 cm in right parotid region raising the ear lobe. No other congenital abnormalities were present. All routine investigations were with in normal limits. Ultrasound showed hypertrophy of tongue with multiple cystic areas seen in parotid and submandibular gland. Magnetic resonance imaging revealed enlargement of tongue in anterior 2/3 region and hyper intensity on T2W and T1W images. It showed heterogeneous enhancement on CEMR. Lymphangiomas seen in bilateral parotid and submandibular glands with enlarged right parotid gland. Rest of the systemic examination was normal. Child was pre-medicated with intranasal midazolam 0.5 mg 30 min prior to shifting to the operation theatre. On arrival to the operating room, routine monitors, including a pulse oximeter, non-invasive blood pressure cuff and electrocardiographic monitor were applied. Difficult airway cart, and tracheostomy set were arranged. Suture was kept ready. It was planned to pull the tongue with suture if obstruction occurred. Child was induced with sevoflurane 2-6% in increments using size 3 transparent facemask (Figure 3). We used bigger size face mask, i.e., size 3 to oxygenate and ventilate the patient, as we were unable to maintain proper seal with smaller mask because of large protruding tongue. It was difficult to assist ventilation in supine position so he was turned to right lateral position. Intravenous line was secured with 24 G cannula in right hand. Injection glycopyrrolate 50 µg and fentanyl 20 µg were given IV. After checking for ability to ventilate, check laryngoscopy done and succinylcholine 20 mg given IV. Child was successfully intubated orally with 4.5 mm uncuffed tube in a single attempt. Anesthesia was maintained with N₂O 67%, oxygen 33%, isoflurane 1% and atracurium. Injection paracetamol 150 mg IV, hydrocortisone 20 mg IV was given intraoperatively. The inverted V shaped anterior half of the tongue was removed. The tumor had large cystic spaces filled with lymph like fluid. After completion of surgery, trachea was extubated without any adverse event. The intra-operative and recovery period were uncomplicated and baby was shifted to the ward after being  observed for  2  h  in  the recovery room. Child’s stay in the hospital was uneventful and was discharged home after 5 days.

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