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造血干细胞移植纠正了一名携带FERMT3基因新纯合突变儿童的骨质石化症。

Hematopoietic stem cell transplantation corrects osteopetrosis in a child carrying a novel homozygous mutation in the FERMT3 gene.

作者信息

Palagano Eleonora, Slatter Mary A, Uva Paolo, Menale Ciro, Villa Anna, Abinun Mario, Sobacchi Cristina

机构信息

Humanitas Clinical and Research Institute, Rozzano, Italy; Department of Medical Biotechnologies and Translational Medicine, University of Milan, Milan, Italy.

Bone Marrow Transplantation Unit, Great North Children's Hospital, The Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UK; Primary Immunodeficiency Group, Institute of Cellular Medicine, Newcastle University, Newcastle upon Tyne, UK.

出版信息

Bone. 2017 Apr;97:126-129. doi: 10.1016/j.bone.2017.01.012. Epub 2017 Jan 14.

DOI:10.1016/j.bone.2017.01.012
PMID:28095295
Abstract

Osteopetrosis (OPT) is a rare skeletal disorder with phenotypic and genotypic heterogeneity: a variety of clinical features besides the bony defect may be present, and at least ten different genes are known to be involved in the disease pathogenesis. In the framework of this heterogeneity, we report the clinical description of a neonate, first child of consanguineous parents, who had osteoclast-rich osteopetrosis and bone marrow failure in early life, but no other usual classical features of infantile malignant OPT, such as visual or hearing impairments. Because of the severe presentation at birth, the patient received Hematopoietic Stem Cell Transplantation (HSCT) at 2months of age with successful outcome. Post-HSCT genetic investigation by means of exome sequencing identified a novel homozygous mutation in the Fermitin Family Member 3 (FERMT3) gene, which was predicted to disrupt the functionality of its protein product kindlin 3. Our report provides information relevant to physicians for recognizing patients with one of the rarest forms of infantile malignant OPT, and clearly demonstrates that HSCT cures kindlin 3 deficiency with severe phenotype.

摘要

骨质石化症(OPT)是一种罕见的骨骼疾病,具有表型和基因型异质性:除骨缺陷外,可能还存在多种临床特征,并且已知至少有十个不同的基因参与该疾病的发病机制。在这种异质性的背景下,我们报告了一名新生儿的临床情况,该新生儿是近亲结婚父母的第一个孩子,患有富含破骨细胞的骨质石化症且在生命早期出现骨髓衰竭,但没有婴儿恶性OPT的其他常见典型特征,如视力或听力障碍。由于出生时病情严重,该患者在2个月大时接受了造血干细胞移植(HSCT),结果成功。HSCT后通过外显子组测序进行的基因研究在FERMT3(FERM结构域含蛋白家族成员3)基因中发现了一种新的纯合突变,预计该突变会破坏其蛋白质产物整合素连接激酶3的功能。我们的报告为医生识别患有最罕见形式的婴儿恶性OPT的患者提供了相关信息,并清楚地表明HSCT可治愈具有严重表型的整合素连接激酶3缺乏症。

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J Clin Immunol. 2023 Aug;43(6):1250-1258. doi: 10.1007/s10875-023-01479-7. Epub 2023 Apr 4.
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Bone marrow transplantation as a therapy for autosomal dominant osteopetrosis type 2 in mice.骨髓移植作为治疗常染色体显性遗传骨硬化症 2 型的方法在小鼠中。
FASEB J. 2022 Sep;36(9):e22471. doi: 10.1096/fj.202200678R.
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RNA-seq analysis of chondrocyte transcriptome reveals genetic heterogeneity in LG/J and SM/J murine strains.
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Osteoarthritis Cartilage. 2020 Apr;28(4):516-527. doi: 10.1016/j.joca.2020.01.001. Epub 2020 Jan 14.
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One Disease, Many Genes: Implications for the Treatment of Osteopetroses.一种疾病,多个基因:对骨硬化症治疗的启示
Front Endocrinol (Lausanne). 2019 Feb 19;10:85. doi: 10.3389/fendo.2019.00085. eCollection 2019.
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