Touzot Maxime, Terrier Cécile Saint-Pastou, Faguer Stanislas, Masson Ingrid, François Hélène, Couzi Lionel, Hummel Aurélie, Quellard Nathalie, Touchard Guy, Jourde-Chiche Noémie, Goujon Jean-Michel, Daugas Eric
AURA Paris Plaisance, Paris Service de médecine interne, Hôpital Haut-Levesque, CHU de Bordeaux Département de Néphrologie et Transplantation d'organes, Hôpital Rangueil, CHU de Toulouse Service de néphrologie, Service de néphrologie, CHU Saint-Etienne Service de Médecine interne et immunologie clinique, CHU Bicêtre, Kremlin-Bicêtre Service de néphrologie-transplantation, CHU de Bordeaux, FHU ACRONYM, CNRS-UMR 5164 Immuno Concept Service de néphrologie CHU Necker, Paris Service d'anatomo-pathologie, CHU de Poitiers Aix-Marseille Univ, service de néphrologie, AP-HM, Marseille Service de néphrologie, CHU Bichat, AP-HP, INSERM U1199, Paris Diderot University and DHU FIRE, Paris, France.
Medicine (Baltimore). 2017 Dec;96(48):e9017. doi: 10.1097/MD.0000000000009017.
Severe lupus nephritis in the absence of systemic lupus erythematosus (SLE) is a rare condition with an unclear clinical presentation and outcome.We conducted a historical observational study of 12 adult (age >18 years) patients with biopsy-proven severe lupus nephritis or lupus-like nephritis without SLE immunological markers at diagnosis or during follow-up. Excluded were patients with chronic infections with HIV or hepatitis B or C; patients with a bacterial infectious disease; and patients with pure membranous nephropathy. Electron microscopy was retrospectively performed when the material was available. End points were the proportion of patients with a complete response (urine protein to creatinine ratio <0.5 g/day and a normal or near-normal eGFR), partial response (≥50% reduction in proteinuria to subnephrotic levels and a normal or near-normal eGFR), or nonresponse at 12 months or later after the initiation of the treatment.The study included 12 patients (66% female) with a median age of 36.5 years. At diagnosis, median creatinine and proteinuria levels were 1.21 mg/dL (range 0.5-11.6) and 7.5 g/day (1.4-26.7), respectively. Six patients had nephrotic syndrome and acute kidney injury. Renal biopsy examinations revealed class III or class IV A/C lupus nephritis in all cases. Electron microscopy was performed on samples from 5 patients. The results showed mesangial and subendothelial dense deposits consistent with LN in 4 cases, and a retrospective diagnosis of pseudo-amyloid fibrillary glomerulonephritis was made in 1 patient.Patients received immunosuppressive therapy consisting of induction therapy followed by maintenance therapy, similar to treatment for severe lupus nephritis. Remission was recorded in 10 patients at 12 months after the initiation of treatment. One patient reached end-stage renal disease. After a median follow-up of 24 months, 2 patients relapsed.Lupus nephritis in the absence of overt SLE is a nosological entity requiring careful etiological investigation, including systematic electron microscopy examination of renal biopsies to rule out fibrillary glomerulonephritis. In this series, most patients presented with severe glomerulonephritis, which was highly similar to lupus nephritis at presentation and in terms of response to immunosuppressive therapy.
无系统性红斑狼疮(SLE)的重症狼疮性肾炎是一种罕见疾病,其临床表现和预后尚不清楚。我们对12例成年(年龄>18岁)患者进行了一项历史性观察研究,这些患者经活检证实患有重症狼疮性肾炎或狼疮样肾炎,在诊断时或随访期间无SLE免疫标志物。排除患有HIV或乙型或丙型肝炎慢性感染的患者;患有细菌性传染病的患者;以及患有单纯膜性肾病的患者。当有材料时,回顾性地进行电子显微镜检查。终点是治疗开始后12个月或更晚时完全缓解(尿蛋白与肌酐比值<0.5g/天且估算肾小球滤过率正常或接近正常)、部分缓解(蛋白尿减少≥50%至亚肾病水平且估算肾小球滤过率正常或接近正常)或无反应的患者比例。该研究纳入了12例患者(66%为女性),中位年龄为36.5岁。诊断时,肌酐和蛋白尿水平中位数分别为1.21mg/dL(范围0.5 - 11.6)和7.5g/天(1.4 - 26.7)。6例患者患有肾病综合征和急性肾损伤。所有病例的肾活检检查均显示为III级或IV A/C级狼疮性肾炎。对5例患者的样本进行了电子显微镜检查。结果显示4例患者有与狼疮性肾炎一致的系膜和内皮下致密沉积物,1例患者被回顾性诊断为假性淀粉样纤维性肾小球肾炎。患者接受了免疫抑制治疗,包括诱导治疗后进行维持治疗,类似于重症狼疮性肾炎的治疗。治疗开始后12个月时,10例患者达到缓解。1例患者发展为终末期肾病。中位随访24个月后,2例患者复发。无明显SLE的狼疮性肾炎是一种需要仔细进行病因调查的疾病实体,包括对肾活检进行系统电子显微镜检查以排除纤维性肾小球肾炎。在本系列中,大多数患者表现为重症肾小球肾炎,在临床表现和对免疫抑制治疗的反应方面与狼疮性肾炎高度相似。