Kahn Cameron, Singh Sukhraj, Mathew Reshmi, Ramrattan Laurie A, Mohammed Ibraheem J, Omman Reeba
Medicine, University of Florida College of Medicine - Jacksonville, Jacksonville, USA.
Rheumatology, University of Florida College of Medicine - Jacksonville, Jacksonville, USA.
Cureus. 2022 Jul 2;14(7):e26516. doi: 10.7759/cureus.26516. eCollection 2022 Jul.
Nonuremic calciphylaxis (NUC) is a rare and debilitating form of panniculitis. NUC is associated with a high mortality rate within the first year of diagnosis. Connective tissue diseases account for a small fraction of the reported cases. However, there have also been reported cases of patients developing NUC while on treatment with chronic corticosteroid immunosuppressive therapy. The pathophysiology of NUC is still not fully established. Several risk factors including underlying diseases, obesity, female gender, and medications have been associated with the development of NUC. The diagnosis remains challenging due to the condition's similarities with other forms of panniculitis. The gold standard for diagnosis is a tissue biopsy showing calcifications within the medial layer of arterioles and the presence of microthrombi with surrounding necrosis. The treatment for NUC has not advanced much in recent years and focuses on the management of the underlying condition, wound care, and treating any superimposed infection. Treating superimposed infections remains important as most of the associated mortality from NUC occurs due to sepsis. We describe a case of a young woman with lupus nephritis who developed NUC while on prolonged corticosteroid therapy. She did not respond to several immunosuppressive agents and was ultimately treated with rituximab, a monoclonal antibody against CD20 antigen, as salvage therapy.
非尿毒症性钙化防御(NUC)是一种罕见且使人衰弱的脂膜炎形式。NUC在诊断后的第一年内死亡率很高。结缔组织病在报告的病例中占一小部分。然而,也有报道称患者在接受慢性糖皮质激素免疫抑制治疗时发生NUC。NUC的病理生理学仍未完全明确。包括基础疾病、肥胖、女性性别和药物在内的多种危险因素与NUC的发生有关。由于该病症与其他形式的脂膜炎相似,诊断仍然具有挑战性。诊断的金标准是组织活检显示小动脉中层有钙化以及存在微血栓并伴有周围坏死。近年来,NUC的治疗进展不大,主要集中在基础疾病的管理、伤口护理以及治疗任何叠加感染。治疗叠加感染仍然很重要,因为NUC相关的死亡大多是由败血症引起的。我们描述了一例患有狼疮性肾炎的年轻女性,她在长期接受糖皮质激素治疗时发生了NUC。她对多种免疫抑制剂均无反应,最终接受了利妥昔单抗(一种抗CD20抗原的单克隆抗体)作为挽救治疗。