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常染色体显性营养不良型大疱性表皮松解症成年男性合并局灶性硬化和肾淀粉样变性的弥漫性膜性增生性肾小球肾炎:一例报告。

Diffuse membranoproliferative glomerulonephritis with focal sclerosis and renal amyloidosis in an adult male with autosomal dominant dystrophic epidermolysis bullosa: a case report.

机构信息

Department of Medicine, Division of Nephrology, Medical University of South Carolina , Charleston , SC , USA.

Department of Medicine, Division of Nephrology, Cairo University , Cairo , Egypt.

出版信息

Ren Fail. 2019 Nov;41(1):850-854. doi: 10.1080/0886022X.2019.1614056.

DOI:10.1080/0886022X.2019.1614056
PMID:31498016
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6746271/
Abstract

Previous reports of glomerular disease in adult patients with autosomal dominant dystrophic epidermolysis bullosa (EB) are limited and include post-infectious glomerulonephritis, IgA nephropathy, amyloidosis, and leukocytoclastic vasculitis. To our knowledge, membranoproliferative glomerulonephritis (MPGN) has not been described before. We report a case of a 39-year-old male with autosomal dominant dystrophic EB, presenting with bilateral leg swelling of one-week duration. There was no other significant past medical history. The physical examination was remarkable for scars and erosions over all body areas, with all extremities with blisters and ulcers covered, absent finger and toenails and bilateral lower extremity edema. Serum creatinine was 0.9 mg/dL, albumin 1.3 g/dL and urine protein excretion 3.7 g/24 h. Viral markers (hepatitis-B, C, and HIV), complement c3 and c4 levels and auto-immune antibody profile all remained negative or within normal limits. Renal ultrasound and echocardiogram were normal. Renal biopsy recovered 14 glomeruli, all with proliferation of mesangial and endothelial cells as well as an expansion of the mesangial matrix, focal segmental sclerosis and amorphous homogeneous deposits demonstrating apple-green birefringence under polarized light with Congo red stain. Our observation emphasizes the importance of recognizing MPGN and secondary amyloidosis in patients with EB, especially with the availability of newer treatment modalities.

摘要

先前关于常染色体显性营养不良性大疱性表皮松解症(EB)成年患者肾小球疾病的报道有限,包括感染后肾小球肾炎、IgA 肾病、淀粉样变性和白细胞碎裂性血管炎。据我们所知,膜增生性肾小球肾炎(MPGN)以前尚未描述过。我们报告了一例 39 岁男性,患有常染色体显性营养不良性 EB,表现为双侧腿部肿胀,持续一周。无其他重大既往病史。体格检查的显著特征是全身所有区域的疤痕和侵蚀,所有四肢均有水疱和溃疡,无指甲和趾甲,双侧下肢水肿。血清肌酐为 0.9mg/dL,白蛋白 1.3g/dL,尿蛋白排泄 3.7g/24h。病毒标志物(乙型肝炎、丙型肝炎和 HIV)、补体 c3 和 c4 水平以及自身免疫抗体谱均为阴性或在正常范围内。肾脏超声和超声心动图正常。肾脏活检获得 14 个肾小球,所有肾小球均表现为系膜细胞和内皮细胞增生,以及系膜基质扩张、局灶性节段性硬化和无定形均匀沉积物,在偏振光下刚果红染色呈苹果绿双折射。我们的观察结果强调了在 EB 患者中识别 MPGN 和继发性淀粉样变性的重要性,特别是在有新的治疗方法的情况下。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/513b26f115a1/IRNF_A_1614056_F0005_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/393ce3b92993/IRNF_A_1614056_F0001_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/ddfef30dc3a4/IRNF_A_1614056_F0002_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/bdf246afe9ee/IRNF_A_1614056_F0003_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/b3c5306d31dd/IRNF_A_1614056_F0004_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/513b26f115a1/IRNF_A_1614056_F0005_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/393ce3b92993/IRNF_A_1614056_F0001_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/ddfef30dc3a4/IRNF_A_1614056_F0002_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/bdf246afe9ee/IRNF_A_1614056_F0003_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/b3c5306d31dd/IRNF_A_1614056_F0004_C.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/667f/6746271/513b26f115a1/IRNF_A_1614056_F0005_C.jpg

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