Dermatology Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy -
G Ital Dermatol Venereol. 2013 Oct;148(5):525-30.
Infantile hepatic hemangioma (IHH) is a common liver tumors of infancy with a higher incidence in females. Various treatments for infantile hepatic hemangioma such as systemic corticosteroids, interferon-alpha, vincristine and cyclophosphamide have been suggested, though no consensus exists about the first-choice treatment. Recent evidences suggest that propranolol, a nonselective β-blocker, may be effective and safe as first-line therapy for infantile hepatic hemangioma. We report a case of female born at term with a weight of 2.450 g started to develop multiple cutaneous IHs at 10 days of age and presenting concomitant multiple cutaneous and hepatic infantile hemangiomas confirmed on magnetic resonance imaging. Propranolol, used as monotherapy, was started at 14 days of age at a dose of 2 mg/kg/day orally and maintained for 6 months. Patient was monitored in the hospital during first days of treatment with propranolol, and discharged after no side-effects were detected. Hepatic and cutaneous lesions had complete resolution in three months, although the fibro-fatty residuum of largest cutaneous nodule was still palpable at month 6. A further control after 6 months showed no recurrences. Our report case suggests that propranolol can be a safe and effective first-line therapy for neonates with concomitant multiple cutaneous and hepatic infantile hemangiomas.
婴儿型肝脏血管瘤(IHH)是一种常见的婴儿期肝脏肿瘤,女性发病率较高。已提出多种治疗婴儿型肝脏血管瘤的方法,如全身皮质类固醇、干扰素-α、长春新碱和环磷酰胺等,但对于首选治疗方法尚无共识。最近的证据表明,非选择性β受体阻滞剂普萘洛尔可能是治疗婴儿型肝脏血管瘤的有效和安全的一线治疗药物。我们报告了一例女性足月出生,体重 2450 克,出生后 10 天开始出现多发性皮肤 IHH,并通过磁共振成像证实同时存在多发性皮肤和肝脏婴儿型血管瘤。普萘洛尔于 14 天龄时开始作为单药治疗,剂量为 2mg/kg/天口服,维持 6 个月。在开始使用普萘洛尔的最初几天,患者在医院接受监测,在未发现副作用后出院。肝脏和皮肤病变在 3 个月内完全消退,尽管最大皮肤结节的纤维脂肪残余仍可触及 6 个月。6 个月后的进一步随访显示无复发。我们的报告病例表明,普萘洛尔可作为同时存在多发性皮肤和肝脏婴儿型血管瘤的新生儿的安全有效一线治疗药物。