Abstract

Cleft lip palate is a congenital anomaly that requires surgical reconstruction, and patients rarely develop tongue edema after palatoplasty. We describe a 1-year-and-8-month-old boy who underwent palatoplasty for left-sided cleft lip palate accompanied by exudative otitis media. Although previous reports have described that tongue edema usually sets in early after surgery, the symptoms of edema persisted more than 4 hours postoperatively in our case. We suggest that careful observation for edema is necessary for 24 hours at least after palatoplasty.

Keywords

cleft palate; reconstructive surgical procedures; anesthesiapediatrics;

1. Introduction

Cleft lip palate is a congenital anomaly with incidence of about 1 in 500–700 neonates. Palatoplasty, a surgical treatment for cleft palate, is performed at the age of 1 year and 6 months to 2 years, and the main postoperative complications include infection, bleeding, and oropharyngeal edema.¹ Tongue edema usually occurs early after surgery, and subsequent upper airway obstruction causing a serious condition is infrequently observed.² We report here a child who developed tongue edema as a late postoperative complication after palatoplasty.

2. Case report

The patient was a 1-year-and-8-month-old boy, 81 cm tall and weighing 12 kg. Left-sided cleft lip palate was diagnosed at birth, and the patient was brought to our hospital for the preparation of Hotz’ plate and suckling guidance. Thereafter, suckling became possible with stabilization by a Hotz’ plate, and labioplasty was performed at the age of 3 months. There were no remarkable complications during or after this surgery.

No premedication was administered, and anesthesia was induced slowly with nitrous oxide and sevoflurane in oxygen. Intubation was achieved easily after administering a muscle relaxant, and then manual ventilation was performed using a Jackson–Rees circuit. Thereafter the patient was placed in the supine position with the head hanging, and a Dingman mouth gag was applied by the surgeon. Anesthesia was maintained with nitrous oxide and sevoflurane in oxygen, and there were no intraoperative complications. Surgery was completed uneventfully, and there was no abnormal vascularity noted in tongue. The mouth gag was removed at the end of surgery, and spontaneous respiration promptly restarted. There was no abnormality in the respiratory state or hemodynamics after extubation. The surgical duration was 4 hours and 47 minutes; anesthesia time was 6 hours and 36 minutes.

The patient was brought to the ICU and observed carefully over more than 2 hours. During this period, there was no abnormality in the oral cavity or compromise of respiration. The patient was then returned to a general ward, and tongue edema was noted 2 hours after that. The tongue became too large to remain in the oral cavity and subsequently began to protrude out of the lips (Fig. 1). Furthermore, edema extended to the oral floor, causing neck swelling. Since pharyngeal stridor was heard, hydrocortisone was administered and percutaneous oxygen saturation was monitored continuously. Although edema persisted overnight, it did not deteriorate to obstruct the upper airway, and was gradually alleviated on the next day with disappearance of pharyngeal stridor. On the 4th postoperative day, tongue edema was almost fully resolved, and oral intake became possible.

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Fig. 1. Tongue edema over more than 4 hours after palatoplasty.

3. Discussion

Past reports on tongue edema after palatoplasty have suggested the necessity of careful observation for 2 hours after surgery, because tongue edema usually occurs immediately or within 2 hours after the end of surgery in most cases reported (Table 1). However, in our patient, tongue edema occurred over more than 4 hours after surgery. Trauma during difficult intubation, emphysema caused by dental abrasion instruments, suppression of circulation by mouth gags, and impairment of the blood or lymph flow in the supine position with the head hanging are considered possible causes of edema of the tongue and oral floor after palatoplasty.3, 4, 5 In the present case, intubation was performed easily and smoothly, and there was no sign of emphysema on postoperative radiography. Therefore, application of the mouth gag and sustained head-hanging posture during prolonged surgery were considered to be possible causes of the tongue and mouth edema. Schettler et al. reported that the risk of upper airway obstruction due to edema of the tongue or oral floor increases 10 times, from 0.1% to 1%, if the surgical duration exceeds 2 hours.⁶ Lee et al. recommended removal of the mouth gag for about 5 minutes every 1 hour as a countermeasure against severe upper airway obstruction.⁷ Although we have not had the experience of the tongue edema after palatoplasty in the past, the present surgery which lasted over more than 4 hours reconfirms the necessity of such an approach in prolonged palatoplasty.

Table 1. Time when tongue edema occurred after surgery.

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Tongue swelling after palatoplasty mostly occurs immediately or within 2 hours after operation,8, 10 and there are some opinions that we had better be unhurried for extubation.2, 10 In particular, when the operation time gets longer like our instance, the tongue edema is easy to occur because of impairment of the blood or lymph flow in the supine position with the head hanging. However, the damage of the surgical site could be caused by the retaining tube. Therefore, if the operation time was shorter, early extubation in the presence of restoration to spontaneous breathing, should have been done.

There is controversy about re-intubation in airway obstruction after palatoplasty. Many reports say that re-intubation should be done early.7, 8, 9, 11, 12 On the other hand, Bell et al.¹¹ and Chan et al.³ stated that patients after palatoplasty are in danger of tissue damage or bleeding at the surgical site in the act of re-intubation, which may result in a more serious event. Therefore unless percutaneous oxygen saturation deteriorates, mechanical stimulation by re-intubation should better be avoided.¹⁰ In case of complicated operation or predictable difficult airway, the patients should be under sedation without extubation.2, 10 If the patient has airway obstruction, with difficult intubation, needle cricothyroidotomy should be performed. However, such airway management, which is very likely accompanied by severe complications, has to be avoided with utmost effort.³

It is important for medical staff to realize that edema may occur in the late postoperative recovery period although it is not yet clear whether the causes are the same as those of immediate edema. Careful observation for late onset of edema after palatoplasty, especially after prolonged surgery as in our case, should be considered to be a necessity. Although we extubated our patient immediately after surgery, extubation should have been delayed considering the possibility of edema occurring late after surgery.

4. Conclusion

Here, we present a child who developed tongue edema several hours after palatoplasty under general anesthesia. This case emphasizes the possibility of tongue edema that could occur several hours after surgery, and the necessity of sufficient postoperative observation as well as countermeasures for prevention.