评估低碱性磷酸酶水平在临床实践:对诊断低磷酸症的意义。

IF 3.2 3区 医学 Q2 ENDOCRINOLOGY & METABOLISM
Gonul Buyukyilmaz, Serkan Bilge Koca, Refika Gören, Andac Uzdogan, Keziban Toksoy Adıguzel, Aylin Kılınç Uğurlu, Gonul Yardimci, Pınar Kocaay, Derya Tepe, Mehmet Boyraz, Esra Kilic, Fatih Gürbüz
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引用次数: 0

摘要

持续的低血清碱性磷酸酶(ALP)水平是识别遗传性疾病如低磷酸酶(HPP)的关键。本研究探讨儿童低ALP水平的原因,旨在评估诊断为HPP的人口统计学和临床特征。我们在2019年9月至2024年7月期间评估了2243名年龄在0至19岁之间的儿童和青少年,他们表现出至少两个ALP水平低于年龄和性别的下限。在患者组中,95.4%(2140例)表现为一过性低ALP水平,4.6%(103例)表现为持续低ALP水平。在持久组中,除HPP外,确定了11种额外的医疗条件,其中卡路里消耗(厌食症,营养不良)是最常见的原因。研究发现16例HPP患者(10名女性,6名男性)即使在相同的变异中也具有高表型变异性,占整个组的0.71%。基因检测发现13个致病或可能致病的ALPL基因变异(10个杂合,3个纯合),其中2个为新变异。在HPP患者中,56.2%出现HPP相关症状,最常见的是身材矮小。我们发现总ALP与血清磷酸吡哆醛(PLP)水平呈显著负相关(Rho = - 0.55, p = 0.039),但与尿磷酸乙醇胺无相关性。持续低ALP水平是一系列疾病的重要临床指标,尤其是HPP。本研究扩展了HPP的表型和基因型谱,同时提高了我们对儿童疾病的认识。提高对疾病的认识,特别是对较轻疾病的认识,对于避免延误诊断至关重要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Evaluation of Low Alkaline Phosphatase Levels in Clinical Practice: Implications for Diagnosing Hypophosphatasia.

Persistent low serum alkaline phosphatase (ALP) levels are crucial in identifying genetic disorders such as hypophosphatasia (HPP). This study investigates the causes of low ALP levels in children, aiming to evaluate the demographic and clinical characteristics of those diagnosed with HPP.We evaluated 2243 children and adolescents, ranging from 0 to 19 years old between September 2019 and July 2024, who exhibited at least two ALP levels below the age- and gender-specific lower limit.In the patient group, 95.4% (2140 patients) exhibited transient low ALP levels, while 4.6% (103 patients) showed persistently low levels. In the persistent group, eleven additional medical conditions were identified, excluding HPP, with calorie depletion (anorexia, malnutrition) being the most common cause. The study identified 16 HPP patients (10 females, 6 males) with high phenotypic variability even within the same variants, comprising 0.71% of the whole group. Genetic testing identified 13 pathogenic/likely pathogenic ALPL gene variants (10 heterozygous, 3 homozygous), two of which were novel. Among HPP patients, 56.2% presented with HPP-related symptoms, most commonly short stature. We found a significant negative correlation between total ALP and serum pyridoxal phosphate (PLP) levels (Rho = - 0.55, p = 0.039), but no correlation with urine phosphoethanolamine.Persistently low ALP levels are a vital clinical indicator for a wide range of disorders, especially HPP. This study expands the phenotypic and genotypic profiles of HPP while improving our understanding of the disease in children. Increasing disease awareness, particularly for milder forms, is essential to avoid delayed diagnosis.

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来源期刊
Calcified Tissue International
Calcified Tissue International 医学-内分泌学与代谢
CiteScore
8.00
自引率
2.40%
发文量
112
审稿时长
4-8 weeks
期刊介绍: Calcified Tissue International and Musculoskeletal Research publishes original research and reviews concerning the structure and function of bone, and other musculoskeletal tissues in living organisms and clinical studies of musculoskeletal disease. It includes studies of cell biology, molecular biology, intracellular signalling, and physiology, as well as research into the hormones, cytokines and other mediators that influence the musculoskeletal system. The journal also publishes clinical studies of relevance to bone disease, mineral metabolism, muscle function, and musculoskeletal interactions.
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