Severe Glossal Edema After Primary Palatoplasty

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GLOSSAL EDEMA PRIMARY PALATOPLASTY

This made it possible to repair the defect using the expanded skin. When the incision was made to remove the tumor, we tried to preserve the normal skin as much as possible. An extra incision from the lateral neck to the nuchal region made the flap extend forward. Next, the submandibular incision was extended medially and inferiorly. This made the advancement flap in the submandibular region move upward easily. The defect was repaired using these local flaps. Local flaps possess the following advantages. First, the color and texture of the flaps will be similar to the skin surrounding the defect. Also, they avoid surgical injury and the functional and cosmetic deformities of the donor areas resulting with distal or free flaps. Finally, this procedure is simple and diminished the time requirements. Giant lipomas involving multiple areas of the face and neck are extremely rare. Computed tomography and magnetic resonance imaging are both accurate preoperative investigative methods. Preoperative histologic biopsy will also be helpful in determining the operative management. Surgical management of a giant lipoma involving the cervical sheath and the seventh cranial nerve branch should be performed by experienced surgeons because of the need for meticulous dissection of the cervical sheath and the facial nerve branches. The identification and preservation of most of the parotid gland not involved by tumor might contribute to a better postoperative aesthetic and functional result. The use of local flaps is preferable for defect repair in selected cases.

Acknowledgments We thank the charity foundation of the First Affiliated Hospital of Kunming University of Medical Science for the total financial support for the treatment of this patient.

References 1. Kransdorf MJ: Benign soft-tissue tumors in a large referral population: distribution of specific diagnoses by age, sex, and location. AJR Am J Roentgenol 164:395, 1995 2. El-Monem MH, Gaafar AH, Magdy EA: Lipomas of the head and neck: Presentation variability and diagnostic work-up. J Laryngol Otol 120:47, 2006 3. Som PM, Scherl MP, Rao VM, Biller HF: Rare presentations of ordinary lipomas of the head and neck: A review. AJNR Am J Neuroradiol 7:657, 1986 4. Lewis WR: Fibrolipoma of jaw and neck. Ann Surg 43:500, 1906 5. Korentager R, Noyek AM, Chapnik JS, Steinhardt M, Luk SC, Cooter N: Lipoma and liposarcoma of the parotid gland: Highresolution preoperative imaging diagnosis. Laryngoscope 98: 967, 1988 6. Weiner GM, Pahor AL: Deep lobe parotid lipoma: A case report. J Laryngol Otolaryngol 109:772, 1995 7. Ozcan C, Unal M, Talas D, Gorur K: Deep lobe parotid gland lipoma. J Oral Maxillofac Surg 60:449, 2002 8. Kimura Y, Ishikawa N, Goutsu K, Kitamura K, Kishimoto S: Lipoma in the deep lobe of the parotid gland: A case report. Auris Nasus Larynx 29:391, 2002 9. Ulku CH, Uyar Y, Unaldi D: Management of lipomas arising from deep lobe of the parotid gland. Auris Nasus Larynx 32:49, 2005 10. Gritzmann N, Schratter M, Traxler M, Helmer M: Sonography and computed tomography in deep cervical lipomas and lipomatosis of the neck. J Ultrasound Med 7:451, 1988 11. Chikui T, Yonetsu K, Yoshiura K, et al: Imaging findings of lipomas in the orofacial region with CT, US, and MRI. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 84:88, 1997

J Oral Maxillofac Surg 67:1326-1328, 2009

Severe Glossal Edema After Primary Palatoplasty Shahid R. Aziz, DMD, MD,* and Vincent B. Ziccardi, DDS, MD† At most American centers, primary palatoplasty to repair a cleft palate is generally performed in patients by the age of 12 months to allow for appropriate speech development. Surgical access for the palato-

plasty is often obtained with a Dingman mouth gag, which allows for maximum access and visibility of the surgical field. There are multiple techniques used by cleft surgeons to repair cleft palates, including War-

Received from the Department of Oral and Maxillofacial Surgery, New Jersey Dental School, University of Medicine and Dentistry of New Jersey, Newark, NJ. *Associate Professor. †Associate Professor and Chair. Address correspondence and reprint requests to Dr Aziz: Department of Oral and Maxillofacial Surgery, New Jersey Dental

School, University of Medicine and Dentistry of New Jersey, 110 Bergen St, Room B854, Newark, NJ 07103; e-mail: [email protected] edu © 2009 American Association of Oral and Maxillofacial Surgeons

0278-2391/09/6706-0030$36.00/0 doi:10.1016/j.joms.2008.12.046

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dill-Kilner, von Langenbeck, Bardach, and Furlow Zplasty. Common complications of these procedures in the immediate postoperative phase include bleeding and oropharyngeal edema. Airway obstruction is rare and most commonly results from laryngeal edema from a traumatic intubation. This case report documents severe glossal edema immediately after primary palatoplasty, causing airway obstruction and requiring reintubation in the immediate postoperative period. This is a rarely documented complication of palatoplasty, with only 4 reports noted in the medical literature since 1950, 3 in the anesthesia literature and 1 in the surgical literature.1-4

Report of a Case An 11-month-old, 10-kg male infant born with a complete left cleft lip, alveolus, and palate was brought to the operating room for primary palatoplasty. He was intubated via an oral approach, by use of a 4-mm endotracheal tube. He was prophylactically administered intravenous ampicillin and 2 mg of dexamethasone. A Dingman self-retaining mouth gag was placed in atraumatic fashion by use of the medium-sized grooved endotracheal tongue blade to secure the endotracheal tube and retract the tongue out of the operative field. The cleft palate was repaired via the 2-finger flap technique advocated by Bardach.5 The total surgical time was approximately 2 hours 30 minutes. The patient was subsequently extubated without incident and brought directly to the pediatric intensive care unit (PICU), per our usual protocol for infant cleft palate repair. On arrival to the PICU, the patient was on 40% oxygen via a face mask with an oxygen saturation of 92% to 100%; however, marked tachypnea with 40 to 50 breaths per minute. At this point, glossal edema, inspiratory stridor, and abdominal breathing were noted. The PICU staff administered racemic epinephrine via a nebulizer, which partially relieved the stridor. One hour postoperatively, the patient continued to maintain an oxygen saturation of 90% to 100% while receiving 40% oxygen via a face mask, with continued tachypnea in addition to noted macroglossia with the tongue protruding from the mouth. On intraoral examination, the tongue was firm and dry from air exposure, and it was not possible to visualize the palatal repair beyond the hard palate. A decision was made at this point to reintubate the patient for airway protection. The patient was then returned to the operating room and reintubated fiberoptically, and he was kept sedated on a ventilator (Fig 1). The patient remained in the intensive care unit for the next 5 days, intubated and sedated. During this time, he was administered intravenous fluids, sedation medications, antibiotics, and 1.5 mg of dexamethasone every 6 hours. The glossal edema was noted to have started resolving on postoperative day 2 and had completely resolved on postoperative day 4, at which time an air leak around the endotracheal tube was appreciated, allowing for extubation. The patient remained hospitalized for 2 more days until adequate oral intake was noted. At discharge, he was tolerating liquids and breathing at 100% oxygen saturation on room air. The palatal repair appeared to be intact at the time of discharge and has

FIGURE 1. Two hours after completion of palatoplasty, immediately after reintubation: lateral view (A) and anterior view (B). Marked glossal edema was noted. Aziz and Ziccardi. Glossal Edema Primary Palatoplasty. J Oral Maxillofac Surg 2009.

remained so 6 months after surgery during routine follow-up.

Discussion Glossal edema after primary palatoplasty is a rare and potentially life-threatening complication in the immediate postoperative period.2 It is hypothesized that the edema is a direct result of compression of the tongue by the Dingman mouth gag (Fig 2). The Dingman mouth gag, named after Reed O. Dingman, an oral and plastic surgeon from the University of Michigan (Ann Arbor, MI), is a device that allows for excellent visualization and access to the palate and

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FIGURE 2. Placement of Dingman mouth gag at beginning of case. The tongue is retracted down; compression of the tongue gives the glossal tissue an engorged strawberry color. Aziz and Ziccardi. Glossal Edema Primary Palatoplasty. J Oral Maxillofac Surg 2009.

oropharynx by retracting the cheeks laterally, stabilizing the anterior maxillary alveolus, and retracting the tongue inferiorly under the endotracheal tube. Retraction of the tongue is achieved by a tongue blade with a groove centered to fit the endotracheal tube. This blade pushes inferiorly on the tongue and tongue base. Too much compression can cause pressure necrosis of the glossal tissue and impede lymphatic and venous outflow from the tongue, resulting in significant glossal edema and potential airway obstruction.3 In addition, the classic Rose positioning of the head when repairing a palate by placing the patient supine with the neck extended and head slightly off the operating table further reduces venous and lymphatic drainage.6 The glossal edema that develops is often swift in onset—within 2 hours postoperatively—and can cause airway obstruction. Edema of the glossal musculature causes the tongue to protrude superiorly and anteriorly out of the mouth.4 Prevention of glossal edema from palatoplasty requires minimizing the compression of the tongue while allowing for visualization of the surgical field. This scenario may be

GLOSSAL EDEMA PRIMARY PALATOPLASTY

difficult to achieve during palate repair, given the nature of the surgery. A reasonable compromise may be to release the Dingman mouth gag hourly for 5 to 10 minutes during palatoplasty to allow for decompression and lymphatic drainage of the tongue. In addition, at the end of the surgery, before extubation but after removal of the Dingman mouth gag, inspection of the tongue to evaluate for areas of edema or necrosis is warranted. If the tongue seems edematous, the extubation should be postponed for 2 to 3 hours to determine whether glossal edema will occur. If glossal edema does occur, the patient should be kept intubated in a monitored setting and administered intravenous steroids until the edema resolves. Only when the edema resolves and an air leak is noted around the endotracheal tube should extubation be considered. Reintubation in a patient with glossal edema after cleft palate repair can be extremely challenging and may traumatize the palate repair. Glossal edema is a rare and potentially life-threatening complication of palatoplasty. It is thought to result from compression of the tongue from the Dingman mouth gag. To prevent this, releasing the Dingman mouth gag during surgery will allow for decompression of the tongue. In addition, before extubation, if there is a concern of glossal edema developing, the patient should be kept intubated until the edema has resolved and a safe extubation can be obtained.

References 1. Gupta R, Chabra B, Mahajan R, et al: Macroglossia following palatoplasty causing upper airway obstruction: Case report. J Oral Maxillofac Surg 59:940, 2001 2. Lee J, Kingston H: Airway obstruction due to massive lingual oedema following cleft palate surgery. Can Anaesth Soc J 32:265, 1985 3. Bell C, Oh TH, Loeffler JR: Massive macroglossia and airway obstruction after cleft palate repair. Anesth Analg 67:71, 1988 4. Antony A, Sloan GM: Airway obstruction following palatoplasty: Analysis of 247 consecutive operations. Cleft Palate Craniofac J 39:145, 2002 5. Bardach J. Two flap palatoplasty: Bardach’s technique. Oper Tech Plast Reconstr Surg 2:211, 1995 6. Greene A, Kreuter M, Mulliken J: Professor Edmund Rose: His position in surgery. Plast Reconstr Surg 111:383, 2003

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