Sir,
Hemangiomas along with lymphangiomas constitute 30% of oral cavity tumors in the pediatric population.[1] A 3-year-old girl child presented with swelling over the right side of the tongue since birth which was gradually increasing in size. There were no complaints of bleeding or airway obstruction. She was earlier treated with oral propranolol and multiple kenocort injections locally without any significant reduction in the size of the swelling. She was posted for debulking of the hemangioma. On examination, the tongue looked bulky and was protruding outside the oral cavity and the child was not able to close her mouth [Figure 1a]. The swelling was firm in consistency and was involving the right side of the tongue extending posteriorly. MRI imaging showed hemangioma involving the right buccal and masticator space with diffuse infiltration.
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Figure 1: (a) Hemangioma tongue protruding out of the oral cavity and (b) swollen tongue on postoperative day 1
The child was taken into theater and inhalation induction with 8% sevoflurane in oxygen was attempted. There was airway obstruction which was managed by pulling the tongue out and a mask was held over her face keeping the tongue pulled out. Once induced intravenous access was secured and glycopyrrolate was given. Laryngoscopy with C-MAC videolaryngoscope with tongue pulled out showed the glottis without difficulty. The trachea was then intubated nasally with 4.5-size cuffed Ring-Adair-Elwin endotracheal tube under videolaryngoscopic guidance. Fentanyl 2 mcg/kg and atracurium 0.5 mg/kg were given and anesthesia was maintained with isoflurane in an air-oxygen (1:1) mixture.
Hemangioma involving the anterior and lateral parts of the tongue was excised. Reshaping and recreating the tip of the tongue was performed. The posterior part of the tongue was untouched and there was adequate space without any obstruction. The debulked tongue remained inside the oral cavity at the end of surgery. Overnight ventilation and trial of extubation the next day were considered initially. But a decision for elective tracheostomy was made in view of anticipated postoperative swelling of the tongue, which if extended posteriorly, would make reintubation practically impossible. The child was tracheostomized with a 4.5 binova tube. Neuromuscular blockade was reversed and she was shifted to postoperative ICU on T piece.
In the immediate postoperative period, the child was able to close her mouth. Gradually, tongue size increased, and over a few hours, swollen tongue occupied the entire oral cavity and protruded outside the mouth as well [Figure 1b]. Since the child was already tracheostomised, there was no airway compromise.
The gravest complication of macroglossia is airway compromise.[2] Maintenance of spontaneous ventilation during induction is crucial. Pulling out the tongue gives room for the laryngoscope to be passed behind the tongue and eases the passage of the endotracheal tube.[3] Following debulking of tongue hemangioma, postoperative swelling either due to edema or hematoma leading to airway compromise should be anticipated. The incidence of complications associated with tracheostomy is higher in children than in adults.[4] Although tracheostomy is mostly a life-saving procedure, late complications of pediatric tracheostomy like tracheal stenosis, tracheocutaneous fistula, tracheoesophageal fistula, and subglottic stenosis have significant morbidity.[5]
It is concluded that following tongue debulking surgery need of tracheostomy for maintaining the airway in the postoperative period should be carefully weighed against long-term complications and should be chosen if deemed lifesaving.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
References
1. Barrón-Peña A, Martínez-Borras MA, Benítez-Cárdenas O, Pozos-Guillén A, Garrocho-Rangel A. Management of the oral hemangiomas in infants and children: Scoping review Med Oral Patol Oral Cir Bucal. 2020;25:e252–61
2. Balaji SM, Balaji P. Large hemangioma of the tongue Indian J Dent Res. 2020;31:979–82
3. Hajipour A, Javid MJ, Saedi B. Airway management in a toddler with a giant hemangioma of the tongue Iran J Pediatr. 2012;22:551–4
4. Kremer B, Botos-Kremer AI, Schlondorff G. Indications, complications, and surgical techniques for pediatric tracheostomies — An update J Pediatr Surg. 2002;37:1556–62
5. Lubianca Neto JF, Castagno OC, Schuster AK. Complications of tracheostomy in children: A systematic review Braz J Otorhinolaryngol. 2020 S1808-8694 (20) 30247-0. doi: 10.1016/j.bjorl. 2020.12.006. Online ahead of print
