Carrasco Cubero Carmen, Bejarano Moguel Verónica, Fernández Gil M Ángeles, Álvarez Vega Jose Luis
Servicio de Reumatología, Hospital Perpetuo Socorro, Complejo Hospitalario Universitario de Badajoz, Badajoz, España.
Centro de Salud San Fernando, Badajoz, España.
Reumatol Clin. 2016 Jul-Aug;12(4):219-22. doi: 10.1016/j.reuma.2015.08.003. Epub 2015 Oct 29.
Tuberous sclerosis, also called Bourneville Pringle disease, is a phakomatosis with potential dermal, nerve, kidney and lung damage. It is characterized by the development of benign proliferations in many organs, which result in different clinical manifestations. It is associated with the mutation of two genes: TSC1 (hamartin) and TSC2 (tuberin), with the change in the functionality of the complex target of rapamycin (mTOR). MTOR activation signal has been recently described in systemic lupus erythematosus (SLE) and its inhibition could be beneficial in patients with lupus nephritis. We report the case of a patient who began with clinical manifestations of tuberous sclerosis complex (TSC) 30 years after the onset of SLE with severe renal disease (tipe IV nephritis) who improved after treatment with iv pulses of cyclophosphamide. We found only two similar cases in the literature, and hence considered the coexistence of these two entities of great interest.
结节性硬化症,也称为博恩维尔-普林格尔病,是一种可导致皮肤、神经、肾脏和肺部潜在损害的错构瘤病。其特征是在许多器官中出现良性增生,从而导致不同的临床表现。它与两个基因的突变有关:TSC1(错构瘤蛋白)和TSC2(结节蛋白),这会导致雷帕霉素复合物靶标(mTOR)功能的改变。最近在系统性红斑狼疮(SLE)中发现了mTOR激活信号,对其进行抑制可能对狼疮性肾炎患者有益。我们报告了一例患者,该患者在SLE发病30年后出现了结节性硬化症复合体(TSC)的临床表现,伴有严重的肾脏疾病(IV型肾炎),经静脉注射环磷酰胺脉冲治疗后病情有所改善。我们在文献中仅发现两例类似病例,因此认为这两种疾病的共存非常值得关注。