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Pierson 综合征严重表型患儿的 5 年随访及肾移植成功

Five-Year Follow-Up and Successful Kidney Transplantation in a Girl with a Severe Phenotype of Pierson Syndrome.

机构信息

Department of Pediatric Nephrology, Medical University of Lublin, Lublin, Poland.

Department of Nephrology, Kidney Transplantation and Hypertension, Children's Memorial Health Institute, Warsaw, Poland.

出版信息

Nephron. 2021;145(5):579-584. doi: 10.1159/000516247. Epub 2021 May 31.

DOI:10.1159/000516247
PMID:34058744
Abstract

Pierson syndrome (PIERSS) is a rare autosomal recessive disorder characterized by the combination of congenital nephrotic syndrome (CNS) and extrarenal symptoms including ocular malformations and neurodevelopmental deficits. PIERSS is caused by biallelic pathogenic variants in the LAMB2 gene leading to the defects of β2-laminin, the protein mainly expressed in the glomerular basement membrane, ocular structures, and neuromuscular junctions. Severe complications of PIERSS lead to the fatal outcome in early childhood in majority of the cases. We report a case of 5-year-old girl with severe phenotype of PIERSS caused by biallelic functional null variants of the LAMB2 gene. Due to consequences of CNS, the patient required bilateral nephrectomy and peritoneal dialysis since early infancy. The course was additionally complicated by tubulopathy, life-threatening infections, severe hypertension, erythropoietin-resistant anemia, generalized muscular hypotonia, neurogenic bladder, profound neurodevelopmental delay, epilepsy, gastrointestinal problems, secondary hypothyroidism, and necessity of repeated ocular surgery due to microcoria, cataract, and nystagmus. Due to multidisciplinary efforts, at the age of 4 years, the kidney transplantation was possible. Currently, the renal graft has an excellent function; however, the girl presents severe neurodevelopmental delay. The report presents a unique long-term follow-up of severe PIERSS with a few new phenotypical findings. It highlights the clinical problems and challenges in management of this rare condition.

摘要

皮尔逊综合征(PIERSS)是一种罕见的常染色体隐性遗传病,其特征是先天性肾病综合征(CNS)与眼部畸形和神经发育缺陷等肾外表现同时存在。PIERSS 由 LAMB2 基因的双等位致病性变异引起,导致β2-层粘连蛋白缺陷,该蛋白主要表达在肾小球基底膜、眼部结构和神经肌肉接头。PIERSS 的严重并发症导致大多数病例在幼儿期即致命。我们报告了一例 5 岁女孩,因 LAMB2 基因的双等位功能性缺失变异导致严重的 PIERSS 表型。由于 CNS 的后果,该患者从婴儿早期开始就需要双侧肾切除术和腹膜透析。病程还伴有肾小管病、危及生命的感染、严重高血压、促红细胞生成素抵抗性贫血、全身肌肉张力减退、神经源性膀胱、严重的神经发育迟缓、癫痫、胃肠道问题、继发甲状腺功能减退以及由于瞳孔小、白内障和眼球震颤需要反复眼部手术。由于多学科的努力,在 4 岁时进行了肾移植。目前,移植肾功能良好;然而,该女孩仍存在严重的神经发育迟缓。该报告对一例严重 PIERSS 进行了独特的长期随访,并提出了一些新的表型发现。它突出了管理这种罕见疾病的临床问题和挑战。

相似文献

1
Five-Year Follow-Up and Successful Kidney Transplantation in a Girl with a Severe Phenotype of Pierson Syndrome.Pierson 综合征严重表型患儿的 5 年随访及肾移植成功
Nephron. 2021;145(5):579-584. doi: 10.1159/000516247. Epub 2021 May 31.
2
A novel LAMB2 gene mutation associated with a severe phenotype in a neonate with Pierson syndrome.在一名患有皮尔逊综合征的新生儿中发现一种与严重表型相关的新型LAMB2基因突变。
Eur J Med Res. 2016 Apr 30;21:19. doi: 10.1186/s40001-016-0215-z.
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Kidney transplantation in a child with Pierson syndrome.患有皮尔逊综合征的儿童肾移植
Pediatr Transplant. 2017 Dec;21(8). doi: 10.1111/petr.13076. Epub 2017 Nov 1.
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Skeletal impairment in Pierson syndrome: Is there a role for lamininβ2 in bone physiology?皮尔逊综合征的骨骼损伤:层粘连蛋白β2在骨骼生理学中起作用吗?
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Neurodevelopmental deficits in Pierson (microcoria-congenital nephrosis) syndrome.皮尔森(小眼畸形-先天性肾病)综合征中的神经发育缺陷。
Am J Med Genet A. 2007 Feb 15;143(4):311-9. doi: 10.1002/ajmg.a.31564.
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Gastrointestinal symptoms as an extended clinical feature of Pierson syndrome: a case report and review of the literature.胃肠道症状作为 Pierson 综合征的扩展临床特征:病例报告及文献复习。
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