Abstract
Picibanil (OK-432) and bleomycin have been used as alternative sclerosing agents for lymphatic malformations. This study evaluated the clinical curative effect of sclerotherapy using fibrin glue combined with OK-432 and bleomycin for the treatment of macrocystic lymphatic malformations of the face and neck. Fifteen paediatric patients (6 males; 9 females, aged 13 months to 14 years) who had received percutaneous sclerotherapy for massive macrocystic lymphatic malformations of the face and neck were retrospectively reviewed. Affected regions included the neck, parotid region and parapharynx, mouth floor, face and cheek, and orbital regions. All patients showed preoperative symptoms of space-occupying lesions between 4 cm × 5 cm and 12 cm × 16 cm in size. Fibrin glue with OK-432 and bleomycin was injected under general anaesthesia. All patients received preoperative and follow-up CT scans. Outcomes were assessed by three surgeons. All patients exhibited mid-facial swelling for 3–4 weeks after surgery, but no major complications. Follow-up periods ranged from 8 to 16 months. Eight lesions were completely involuted, five were mostly involuted, and two were partially involuted. Percutaneous sclerotherapy using fibrin glue with OK-432 and bleomycin provided a simple, safe, and reliable alternative treatment for massive macrocystic lymphatic malformations of the face and neck.
Lymphatic malformations are benign vascular lesions that can cause disfigurement and functional impairment. The most common preoperative symptoms are intralesional haemorrhage and infection; more serious symptoms such as dysphasia and airway obstruction may also occur . Surgical excision has been the standard treatment for such malformations , but it has been associated in some cases with postoperative recurrence and surgical complications, such as facial nerve paralysis, bleeding or haematoma, Frey’s syndrome, Stant’s duct obstruction, and contour defects . A variety of nonsurgical alternatives have been investigated, with the goal of reducing morbidity whilst effectively treating the lesion. Recent attention has focussed on the sclerosants picibanil (OK-432) and bleomycin . Some studies have suggested that the intralesional injection of fibrin glue is an effective treatment modality for orbital cystic lymphangiomas . The authors have previously reported the use of fibrin glue combined with OK-432 and bleomycin to perform percutaneous sclerotherapy on massive venous malformations of the face and neck and on juvenile nasopharyngeal angiofibromas . In the present report, the authors discuss their experience with percutaneous sclerotherapy using fibrin glue with OK-432 and bleomycin to treat macrocystic lymphatic malformations of the face and neck.
Patients and methods
A retrospective review of 15 patients diagnosed with macrocystic lymphatic malformations of the face and neck was carried out. These patients received percutaneous sclerotherapy using fibrin glue with OK-432 and bleomycin between February 2008 and August 2009. The Institutional Review Board of Sun Yat-Sen Hospital provided ethical approval for the study. The study sample comprised 6 males and 9 females, aged 13 months to 14 years (mean 6.1 years). Affected regions included the neck ( n = 6), parotid region and parapharynx ( n = 4), mouth floor ( n = 2), face and cheek ( n = 2), and orbital regions ( n = 1). All of the patients showed preoperative symptoms of space-occupying lesions ( Fig. 1 A and B ) and underwent a computer tomography (CT) angiography of the lesion ( Fig. 1 C). The lesions ranged in size from 4 cm × 5 cm to 12 cm × 16 cm (mean 7.7 cm × 9.5 cm).
The patients included in the study sample had received no previous treatment (e.g. surgery, alcohol sclerotherapy, or systemic corticosteroids) and exhibited no symptoms (e.g. haemorrhage, infection, dysphasia, or airway obstruction). None of the patients reported a penicillin allergy before treatment. Preoperative immunological testing yielded negative results for all patients.
Under general anaesthesia, the patients were given an injection of fibrin glue (Guangzhou Bioseal Co., Ltd., Guangzhou, China) combined with OK-432 (streptococcal pyrogenic exotoxin A; Shandong Lukuang Pharmaceutical Group Luya Co., Ltd., Jinan, China) and bleomycin (Nipponkayaku Co., Ltd., Tokyo, Japan). The fibrin glue components were packaged in separate bottles that contained freeze-dried fibrinogen powder (50–75 mg); blood coagulation factor XIII (10–70 U); potassium dihydrogen phosphate (6.8 mg) buffer for the main gel; freeze-dried thrombin powder (400 IU) for the catalyst; or calcium chloride (40 mmol) solution for the catalyst. Two syringes were provided with the fibrin glue; the fibrinogen, blood coagulation factor XIII, and potassium dihydrogen phosphate buffer were loaded into syringe A, and the thrombin and calcium chloride solution were loaded into syringe B. The contents of syringes A and B were mixed to produce 20 ml of fibrin glue.
The OK-432 solution was prepared by dissolving lyophilized OK-432 at a concentration of 1 mg (1 klinische Einheit (KE), clinical unit per 1 ml of normal saline (NS)). The bleomycin solution was prepared by dissolving lyophilized bleomycin at a concentration of 15 mg per 2 ml of NS. The OK-432 solution was added to syringe A, and the bleomycin solution was added to syringe B, according to each patient’s dosage requirements. The lesions were punctured with 18-gauge needles and completely aspirated, after which the needles were left in place. The two syringes containing the reagents were fitted onto the needles, and the fibrin glue containing OK-432 and bleomycin was injected into the lesions from several directions at two to three sites ( Fig. 2 ). Intravenous dexamethasone (0.1 mg/kg) and NS (60 ml/kg) were given for 3 days postoperatively.
All the patients received these injections during a single sclerotherapy procedure. The mean total doses of fibrin glue, OK-432, and bleomycin were 18.3 ml (range 15–30 ml), 3.6 mg (range 2–6 mg), and 11.2 mg (range 5–20 mg), respectively. The volume of injected fibrin glue equalled approximately one-fourth to one-third of the cavity volume for each lesion. Treatment success was defined as clinically reduced lesion size, which was determined by serially photographing and measuring the lesions.
All the patients received CT scans preoperatively and at follow-up, 8–16 months after surgery. A panel of three surgeons assessed the outcomes according to the following categories: complete involution, >90% reduction in size ( Fig. 3 ); mostly involuted, 75–90% reduction; partial involution, 50–75% reduction; small involution, 25–50% reduction; and non-involution, <25% reduction .
Results
All the patients exhibited mid-facial swelling for 3–4 weeks after surgery, but no major complications occurred ( Table 1 ). The follow-up period ranged from 8 to 16 months (mean 11.5 months). Eight lesions (53%) were completely involuted; five (33%), mostly involuted; and two (13%), partially involuted ( Table 1 ). None of the patients underwent subsequent excision. All of the patients showed normal liver and kidney functions, and no haematologic toxic effects or signs of pulmonary involvement were observed.
| Case no. | Age (y)/gender | Lesion site | Lesion size (cm) | FG (ml), OK (mg), BL (mg) | Complications | Follow-up (months) | Outcome |
|---|---|---|---|---|---|---|---|
| 1 | 9/F | Neck | 8 × 9 | 20, 4, 15 | – | 16 | CI |
| 2 | 4/F | Neck | 6 × 8 | 15, 3, 10 | – | 13 | MI |
| 3 | 12/F | Parotid region and parapharynx | 12 × 16 | 30, 6, 20 | – | 14 | PI |
| 4 | 13/M | Parotid region and parapharynx | 8 × 10 | 20, 4, 15 | – | 14 | CI |
| 5 | 6/M | Face and cheek | 6 × 8 | 15, 3, 10 | – | 14 | CI |
| 6 | 2/F | Orbital regions | 4 × 5 | 10, 2, 5 | – | 13 | MI |
| 7 | 2/F | Neck and throat | 8 × 10 | 20, 4, 10 | – | 12 | CI |
| 8 | 1.17/F | Mouth floor | 6 × 8 | 15, 2, 5 | – | 12 | PI |
| 9 | 1.5/M | Parotid region | 8 × 8 | 15, 3, 7.5 | – | 12 | MI |
| 10 | 1.25/F | Neck | 8 × 10 | 20, 4, 10 | 10 | CI | |
| 11 | 14/F | Face and cheek | 12 × 14 | 25, 5, 15 | HP | 9 | MI |
| 12 | 6/M | Parotid region | 6 × 8 | 15, 3, 10 | 9 | CI | |
| 13 | 2/M | Neck | 6 × 6 | 15, 3, 10 | 8 | CI | |
| 14 | 10/F | Mouth floor | 10 × 12 | 20, 4, 15 | 8 | MI | |
| 15 | 8/M | Neck | 8 × 10 | 20, 4, 10 | 8 | CI |
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