Waguespack Steven G, Hui Siu L, White Kenneth E, Buckwalter Kenneth A, Econs Michael J
Department of Medicine, Indiana University School of Medicine, Indianapolis, Indiana 46202, USA.
J Clin Endocrinol Metab. 2002 May;87(5):2212-7. doi: 10.1210/jcem.87.5.8497.
Autosomal dominant osteopetrosis type II (ADO2) is typically diagnosed from radiographs, which demonstrate the pathognomonic findings of osteosclerosis and endobone formation. Individuals with ADO2 also have elevated serum levels of tartrate-resistant acid phosphatase (TRAP) and the BB isoenzyme of creatine kinase (CK-BB). In the current study, we tested the utility of these enzymes in making or refuting a diagnosis of ADO2. Furthermore, because ADO2 has incomplete penetrance, we examined whether TRAP and CK-BB were helpful in identifying gene carriers. We studied eight families, measured serum levels of TRAP and CK-BB in 52 affected individuals and 12 obligate gene carriers, and compared their values with age-matched controls. Our results demonstrate that affected patients have significantly elevated levels of both TRAP and CK-BB. In contrast, gene carriers have values that are not different from controls. Furthermore, in our study population, TRAP and CK-BB have a high diagnostic sensitivity and specificity, particularly in children. From this large study of ADO2 patients and carriers, we conclude that: 1) TRAP and CK-BB are significantly elevated in patients with ADO2, 2) obligate carriers cannot be adequately identified by measurement of these analytes, and 3) TRAP and CK-BB are highly sensitive and specific diagnostic tests that can efficiently and effectively screen high-risk individuals who have not had previous radiographic assessment.
常染色体显性遗传性II型骨硬化症(ADO2)通常通过X线片进行诊断,X线片可显示骨硬化和骨内形成的特征性表现。ADO2患者的血清抗酒石酸酸性磷酸酶(TRAP)和肌酸激酶BB同工酶(CK-BB)水平也会升高。在本研究中,我们测试了这些酶在确诊或排除ADO2诊断中的作用。此外,由于ADO2具有不完全外显率,我们研究了TRAP和CK-BB是否有助于识别基因携带者。我们研究了8个家系,检测了52例患者和12例确定的基因携带者的血清TRAP和CK-BB水平,并将其值与年龄匹配的对照组进行比较。我们的结果表明,患者的TRAP和CK-BB水平均显著升高。相比之下,基因携带者的值与对照组无差异。此外,在我们的研究人群中,TRAP和CK-BB具有较高的诊断敏感性和特异性,尤其是在儿童中。通过对ADO2患者和携带者的这项大型研究,我们得出以下结论:1)ADO2患者的TRAP和CK-BB显著升高;2)通过检测这些分析物无法充分识别确定的携带者;3)TRAP和CK-BB是高度敏感和特异的诊断测试,能够有效且高效地筛查未进行过先前X线评估的高危个体。