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Lymphatic Malformation of the Lingual Base and Oral Floor

Edwards, Paul D. M.D.; Rahbar, Reza D.M.D., M.D.; Ferraro, Nalton F. D.M.D., M.D.; Burrows, Patricia E. M.D.; Mulliken, John B. M.D.

Plastic & Reconstructive Surgery: June 2005 - Volume 115 - Issue 7 - pp 1906-1915
doi: 10.1097/01.PRS.0000165071.48422.A4
Original Articles: Reconstructive, Head/Neck

Background: Lymphatic malformation of the tongue and floor of the mouth is associated with chronic airway problems, recurrent infection, and functional issues related to speech, oral hygiene, and malocclusion. There are no accepted anatomic guidelines or treatment protocols.

Methods: This retrospective review focused on anatomic extent, treatment, complications, and airway management in 31 patients with lymphatic malformation of the lingual base and oral floor.

Results: Involved adjacent structures included the neck (77 percent), mandible (41 percent), face (42 percent), lips (10 percent), pharynx (45 percent), and larynx (26 percent). Fifty-eight percent of patients required tracheostomy during infancy; decannulation was possible in two-thirds of these patients. Management included resection alone (42 percent), resection and sclerotherapy (26 percent), resection and laser coagulation (16 percent), sclerotherapy and laser coagulation (16 percent), and resection and radiofrequency ablation (3 percent). Resection involved the neck (58 percent), floor of the mouth (52 percent), and tongue (42 percent); there were often multiple procedures. Aspiration was tried with little success in 10 percent of patients. Virtually all patients had residual abnormal lymphatic tissue. Complications and posttherapeutic problems included infection (81 percent), neural damage (27 percent), difficulty in speech (23 percent), feeding problems (10 percent), and seroma or hematoma (6 percent). Associated dental/orthognathic conditions, particularly prognathism and anterior open bite, were documented in one-third of patients.

Conclusions: The initial step in the protocol is control of the neonatal airway. Staged cervical resection is undertaken in late infancy to early childhood; resection should also include abnormal tissue in the oral floor. Sclerotherapy is primarily for macrocystic disease or secondarily for recurrent cysts following partial extirpation. Vesicles of the mucous membranes and dorsal tongue are treated either by sclerotherapy, laser (carbon dioxide, yttrium-aluminum-garnet, or potassium-titanyl-phosphate), or radiofrequency ablation. Reduction for macroglossia is indicated for persistent protrusion or to allow correction of malocclusion. Embolization controls lingual bleeding. Orthognathic procedures are undertaken at the appropriate age, only after lingual size and position are acceptable.

Boston, Mass.

From the Craniofacial Center, Division of Plastic and Oral Surgery, Department of Radiology, and Department of Otolaryngology and Communicative Disorders, Children’s Hospital, Harvard Medical School.

Received for publication November 14, 2003; revised August 4, 2004.

Presented at the 81st Annual Meeting of the American Association of Plastic Surgeons, in Seattle, Washington, April 29, 2002.

John B. Mulliken, M.D., Division of Plastic Surgery, Children’s Hospital, 300 Longwood Avenue, Boston, Mass. 02115, john.mulliken@childrens.harvard.edu

©2005American Society of Plastic Surgeons