J Oral Maxillofac Surg59:940-941, 2001

Macroglossia Following PalatoplastyCausing Upper Airway Obstruction:

Case ReportRuchi Gupta, MD, DNBE,* Balbir Chhabra, DA, MS,†

Ravi K. Mahajan, MS, Mch,‡ and Nandini, MD§

Because the incidence of cleft lip and palate is 1 in800 live births, palatoplasty is a common surgicalprocedure in the pediatric age group.1 Although pal-atoplasty in children with syndromes can cause vari-ous problems, anesthetic management of cleft palaterepair in pediatric patients with a normal airway israrely complicated.2,3 Common postoperative compli-cations in these children include hemorrhage andupper airway obstruction mainly caused by laryngo-spasm or edema.4 Macroglossia, a rare complication,is related to the duration of the surgery. We report acase of massive lingual swelling in an otherwisehealthy child shortly after routine palatoplasty of 1hour and 30 minutes duration.

Report of Case

A 2.5-year-old, male child weighing 15 kg presented forelective cleft palate repair. He had been delivered vaginallyat term following an uncomplicated pregnancy. His birthweight was 2.9 kg, and he had left-sided complete cleft lipand palate. His lip and anterior palate had been repaired atthe age of 1 year. There had been no anesthetic or surgicalcomplication at that time.

Preoperatively, his general physical and systemic exami-nation were essentially normal. The routine laboratory in-vestigations were within normal limits. He was given syrupof trimeprazine, 30 mg orally, as a premedicant 2 hoursbefore surgery.

After induction with oxygen, nitrous oxide (40:60), and2% halothane, an intravenous infusion of 5% dextrose in

normal saline was started at 100 mL/hr. After the child wasgiven 30 mg suxamethonium, the trachea was intubatedwith a 20F gauge oxford tube (Rusch, West Germany)without any difficulty. Thereafter, 1 mg pancuronium and7.5 mg meperidine were given intravenously, and the patientwas maintained on positive-pressure ventilation with oxygen,nitrous oxide, and 0.5% halothane.

The child was placed in the horizontal position with theneck hyperextended. A Dingman-Dott self-retaining tongueretractor (Padgett, Kansas City, MO) was inserted to opti-mize surgical exposure. The palatal shelves were infiltratedwith 5 mL of 1.0% lidocaine with 1:400,000 adrenalin, andan V-Y pushback palatoplasty was then performed. Theoperation was technically difficult, but was still completedin 1.5 hours. The tongue retractor was in place throughoutthe entire time. Blood loss was only 30 mL, and a total of210 mL of fluid was given.

Near the end of the operation, the surgeon noticed bluishdiscoloration of the tongue. Immediately, oxygen saturation(SaO2) and vital signs were checked, and the chest wasauscultated, but no abnormality was detected. As soon asthe retractor was released, the discoloration disappeared.

The residual neuromuscular block was antagonized withneostigmine and atropine, and the patient was extubatedonce fully awake. The tonsils were enlarged, but were notcausing any airway compromise. He was observed in therecovery room and shifted to the ward after 1 hour. Half anhour later, the patient showed the features of respiratoryobstruction and tongue edema. There was no evidence ofan allergic reaction, such as a rash, generalized edema, orerythema, and the child was otherwise hemodynamicallystable. He was placed in a left lateral position and a glos-sopexy was done. The obstruction was relieved with thesemeasures.

The child was carefully watched and heavy doses ofsteroids were started. While under observation, the tonguecontinued to increase in size over 4 hours and startedprotruding out of the oral cavity. Because he did not showany further features of obstruction, the vital signs werestable, and swelling did not further increase in size, it wasdecided to manage the patient conservatively, with facilitiesfor urgent tracheostomy available. From the next day on-ward, the tongue edema started reducing, and the tonguebecame normal on the fourth postoperative day. The pa-tient was discharged on the seventh postoperative day.

Discussion

Postoperative, massive, lingual swelling is an un-usual complication that can lead to life-threateningairway obstruction.5 Macroglossia may also occurfrom rapid expansion of silent tongue cysts or asymp-

*Lecturer, Department of Anaesthesiology and Critical Care, Pt.

B.D. Sharma, PGIMS, Rohtak (Haryana), India.

†Professor and Head, Department of Anaesthesiology and Criti-

cal Care, Pt. B.D. Sharma, PGIMS, Rohtak (Haryana), India.

‡Reader and Head, Department of Plastic Surgery, Pt. B.D.

Sharma, PGIMS, Rohtak (Haryana), India.

§Senior Resident, Department of Anaesthesiology and Critical

Care, Pt. B.D. Sharma, PGIMS, Rohtak (Haryana), India.

Address correspondence and reprint requests to Dr Gupta: 7/9J,

Medical Enclave, Pt. B.D. Sharma PGIMS, Rohtak-124001, India,

e-mail:raviruchi@vsnl.com

© 2001 American Association of Oral and Maxillofacial Surgeons

0278-2391/01/5908-0018$35.00/0

doi:10.1053/joms.2001.25042

940

tomatic lymphangiomas caused by injury or infec-tion.6,7 It can also be seen after difficult and traumaticoral intubation, dental procedures, and prolongedneurosurgical operations done in the sitting posi-tion.8-12 Hypersensitivity reaction to various agentsused for sterilizing laryngoscopes and drugs, such asdroperidol and angiotensin-converting enzyme inhib-itors, can also cause macroglossia.13,14 However, inthis case, none of the previous reasons were validand, because there were no signs of allergy, an immu-nologic mechanism was unlikely.

Postoperative macroglossia after palatoplasty isa rare complication; the development of which isclosely related to the duration of the surgical repair.In the literature, there have been 2 reports of lingualswelling in patients having various syndromes and 5in patients with normal airways, but in all of these thesurgery lasted longer than 3.5 hours, and Dingmanretractor was used to provide surgical exposure.2 TheDingman-Dott tongue retractor holds the mouth openby exerting pressure on the tongue and alveolar ridge.Excessive pressure exerted for prolonged periods, orinappropriate size of the retractor blade, impairs thevenous and lymphatic drainage of the tongue and re-sults in mucosal edema.2,5,6,15 Although in this casethe surgery was completed in the normal time of 1.5hours, it was technically difficult, and excessive pres-sure was applied on the base of tongue to obtain ade-quate exposure. Excessive retraction, in conjunctionwith the hyperextended neck, could have impairedthe venous drainage from the tongue resulting in thebluish discoloration and later the lingual swelling.

Lingual swelling can occur immediately after re-moval of the mouth gag or it can be delayed for 2 to2.5 hours; in which case it can be dangerous becauseit will not be detected until the patient is on the ward.In our case also, the swelling was noticed on the ward1.5 hours after completion of surgery.

A massively swollen tongue can occlude the oro-pharynx and nasopharynx, resulting in total airwayobstruction and cerebral anoxia.2 Management of anobstructed airway secondary to massive lingual swell-ing includes glossopexy, placement of a nasopharyn-geal airway, or tracheal intubation. However, in diffi-cult cases, where retrograde intubation, use of anIaryngeal mask airway, or flexible fiberoptic-guidedintubation is not possible, tracheostomy or cricothy-rotomy are the other alternatives.16

We recommend that even in surgeries that last 2hours or less and that are otherwise technically diffi-cult, the tongue retractor should be released at leastfor 5 minutes each hour to facilitate venous drainageand decrease trauma, especially in cases where ve-nous stasis is observed. An extreme Trendelenbergposition should be avoided as long as possible. Anappropriately sized blade in relation to the oral cavityshould be chosen. It is further suggested that in all thecleft palate repairs, the entire oropharyngeal cavity,especially the base of the tongue, should be examinedat the completion of surgery. If there is bluish discol-oration or edema of the tongue observed during thesurgery, then these children should be kept underclose observation postoperatively for any kind of ob-struction, and all measures to secure a patent airwayshould be kept ready if this occurs.

References1. Steward RE: Craniofacial malformations—clinical and genetic

considerations. Pediatr Clin North Am 25:485, 19782. Chan MTV, Chan MSH, Mui KSY, et al: Massive lingual swelling

following palatoplasty. Anaesthesia 50:30, 19953. Levin RM: Anesthesia for cleft lip and cleft palate. Anesthesiol-

ogy Rev 6:25, 19794. Wray C, Dann J, Holtmann B: A comparison of three technics of

palatorrhaphy: In-hospital morbidity. Cleft Palate J 16:42, 19795. Patane PS, White SE: Macroglossia causing airway obstruction

following cleft palate repair. Anesthesiology 71:995, 19896. Bell C, Oh TH, Loeffler JR: Massive macroglossia and airway

obstruction after cleft palate repair. Anesth Analg 67:71, 19887. La Bagnara J: Cysts of the base of the tongue in infants: An

unusual cause of neonatal airway obstruction. OtolaryngolHead Neck Surg 101:108, 1989

8. McGoldrick KE, Donlon JV: Sublingual hematoma followingdifficult laryngoscopy. Anesth Analg 58:343, 1979

9. Means LJ, Jones JE, Rao CC: Sublingual emphysema complicat-ing dental anesthesia. Anesth Analg 64:737, 1985

10. McAllister RG: Macroglossia—a positional complication. Anes-thesiology 40:199, 1974

11. Mayhew JF, Miner M, Katz J: Macroglossia in a 16 month-oldchild after a craniotomy. Anesthesiology 62:683, 1985

12. Mocre JK, Chaudhri S, Moore AP, et al: Macroglossia andposterior fossa disease. Anaesthesia 43:382, 1988

13. Clark RJ: Tongue swelling with droperidol. Anaesth IntensiveCare 21:898, 1993

14. Jain M, Armstrong L, Hall J: Predisposition to and late onset ofupper airway obstruction following angiotensin-converting en-zyme inhibitor therapy. Chest 102:871, 1992

15. Lee JT, Kingston HGG: Airway obstruction due to massivelingual oedema following cleft palate surgery. Can Anaesth SocJ 32:265, 1985

16. Cozine K, Stone JG: The take-back patient in ear, nose andthroat surgery. Anesthesiol Clin North Am 11:651, 1993

GUPTA ET AL 941