Pingyangmycin with triamcinolone acetonide effective for treatment of lymphatic malformations in the oral and maxillofacial region

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Abstract

Objective

The aim of this study was to evaluate the therapeutic effects of intralesion injection of pingyangmycin in combination with triamcinolone acetonide or pingyangmycin alone on lymphatic malformations in oral and maxillofacial regions.

Methods

Twenty-nine cases with lymphatic malformations in the oral and maxillofacial region were divided into experimental and control groups. Thirteen patients in the experimental group underwent intralesion injections of pingyangmycin in combination with triamcinolone acetonide, and 16 patients of control group underwent intralesion injections of pingyangmycin alone. The effects of treatments were assessed by measuring lesion volume and facial deformity before and after treatment.

Results

Two years after the treatment, the volumes of macrocystic and microcystic lesions were 3.7% ± 0.3 and 4.2% ± 0.4 of pre-treatment volume in the experimental group, respectively, whereas the volumes of macrocystic and microcystic lesions in control group were 15.4% ± 1.3 and 24.1% ± 3.1, respectively. Facial appearance was satisfactory in all subjects in the experimental group, whereas facial asymmetry remained in varying degree in control group.

Conclusion

Intralesion injection of pingyangmycin with triamcinolone acetonide was more effective than pingyangmycin alone for treatment of lymphatic malformations in oral and maxillofacial regions.

Introduction

Lymphatic malformations are localized areas of abnormal development of the lymphatic system (Bloom et al., 2004). About 90% of cases in the oral and maxillofacial region occur when patients are under 2 years old and account for more than 75% of all lymphatic malformations (Zhou et al., 2011). Medical history and physical examination are generally sufficient to diagnose lymphatic malformations, however magnetic resonance imaging (MRI) and computed tomography (CT) can help to confirm the diagnosis and to differentiate the lesions from venous anomilies and also to determine the extent of the lesion (Watzinger et al., 1997; Tao et al., 2010). Lymphatic malformations can roughly be divided into the macrocystic, microcystic, or mixed lesions based on response to sclerotherapy (Smith et al., 1996).

Lymphatic malformations in the oral and maxillofacial region often lead to dysfunction and cosmetic disfigurement. Some patients may have trouble in breathing, eating, speaking, and lesions can even become life-threatening because of infections, trauma, hemorrhage, or the compression of the respiratory tract by the rapid enlargement of the pathological tissue (Hartl et al., 2000). There are several treatment options for lymphatic malformations documented in the literature, including laser therapy, sclerotherapy, surgical resection or a combination of these methods (Kobus et al., 1982; Watzinger et al., 1997; Zheng et al., 2005; Grimmer et al., 2006; Kang and Song, 2008; Bajaj et al., 2011). Sclerotherapy is currently recommended as one of the major treatments for the lesions (Zhou et al., 2011); sclerosing agents include bleomycin A5 and pingyangmycin, OK-432, etc. (Smith et al., 1996; Laranne et al., 2002; Alonso et al., 2005; Bai et al., 2009; Sainsbury et al., 2011). All these agents can be effective to some extent for treatment of lymphatic malformations. We have used pingyangmycin effectively to treat lymphatic malformations or hemangiomas in the oral and maxillofacial region for more than 20 years (Luo and Zhao, 2011), but with some problems puzzling us. For example, the lesions become harden after several intralesion injections so that the sclerosing agent can not diffuse smoothly in these areas due to sclerosis of the lesions; the clinical effects remain unsatisfactory in some cases. In addition, the side effects of pingyangmycin may be more harmful to the patients with long-term use. Therefore, the side effects of pingyangmycin need to be overcome during sclerotherapy. Furthermore, facial appearances of some patients remain asymmetric after sclerotherapy and may need plastic surgery to correct the facial asymmetry.

Triamcinolone acetonide is a long acting glucocorticoid, which not only inhibits collagen synthesis but also accelerates collagen degradation, in addition to its ability to repress inflammation (Niessen et al., 1999; Basadonna et al., 1999; Oh et al., 2007). Moreover, glucocorticoids also have inhibitory effects on the development of tumor lymphangiogenesis (Yano et al., 2006) or giant cell granuloma (Shirani et al., 2011; Rachmiel et al., 2012), implying that it may also inhibit the development of diseased lymphatic vessels. Corticosteroids can decrease the risk of bleomycin-induced lung fibrosis by reducing the dosage of bleomycin (Jensen et al., 1990). Whether triamcinolone acetonide could enhance the therapeutic effects of pingyangmycin on lymphatic malformations in oral and maxillofacial regions remained to be explored.

To overcome the problems already mentioned, we explored whether intralesion injections of pingyangmycin in combination with triamcinolone acetonide could have a better therapeutic effects than pingyangmycin alone for treatment of lymphatic malformations in the oral and maxillofacial region.

Section snippets

Patients

From August, 2005 to October, 2009, 29 cases of lymphatic malformations in the oral and maxillofacial region were recruited in the Department of Oral & Maxillofacial Surgery, Peking University Stomatologic Hospital. The ages of the patients ranged from 20 days to 19 years. Diagnosis of lymphatic malformations was made on the basis of patient history, clinical examination, B-ultrasound, MRI, and paracentesis. Patients were randomly divided into experimental and control groups. An experimental

Improvement of facial appearance

Two years after the treatment, facial appearances of the patients in the experimental group were significantly improved and all graded as very satisfactory or satisfactory, better than that of the patients in the control group. There was no case showing any sign of facial nerve injury in either group during and after the treatment (Fig. 1, Fig. 2, Fig. 3).

Regression of lesion

The size of macrocystic and microcystic lesions in the experimental group and control group were both reduced significantly after treatment,

Discussion

In this study, we showed that intralesion injection of pingyangmycin in combination with triamcinolone acetonide or pingyangmycin alone could both effectively treat lymphatic malformations in the oral and maxillofacial region, however, pingyangmycin with triamcinolone acetonide showed better effects in involution of the lesions and improvement of facial appearance than that of pingyangmycin alone.

Enhancement of the therapeutic effects of pingyangmycin by triamcinolone acetonide on lymphatic

Conclusions

Intralesion injection of pingyangmycin in combination with triamcinolone acetonide showed better effects than pingyangmycin alone for treatment of lymphatic malformations in the oral and maxillofacial region.

Conflict of interest

The authors declare no conflicts of interest.

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