Abstract
The management of infantile hemangiomas remains a subject of controversy. The purpose of this study was to investigate the indications and treatment results of intralesional injection of pingyangmycin for treatment of infantile hemangiomas. In a prospective study of 66 patients, the effectiveness of intralesional injection of pingyangmycin was evaluated and documented. The lesions were all located in the oral and maxillofacial regions. The smallest lesion was 1.0 cm × 0.7 cm and the largest was 4.6 cm × 3.8 cm. Amongst the 66 infants who underwent sclerotherapy with pingyangmycin, cure was obtained in 74% (49/66) of patients, marked improvement of the treated lesion occurred in 14% (9/66), improvement occurred in 12% (8/66), and a lack of response was not observed in any patient. All patients were regularly followed up for 1–4 years following pingyangmycin treatment, and they demonstrated the same good results over this time. Intralesional injection of pingyangmycin was a reliable and effective therapeutic choice for infantile oral and maxillofacial hemangiomas, as it shortened the involution time and decreased the influence induced by these potentially countenance-influenced tumours with few complications.
Hemangiomas are amongst the most common forms of congenital and neonatal dysmorphogenesis, and they demonstrate a characteristic pattern of rapid postnatal growth followed by slow involution . They can occur in various areas of the body, with 60% being located in the head and neck region. According to their clinical behaviour, the complete process of hemangioma involves proliferating, involuting and involuted phases. Multiple authors reporting on the natural history of hemangiomas have indicated that the rate of involution is relatively consistent. Approximately 50% of hemangiomas are completely resolved by 5 years of age, and about 70% by 7 years of age. Subsequent improvement may occur in the remaining lesions from 10 to 12 years . Their entire natural history may be 10 years or more.
Previous studies have examined the influence of multiple factors on the involution of hemangiomas, and found that lesions of the lip and nose tend to resolve more slowly than others. The tremendous psychosocial consequences of hemangiomas on the affected child and their family should receive more attention. Effective therapy methods should be considered to shorten the involution time of infantile hemangiomas in the oral and maxillofacial region that cannot be hidden from view by clothing.
The last half-century has witnessed major improvements in the treatment of hemangiomas. Many modalities have been used, including operative therapy, corticosteroids, interferon-α-2a/2b, laser therapy, sclerotherapy, cryosurgery, embolization, and radiation therapy . Each of these approaches has advantages and disadvantages based on clinical results. Sclerotherapy is regarded as a suitable choice for some superficial lesions . Finding a suitable sclerosing agent has received considerable attention.
Pingyangmycin is derived from soil that is rich in fungi found in Pingyang County, South-East Zhejiang Province, China. It has a similar chemical structure to bleomycin A5, but the terminal amine moiety is different . The side effects related to use of pingyangmycin are rare and dose related. The therapeutic effect of this compound is derived from its endothelial toxicity, which is achieved at high target-tissue concentrations.
Between January 2003 and December 2008, 66 infants with hemangiomas in the oral and maxillofacial region were treated with intralesional injection of pingyangmycin in the authors’ department with acceptable results. The authors evaluated the experience and investigated the indications, drug concentration and dosage, method of injection, safety, efficacy and prevention of complications.
Materials and methods
From January 2003 to December 2008, 66 consecutive infantile patients with hemangiomas were referred for intralesional pingyangmycin injection. A standardized data collection sheet recorded patient details, including age, sex, weight, location and size of the lesion, clinical history, special investigations, pingyangmycin dose, clinical response, side effects, and follow-up. Colour photographs were taken of every patient before, during, and after completion of the treatment to help the parents see the regression of the lesion over time. 31 patients were male and 35 female (male to female ratio 1:1.13). Their age ranged from 2 to 16 months with a median age of 5 months and a mean age of 5.6 months. 46 patients were younger than 6 months, 17 were between 6 and 12 months, and 3 were between 12 and 16 months. The smallest lesion was 1.0 cm × 0.7 cm and the largest was 4.6 cm × 3.8 cm. The lesions involved different sites: 9 were located on the forehead, 5 on the nose, 7 on the cheek, 16 on the parotid or masseteric area, and 29 on the lip. None of these hemangiomas could be hidden from view by clothing.
60 infants received intralesional pingyangmycin injection alone. Of the remaining patients, 4 underwent Nd:YAG laser therapy and 2 received surgical excision in addition to pingyangmycin injection.
Prior to injection, all the patients underwent hemography, chest radiography, renal function tests, and other routine clinical examinations to exclude systemic diseases and an allergic constitution. Informed consent was acquired from the parents before the commencement of sclerotherapy. This clinical study was approved by the Human Ethics Review Committee of the hospital.
Pingyangmycin (8 mg/ampoule; Tianjin Tai-He Pharmaceutical, Tianjin, China) was dissolved in 4 ml of normal saline solution and 4 ml of 2% lidocaine hydrochloride. The final concentration of pingyangmycin was 1 mg/ml.
The injection was performed through a 23-gauge needle after sterilization of the injection area with povidone-iodine. The procedure was performed under dissociation anaesthesia to ensure the cooperation of the patients. Injection was carried out until the surface of the lesions became a little pale. Multiple injections were needed at different sites and points for larger or more extensive lesions. The maximum dose for one injection was 2 mg, and the total dose for one infant patient did not exceed 40 mg. This is in accordance with published dosage regimens . To avoid local swelling and ulceration in the lips, the dosage for one injection was shorten in some instances. After injection, the lesion was compressed for 5 min to stop bleeding and prevent egress of the pingyangmycin sclerosant. This procedure required hospital admission for 1 day for observation. The patients were followed up weekly, and the procedure was repeated after 2 weeks. Every session included no more than three intralesional pingyangmycin injections. If an additional treatment session was required, the appropriate interval was 1 month. The number of sessions ranged from 1 to 6 with a mean of 1.8 sessions.
All patients were evaluated before injection, between treatment intervals, and at 1 month after the end of treatment. The size of the lesions and changes during treatment were measured and recorded. All patients were followed for 1–4 years. The treatment results were scored according to a four-point scale modified after Z heng et al. and M uir et al. based on improvement in volume, colour, and texture after treatment where: 1 is no response, no change in the size or continued enlargement; 2 is improvement, the lesion decreased in size, but demonstrated less than a 50% improvement in appearance; 3 is marked improvement, the lesion decreased in size by more than 51% but less than 100%, with remarkable improvement in appearance; and 4 is cure, the lesion disappeared completely without recurrence at least 1 year after treatment.
Results
Amongst the 66 infants who received sclerotherapy with pingyangmycin, cure was obtained in 74% (49/66), marked improvement of the lesion occurred in 14% (9/66) of patients, and improvement was observed in 12% (8/66). An overall significant effect was seen in 58 patients (88%). A lack of response was not observed in any patient. All patients were regularly followed up at 6-month intervals over 1–4 years and they all demonstrated the same good results over this period of time.
An excellent response was obtained in 28 patients with small hemangiomas (their largest original lesion dimension was less than 2.0 cm). Their tumour mass gradually shrank, most markedly during the second to sixth weeks ( Fig. 1 ), and an obvious decrease in lesion size was measured after one session of intralesional pingyangmycin injections. The dark-red skin and oral mucosa on the hemangioma body gradually, but clearly, lost its colouration. Subsequently, the dilated lesions disappeared.
The other 38 children underwent regular follow up, and were given repeated intratumoral injections of pingyangmycin due to insufficient shrinkage of the hemangioma body. It was found that the number of intralesional pingyangmycin injections required showed a tendency to relate positively to the size of the hemangiomas ( Fig. 2 ). The larger the lesion, the more injection was required to obtain a satisfactory result. The two biggest lesions (4.6 cm × 3.8 cm and 3.7 cm × 3.0 cm) required six and five sessions, respectively ( Fig. 3 ).
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