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J Oral Maxillofac Surg 55:306-309, 1997

Surgical Excision of Lymphangioma tous Macroglossia: A Case Report

XIN-CHUN JIAN, DDS, MD*

Lymphangioma of the tongue may present as a local- ized or a diffuse growth. Localized lymphangioma oc- curs as a nodular tumor elevated above the surface of the tongue. Its limited extent usually permits complete

* Associate Professor and Head of Oral and Maxillofacial Surgery, Xiang Ya Hospital Hunan Medical University, ChangSha, Hunan, People’s Republic of China.

Address correspondence and reprint requests to Dr Ban: Xiang Ya Hospital Hunan Medical University, ChangSha, Hunan 410008, People’s Republic of China.

surgical excision. The diffuse lymphangioma involves a major portion of the tongue and offers a more difficult management problem.

We describe a patient who had resection of a lymph- angiomatous macroglossia in whom the tongue was reconstructed so that it assumed normal size and shape and the tip consisted of normal tissue.

Report of Case

0 1997 American Association of Oral and Maxillofacial Surgeons

0278-2391/97/5503-0016$3.00/O A lo-year-old girl was admitted on December 13, 1987

with inability to retract her protruded tongue since infancy.

XIN-CHUN JIAN 307

FIGURE 1. Appearance of patient before (AJ) and 3 years after (C,D) resection of the lesion

308 EXCISION OF LYMPHANGIOMATOUS MACROGLOSSIA

FIGURE 2. Diagram of tongue resection. A, Design of incision of sublingual incision. D, The condition after suturing.

on the lingual dorsal surface. B, Incision of lingual lateral margin. 1esign

XIN-CHUN JIAN 309

FIGURE 3. A, Superior and B, inferior views of the operative specimen

It had increased in size with growth of the child, and at the time of admission there was a marked protrusion of the tongue (Fig lA), completely filling the interarch space. The exposed part was red because of recurrent inflammation and was tense and dry. On the dorsal surface, translucent lymph- angiomatous vesicles produced an irregular, granular appear- ance (Fig IB). The mouth could not be closed, and saliva drooled from the corners of the mouth. Speech was thick, guttured, and difficult to comprehend. Pressure from the size and weight of the tongue had produced mandibular defor- mity, which was characterized by an anterior open bite, elon- gation of the mandibular body, increase of the body-ramus angle, and anterior displacement and outward tipping of the incisal edges of the anterior teeth.

On December 21, 1987, the patient underwent surgical resection under general nosotracheal anesthesia. For retrac- tion, two deep sutures were placed in lateral borders of the tongue. A figure-of-eight ligature is placed around the base of the tongue as a temporary method to control bleeding during surgery. Three V-shaped incisions are outlined on the lingual dorsal, lingual lateral, and lingual inferior surfaces of the tongue with methylene blue (Fig 2A-C). The angle of the V on the tongue dorsum was about 1 cm in front of the circumvallate papillae and The inverted V enclosed within it the entire tumor (Fig 3). After completion of the incisions and removal of the lesion the remaining parts of the tongue were approximated in three layers. The reconstruction pro- vided a new tip of the tongue that consisted of normal tissue, and the tongue assumed a relatively normal size and shape (Fig 2D).

The patient was discharged after 2 weeks with a normal- looking tongue and marked improvement in speech. For her mandibular deformity (Fig lC), she was advised to undergo orthodontic treatment. The biopsy report indi- cated lymphangioma. On follow-up 3 years after surgery, there was an improved facial appearance and good tongue function.

Discussion

There have been several different surgical tech- niques described to treat lymphangiomatous macro- glossia.‘-5 V-shaped wedge resection in which the en- tire thickness of the tip of the tongue is removed back to the level of the circumvallate papillae; bilateral mar- ginal resection; a combination of wedge resection and marginal resection; and U-shaped resection with the open end of the incision facing posteriorly. Regardless of the method used, the recurrence rate is high and there is often a need for further surgical procedures. For this reason, we believe that, if possible, lymphangi- omatous lesions should be completely resected.

The method used in this case, which is best suited for lymphangiomatous lesions in the anterior-middle portion of the tongue has the following advantages: 1) After healing, the shape resembles a normal tongue and the suture line resembles the median furrow; 2) The movements of the tongue are not impaired and so articulation is facilitated; and 3) The inverted V-shaped incision leaves normal tissue on the lateral margins of the tongue, which contains taste buds.

References

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2. Robinson F: Lymphangioma of tongue. Br J Plast Surg 6:48, 1953

3. Hendrich JW, Antonio S: Macroglossia or giant tongue. Surgery 39~614, 1956

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5. Dinerman WS, Myers EN: Lymphangiomatous macroglossia. Laryngoscope 86:291, 1976