Basic and patient-oriented research
Sclerotherapy of Microcystic Lymphatic Malformations in Oral and Facial Regions

https://doi.org/10.1016/j.joms.2008.06.046Get rights and content

Purpose

Lymphatic malformations (LMs) are benign vascular lesions that can cause disfigurement and functional impairment. Complete surgical resection is often difficult and intralesional injection of sclerosing agents has been proposed as an alternative to the surgery. The aim of this study was to review our experience with pingyangmycin (bleomycin A5) injection alone or in combination with surgery for treatment of oral and facial LMs, and to observe the histologic changes after intralesional injection of pingyangmycin.

Patients and Methods

Seventy-nine patients who received intralesional injection of pingyangmycin for the treatment of oral and facial LMs were reviewed. There were 47 males and 32 females, at a ratio of 1 to 0.68. Age ranged from 4 months to 17 years. The sites of the lesions occurred primarily in the tongue, which was found in 37 cases, followed by cheeks in 18, involved lips in 10, parotid gland in 7, oropharynx in 5, and floor of the mouth in only 2 cases. Among them, 42 patients received pingyangmycin sclerotherapy solely, 14 patients had sclerotherapy in combination with secondary surgery, and 23 other patients had surgery with sclerotherapy. Patients had been followed up more than 6 months after the last treatment. The rating of the results was 4-graded: excellent, good, fair, and poor (based on clinical outcome). The histologic changes were observed under microscope in 14 resected specimens after the sclerotherapy.

Results

Primary anatomic locations of 79 LMs involved the face in 25 cases, and the oral cavity in 54. Median number of injections received per child was 4.2 (range, 3 to 8). Among the 79 patients, 44 cases (55.7%) were graded as excellent, 23 cases (29.11%) as good, 10 cases (12.66%) as fair, and 2 cases (2.53%) as poor. There were fewer totally cured or near-normal appearance in the sole sclerotherapy group (42.86%) than in the surgery with the sclerotherapy group (73.91%; P < .05). In specimens resected after pingyangmycin injection, histologic examination showed the destructive lymphatic vessels and obvious stromal fibrosis.

Conclusions

Intralesional injection of pingyangmycin was effective for over two thirds of the children with oral and facial LMs. Perioperative sclerotherapy may improve the treatment of these malformations.

Section snippets

Patients and Methods

Intralesional injection of pingyangmycin was used to treat 103 children with LMs between January 1985 and December 2005. In general, when damage to local structures was a concern or lesions were too extensive for complete excision, pingyangmycin sclerotherapy was recommended as the primary treatment. In addition, some patients were recommended to receive sclerotherapy because of residual lesions after surgery. Seventy-nine patients with follow-up of more than 6 months after the last treatment

Results

Among the 79 patients, 71 were involved with 2 or more anatomic sites and the other 8 developed lesions only involving a single site. The total number of injections performed was 386, and the median number of injections per patient was 4.2 (range, 3 to 8). Median duration of follow-up was 2 years, 8 months (range, 6 months to 79 months).

Overall, in this series, 44 patients (55.70%) were graded as cured (Fig 1) or near-normal appearance, 23 (29.11%) as marked improvement (Fig 2). In the

Discussion

The primary treatment for LMs remains debatable. Many authors agree that complete surgical excision is the first choice of treatment for LMs.18, 19, 20, 21 However, surgical treatment of oral and facial LMs in children has usually been disappointing. Studies have shown that the LMs involving the tongue, floor of the mouth, cheek, pharynx, or multiple anatomic sites have more recurrences, require more procedures, have higher complication rates, and were left with persistent symptomatic disease.22

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