Cervical lymphangiomas are rare lymphovascular malformations arising in the neck, which form huge fluid-containing cysts. Treatment of the malformation consists of surgery and sclerotherapy. However, the optimal approach is still controversial. Here, we describe a series of cervical lymphangiomas which have been treated with surgical approaches.
We retrospectively investigated the medical records of 82 patients who had been diagnosed with cervicofacial lymphangioma from 2001 to 2012 in our center. A closed suction drainage with negative pressure was placed on the operative lesion following excision to prevent reaccumulation of lymphatic fluid and the drainage tube was removed after injecting OK-432 through the tube.
Twelve patients underwent surgical excision of cervical lymphangioma. The median patient age was 3 months at the time of the operation. The patients have been followed-up over a period of 34 months. When lesions were located near vital organs such as the trachea or carotid artery or did not respond to repetitive OK-432 injections, surgical treatment might bring good outcomes. However, swallowing difficulty, lip palsy, or dyslalia due to adjacent nerve damage temporarily appeared as postoperative complications. Five children had tracheostomy due to tracheal or subglottic stenosis and 2 patients had gastrostomy due to aspiration while they eat after surgery.
Surgery for cervicofacial lymphangioma should be conducted carefully in selective cases. A well thought-out surgical plan with a multidisciplinary surgical team approach and placement of closed suction drainage tube after surgery and adjuvant OK-432 sclerotherapy through drainage tube seem to be helpful for good outcome.
Pediatric lymphangioma can occur at any site. However the neck is the most common site. There are two treatment modalities (surgical excision and intralesional injection) for lymphangiomas. But, the treatment guide line for lymphangioma has not been established, yet. The aim of this study is to establish the treatment guide line based on our experience with lymphangiomas. Medical records of 82 cases of lymphangioma were reviewed retrospectively. On MRI (magnetic resonance image) findings, lymphangiomas were divided into 4 groups by the proportion of the cyst bigger than 2 cm in diameter of the tumor; group A-proportion of cyst occupies more than 75 %, group B-proportion of the cyst 50~75 %, group C-25~50 %, and D in less than 25 %. All patients were treated with OK-432 intralesional injection as the initial treatment. The effective response rates of OK-432 in group A & B were 88.2 % and 88.8 %, respectively. Group C response was 38.0 % and D only 20.0 %. Twenty-three patients received surgical excision. The result of surgical excision was generally satisfactory. Surgical site infection occurred in 1 case and postoperative bleeding in 1 case. Theses results indicate that intralesional injection of OK-432 could be the first line therapy in group A & B. In group C, OK-432 would be better as the first line therapy than surgery. For the group D, surgical excision should be the first line of treatment.
Lymphangiomas or cystic hygromas are quite commonly seen in children. During a 22-year period, from January 1980 to December 2001, 117 patients with lymphangioma were treated and followed in the Department of Pediatric Surgery at Hanyang University Hospital. The male-to-female ratio was 1.9:1 (77:40) with a male preponderance. As for the age incidence at time of diagnosis, 10 (8.5 %) patients were noted under 1 month of age, 37 (31.6 %) were between 1 month and 1 year of age, 12 (10.3 %) between 1 and 2 years of age, so 59 (50.4 %) were under 2 years of age. Sixty one (52.1 %) lymphangioma-cases were located in the neck, including one case at the nape. Axilla was the second in frequency and the rest were scattered at various sites. The intrascrotal lymphangioma is very rare but we have experienced one case of primary intrascrotal lymphangioma. Eighteen (15.4 %) cases were located in the intraabdominal area, 10 in the mesentery, 2 in the greater omentum and 6 in the retroperitoneum. The chief complaints of intraabdominal lymphangioma were abdominal pain, intestinal obstruction, inguinal hernia, palpable mass, and/or abdominal distension. Among 77 histologically proven cases, 14 cases were cavernous lymphangiomas and the rest were cystic lymphangiomas. Bleeding in the lymphangioma was noted in 20 (17.1 %) cases of all. As for the treatment, a complete excision was performed in 77 (65.8 %) patients and AgNO3 sclerotherapy after incomplete excision was performed in 23 (19.7 %). Picibanil (OK-432) sclerotherapy was performed in 17 (14.5 %) patients. Recurrence rate was 7.7 % and mortality occurred in one case who had a large neck lymphangioma extending into the mediastinum.
Lymphangioma is a congenital malformation of the lymphatic system that expands into the surrounding tissues in a manner similar to a malignant tumor. Surgical excision has been the treatment of choice, but the results are often unsatisfactory due to incomplete resection, fluid collection and recurrences. Intralesional injection of OK-432(lyophilized incubation mixture of Group A
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