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[特发性系统性毛细血管渗漏综合征(克拉克森病):复发性危及生命水肿的罕见病因]

[Idiopathic systemic capillary leak syndrome (Clarkson disease) : A rare cause of recurrent life-threatening edema].

作者信息

Dasdelen S, Grebe S-O

机构信息

Vivantes Humboldt Klinikum Berlin, Am Nordgraben 2, 13509, Berlin Reinickendorf, Deutschland.

Helios Universitätsklinikum Wuppertal, Wuppertal, Deutschland.

出版信息

Internist (Berl). 2018 Jul;59(7):725-735. doi: 10.1007/s00108-018-0409-2.

Abstract

This case report describes the episodic occurrence of severe generalized edema in a young female patient, who developed hypertension with a massive hemoconcentration (hematocrit >0.5, hemoglobin >20g/dl) and hypoalbuminemia during the course of these acute disease phases. After the first two disease exacerbations were overcome, there was a complete regression of symptoms. After a long symptom-free interval, a new exacerbation occurred as a result of which critical organ ischemia occurred due to the severe hypotension and massive edema. Despite all treatment measures a severe compartment syndrome of the lower extemities with subsequent rhabdomyelosis developed. The patient ultimately died as a result of treatment-refractory cardiovascular failure. The idiopathic systemic capillary leak syndrome (SCLS, also known as Clarkson disease) is a rare and potentially life-threatening disease with a high mortality. Since the first description of the disease only approximately 500 cases have been published worldwide. The pathophysiology of this disease remains unclear despite all previous attempts at clarification. Regulation processes of endothelial permeability seem to be essentially disturbed. Affected patients have a monoclonal gammopathy of undetermined signficance conspicuously often; however, the knowledge of the limited treatment options is of fundamental importance for the prognosis and overall survival of patients.

摘要

本病例报告描述了一名年轻女性患者间歇性出现严重全身性水肿的情况,该患者在这些急性疾病阶段出现高血压,并伴有大量血液浓缩(血细胞比容>0.5,血红蛋白>20g/dl)和低白蛋白血症。在前两次疾病发作得到控制后,症状完全消退。经过较长时间的无症状期后,出现了新的发作,由于严重低血压和大量水肿导致关键器官缺血。尽管采取了所有治疗措施,仍发生了下肢严重骨筋膜室综合征并继发横纹肌溶解症。患者最终因治疗无效的心血管衰竭而死亡。特发性系统性毛细血管渗漏综合征(SCLS,也称为克拉克森病)是一种罕见且可能危及生命的疾病,死亡率很高。自该疾病首次被描述以来,全球仅发表了约500例病例。尽管此前进行了所有澄清尝试,但该疾病的病理生理学仍不清楚。内皮通透性的调节过程似乎受到了根本性的干扰。受影响的患者常常明显存在意义未明的单克隆丙种球蛋白病;然而,了解有限的治疗选择对患者的预后和总体生存至关重要。

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