Massive lingual swelling following palatoplasty

Abstract

SummaryWe report a case of upper airway obstruction as a result of delayed massive lingual swelling following routine cleft palate repair in an otherwise healthy 12-month-old girl. We believe that ischaemia and venous congestion were the causes of macroglossia, after prolonged use of the Digman Dott tongue retractor. In any dificult and lengthy repair, we recommend the prophylactic insertion of a nasopharyngeal airway under direct vision by the surgeons after surgery to prevent potential upper airway obstruction. Key wordsAnaesthesia; paediatric. Complication; macroglossia. Airway; obstruction. Surgery; cleft palate.Palatoplasty is a relatively common procedure and the overall incidence of postoperative morbidity varies from 15 to 26% [I, 21. Haemorrhage and airway obstruction are the most frequent major complications [2]. Upper airway obstruction after cleft palate repair is most commonly associated with laryngospasm, but it is also a well recognised complication in patients with pre-existing craniofacial anomalies such as Pierre-Robin syndrome [3] and following primary pharyngeal flap reconstruction [4, 51. Life-threatening airway obstruction as a result of lingual swelling is unusual. Re-establishment of an adequate airway can be extremely difficult, if not impossible. In this report, we describe a case of massive lingual swelling shortly after routine palatoplasty in an otherwise normal child and we review the potential risk factors, pathogenesis, clinical course, preventive measures and the management of this complication. Case historyA 12-month-old, 8.6 kg girl presented for elective cleft palate repair. She had been delivered vaginally at term following an uncomplicated pregnancy. Her birth weight was 2.58 kg and she had a complete bilateral cleft palate. Apart from recurrent middle ear infections and delayed phonologic development, her perinatal history was uneventful. Pre-operatively, physical examination revealed a healthy girl with a central, wide soft palate cleft, but an otherwise normal airway. She was given premedication of trimeprazine 24 mg orally, 30 min before surgery.Following inhalational induction with isoflurane in oxygen and nitrous oxide, an intravenous infusion of 0.18% saline in 4.3% dextrose was started at 40ml.h-'. After administration of atracurium 5 mg and fentanyl 10 pg intravenously, the trachea was intubated easily with a 4.0-mm uncuffed oral-RAE tube (Mallinckrodt Lab. Ltd. Athlone, Ireland).The child was placed in the Trendelenberg position, with the neck hyperextended. A Digman Dott self retaining tongue retractor was inserted to optimise surgical exposure. The cleft edge was infiltrated with lignocaine 0.5% 5ml with 1 : 200 000 adrenaline. A V-Y pushback palatoplasty was performed. The operation was technically difficult because of the large cleft and relatively small mouth. The entire procedure lasted 3 h and 45 min, with the tongue retractor in place during most of the operation. Total blood loss was approximately 60 ml and was replaced with 70 ml of stable plasma protein ...

Massive lingual swelling following cleft palate repair

“…Sporadic reports of similar episodes were noted to cleft palate repair[1348–11] Even in the reported cases, most of them were in Franceschetti’s syndrome and Pierre Robin sequence where, the surgical intervention is more demanding due to micrognathia and glossoptosis and to have a proper surgical access surgeon injudiciously applies more forceful and lengthy tongue retraction. [3]…”

“…1998 suggested excessive pressure exerted on the base of tongue by the retractor producing glossal hematoma, ischemic necrosis of tongue muscles, venous stasis or lymphedema[39] Hyperextension of the head and Tredelenberg position[12] may also contribute to impaired arterial flow and decreased venous drainage of the tongue. [1312] Extreme Tredelenberg is highly detrimental especially when combined with high retractor pressure in prolonged surgery. Most of the authors believe that lingual edema is time dependent and Lee and Kingston suggest periodic release of the mouth gag to prevent prolonged ischemia of the tongue.…”

“…Most of the authors believe that lingual edema is time dependent and Lee and Kingston suggest periodic release of the mouth gag to prevent prolonged ischemia of the tongue. [113] Other suggested mechanisms of tongue swelling are trauma, allergy, infection and massive fluid load perioperatively. [7]…”

“…suggest inserting nasopharyngeal airway of appropriate size under the direct vision by the surgeon before extubation. [1] As part of our institutional protocol, we routinely put tongue sutures.…”

“…The main concerns in palatoplasty, at the time of extubation are hemorrhage and upper airway obstruction. [12] It is well-known that patients with Franceschetti syndrome and Pierre Robin sequence have increased risk for developing airway obstruction following palatoplasty. This phenomenon can be explained as due to shallow nasopharyngeal airway and insufficient maxillofacial growth at the time of repair.…”