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低磷性骨软化症:关于病理生理学、临床表现、诊断及治疗的当前文献

Hypophosphatasia: Current Literature for Pathophysiology, Clinical Manifestations, Diagnosis, and Treatment.

作者信息

Bangura Abdulai, Wright Lisa, Shuler Thomas

机构信息

Department of Research, Trinity School of Medicine, Ratho Mill, VCT.

Department of Orthopaedics, Carilion Clinic, Roanoke, USA.

出版信息

Cureus. 2020 Jun 13;12(6):e8594. doi: 10.7759/cureus.8594.

Abstract

Hypophosphatasia (HPP) is a rare inherited bone disorder identified by impaired bone mineralization. There are seven subtypes of HPP mainly characterized by their age of onset. These subtypes consist of perinatal (prenatal) benign, perinatal lethal, infantile, childhood, adult, odontohypophosphatasia, and pseudohypophosphatasia. Due to limited awareness of the condition, either misdiagnosis or delayed diagnosis is common. Furthermore, the condition is frequently treated with contraindicated drugs. This literature illustrates the most recent findings on the etiology, pathophysiology, clinical manifestations, diagnosing, and treatment for HPP and its subtypes. The etiology of the disease consists of loss-of-function mutations of the ALPL gene on chromosome one, which encodes for tissue nonspecific isoenzyme of alkaline phosphatase (TNAP). A decrease of TNAP reduces inorganic phosphate (Pi) for bone mineralization and allows for an increase in inorganic pyrophosphate (PPi) and phosphorylated osteopontin (p-OPN), which further reduces bone mineralization. The combination of these processes softens bone and mediates a clinical presentation similar to rickets/osteomalacia. HPP has an additional wide range of clinical features depending on its subtype. Although a concrete diagnostic guideline has not yet been established, many studies have supported a similar method of identifying HPP. Clinical features, radiological findings, and/or biomarker levels of the disorder should raise suspicion and encourage the inclusion of HPP as a differential diagnosis. Biomarkers, especially alkaline phosphatase (ALP), are major contributors to diagnosis. However, genetic testing is done for definitive diagnosis. The primary treatment for HPP is the reintroduction of TNAP as a recombinant enzyme called asfotase alfa. There are additional pharmaceutical treatments and in some cases, surgical intervention may be indicated. Pharmaceutical therapies such as bisphosphonates, denosumab, potent antiresorptive agents, and vitamin D are contraindicated in adults with HPP. We hope to raise awareness for HPP in order to prevent delayed diagnosis or misdiagnosis. We plan to encourage appropriate care and avoid treatments that may be contraindicating. We also encourage the development of a diagnostic guideline that will promote a consistently favorable patient prognosis.

摘要

低磷性骨软化症(HPP)是一种罕见的遗传性骨病,其特征为骨矿化受损。HPP有七种亚型,主要根据发病年龄来区分。这些亚型包括围产期(产前)良性型、围产期致死型、婴儿型、儿童型、成人型、牙本质低磷性骨软化症和假性低磷性骨软化症。由于对该病的认识有限,误诊或诊断延迟很常见。此外,该病还经常使用禁忌药物进行治疗。本文阐述了关于HPP及其亚型的病因、病理生理学、临床表现、诊断和治疗的最新研究结果。该病的病因是1号染色体上的ALPL基因发生功能丧失突变,该基因编码组织非特异性碱性磷酸酶(TNAP)同工酶。TNAP减少会降低用于骨矿化的无机磷酸盐(Pi),并使无机焦磷酸盐(PPi)和磷酸化骨桥蛋白(p-OPN)增加,这会进一步降低骨矿化。这些过程的综合作用会使骨骼软化,并导致类似于佝偻病/骨软化症的临床表现。根据亚型不同,HPP还有一系列广泛的临床特征。尽管尚未建立具体的诊断指南,但许多研究都支持采用类似的方法来识别HPP。该疾病的临床特征、影像学表现和/或生物标志物水平应引起怀疑,并促使将HPP纳入鉴别诊断。生物标志物,尤其是碱性磷酸酶(ALP),是诊断的主要依据。然而,确诊需要进行基因检测。HPP的主要治疗方法是重新引入TNAP,即一种名为阿法骨化醇的重组酶。还有其他药物治疗方法,在某些情况下,可能需要进行手术干预。双膦酸盐、地诺单抗、强效抗吸收剂和维生素D等药物治疗对成人HPP患者是禁忌的。我们希望提高对HPP的认识,以防止诊断延迟或误诊。我们计划鼓励进行适当的治疗,并避免使用可能禁忌的治疗方法。我们还鼓励制定诊断指南,以促进患者始终获得良好的预后。

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Clinical Practice Guidelines for Hypophosphatasia.低磷酸酯酶症临床实践指南。
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