Kumar L, Thapa B R, Sarkar B, Walia B N
Department of Pediatrics, Postgraduate Institute of Medical Education and Research, Chandigarh, India.
Ann Rheum Dis. 1995 Feb;54(2):134-6. doi: 10.1136/ard.54.2.134.
To report 10 children younger than 10 years of age with benign cutaneous polyarteritis nodosa (BCPAN).
Ten children aged 1.25-10 years (mean 4.7 years; M:F = 7:3) were admitted with an unusual vasculitis. The clinical features, laboratory investigations, treatment and follow up data were analysed.
Clinical features of these patients included: fever (10), peripheral gangrene (eight), livido reticularis (four), ulceration, nodules and vesiculobullous lesions alone or in combination (10), black necrotic patches over limbs and trunk (three), and arthralgia or swelling of large joints (seven). Cryoglobulinaemia was transient in three children, lasting for eight months in one of them. Histopathology of the skin lesions revealed vasculitis of small and occasionally medium sized blood vessels in nine of the 10 children. Possible association of BCPAN was noted with diphtheria-pertussistetanus immunisation (one), drugs (two), streptococcal infection (two), wasp sting (one), and falciparum malaria (one). The clinical course was interspersed with remissions and exacerbations. Response to corticosteroids alone occurred in seven patients, while three children needed cytotoxic drugs in addition. In a follow up of 5.6 years (mean) no evidence of systemic involvement was noted.
A rare form of vasculitis, BCPAN, is reported in children. The features that distinguished our patients from those reported earlier were onset in the first decade of life and higher incidence of peripheral gangrene.
报告10例10岁以下的儿童良性皮肤结节性多动脉炎(BCPAN)。
10例年龄为1.25至10岁(平均4.7岁;男∶女 = 7∶3)的儿童因一种不寻常的血管炎入院。分析其临床特征、实验室检查、治疗及随访数据。
这些患者的临床特征包括:发热(10例)、外周坏疽(8例)、网状青斑(4例)、单独或合并出现的溃疡、结节及水疱大疱性皮损(10例)、四肢和躯干的黑色坏死斑(3例)以及大关节的关节痛或肿胀(7例)。3例儿童出现短暂性冷球蛋白血症,其中1例持续8个月。10例儿童中有9例皮肤病变的组织病理学显示小血管以及偶尔的中等大小血管的血管炎。注意到BCPAN可能与白喉 - 百日咳 - 破伤风免疫接种(1例)、药物(2例)、链球菌感染(2例)、黄蜂蜇伤(1例)和恶性疟(1例)有关。临床病程呈缓解与加重交替。7例患者单独使用皮质类固醇有反应,而3例儿童还需要细胞毒性药物。在平均5.6年的随访中未发现有全身受累的证据。
报告了儿童中一种罕见的血管炎形式,即BCPAN。我们的患者与早期报告的患者不同的特征为发病于生命的第一个十年以及外周坏疽的发生率较高。