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 A(5)) 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.
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 A(5)) 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.
Authors: June K Wu; Ellen D Hooper; Sherelle L Laifer-Narin; Lynn L Simpson; Jessica Kandel; Carrie J Shawber Journal: Pediatrics Date: 2016-08-25 Impact factor: 7.124