Tiddens H A, van der Net J J, de Graeff-Meeder E R, Fiselier T J, de Rooij D J, van Luijk W H, Herzberger R, van Suijlekom L W, van Venrooij W J, Zegers B J
Department of Immunology, University Hospital for Children and Youth Het Wilhelmina Kinderziekenhuis, Utrecht, The Netherlands.
J Pediatr. 1993 Feb;122(2):191-7. doi: 10.1016/s0022-3476(06)80112-5.
To establish the symptoms and clinical course of juvenile-onset mixed connective tissue disease, we studied 14 patients, classified according the criteria of Kasukawa et al. The patient records were studied retrospectively and all patients were examined in a 1-day follow-up program. Systemic lupus erythematosus and polymyositis/dermatomyositis-like symptoms disappeared in time, whereas scleroderma-like symptoms (such as in the Raynaud phenomenon) and joint abnormalities persisted. Extensive limitation of joint function was found in four patients. At the time of follow-up, no active renal disease was found. Thrombocytopenia was still present in one of the three patients who had had this feature. All patients had abnormalities of esophageal motility. Long-term corticosteroid treatment was associated with aseptic bone necrosis in three patients and growth retardation in one. We conclude that the Kasukawa criteria are easy to apply to children, and that juvenile-onset mixed connective tissue disease has many similarities to the adult form of the disease.
为明确青少年起病的混合性结缔组织病的症状及临床病程,我们依据春日川等人的标准对14例患者进行了分类研究。对患者记录进行了回顾性分析,并在为期1天的随访项目中对所有患者进行了检查。系统性红斑狼疮及多发性肌炎/皮肌炎样症状及时消失,而硬皮病样症状(如雷诺现象)及关节异常持续存在。4例患者存在关节功能的广泛受限。随访时未发现活动性肾脏疾病。有血小板减少症病史的3例患者中,1例仍存在血小板减少。所有患者均有食管动力异常。长期使用糖皮质激素治疗使3例患者出现无菌性骨坏死,1例患者出现生长发育迟缓。我们得出结论,春日川标准易于应用于儿童,且青少年起病的混合性结缔组织病与成人型该疾病有许多相似之处。