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罕见遗传性骨病的分子、表型特征及治疗前景

Molecular, phenotypic aspects and therapeutic horizons of rare genetic bone disorders.

作者信息

Faruqi Taha, Dhawan Naveen, Bahl Jaya, Gupta Vineet, Vohra Shivani, Tu Khin, Abdelmagid Samir M

机构信息

Nova Southeastern University Health Sciences Division, Fort-Lauderdale-Davie, FL 33314, USA.

Florida International University (FIU), Miami, FL 33174, USA.

出版信息

Biomed Res Int. 2014;2014:670842. doi: 10.1155/2014/670842. Epub 2014 Oct 22.

DOI:10.1155/2014/670842
PMID:25530967
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4230237/
Abstract

A rare disease afflicts less than 200,000 individuals, according to the National Organization for Rare Diseases (NORD) of the United States. Over 6,000 rare disorders affect approximately 1 in 10 Americans. Rare genetic bone disorders remain the major causes of disability in US patients. These rare bone disorders also represent a therapeutic challenge for clinicians, due to lack of understanding of underlying mechanisms. This systematic review explored current literature on therapeutic directions for the following rare genetic bone disorders: fibrous dysplasia, Gorham-Stout syndrome, fibrodysplasia ossificans progressiva, melorheostosis, multiple hereditary exostosis, osteogenesis imperfecta, craniometaphyseal dysplasia, achondroplasia, and hypophosphatasia. The disease mechanisms of Gorham-Stout disease, melorheostosis, and multiple hereditary exostosis are not fully elucidated. Inhibitors of the ACVR1/ALK2 pathway may serve as possible therapeutic intervention for FOP. The use of bisphosphonates and IL-6 inhibitors has been explored to be useful in the treatment of fibrous dysplasia, but more research is warranted. Cell therapy, bisphosphonate polytherapy, and human growth hormone may avert the pathology in osteogenesis imperfecta, but further studies are needed. There are still no current effective treatments for these bone disorders; however, significant promising advances in therapeutic modalities were developed that will limit patient suffering and treat their skeletal disabilities.

摘要

根据美国国家罕见病组织(NORD)的数据,一种罕见疾病折磨的患者不到20万。超过6000种罕见疾病影响着约十分之一的美国人。罕见的遗传性骨疾病仍然是美国患者致残的主要原因。由于对潜在机制缺乏了解,这些罕见的骨疾病也给临床医生带来了治疗挑战。本系统综述探讨了关于以下罕见遗传性骨疾病治疗方向的现有文献:纤维发育不良、戈谢病、进行性骨化性纤维发育不良、蜡油样骨病、多发性遗传性骨软骨瘤、成骨不全、颅骨干骺端发育不良、软骨发育不全和低磷酸酯酶症。戈谢病、蜡油样骨病和多发性遗传性骨软骨瘤的发病机制尚未完全阐明。ACVR1/ALK2通路抑制剂可能作为进行性骨化性纤维发育不良的一种可能治疗干预措施。已探索使用双膦酸盐和白细胞介素-6抑制剂治疗纤维发育不良,但仍需更多研究。细胞疗法、双膦酸盐联合疗法和人生长激素可能避免成骨不全的病理状况,但还需要进一步研究。目前这些骨疾病仍然没有有效的治疗方法;然而,在治疗方式上已取得了重大的、有前景的进展,这将减轻患者痛苦并治疗他们的骨骼残疾。

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