Literature DB >> 11994366

Measurement of tartrate-resistant acid phosphatase and the brain isoenzyme of creatine kinase accurately diagnoses type II autosomal dominant osteopetrosis but does not identify gene carriers.

Steven G Waguespack1, Siu L Hui, Kenneth E White, Kenneth A Buckwalter, Michael J Econs.   

Abstract

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.

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Year:  2002        PMID: 11994366     DOI: 10.1210/jcem.87.5.8497

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  12 in total

1.  Interferon Gamma-1b Does Not Increase Markers of Bone Resorption in Autosomal Dominant Osteopetrosis.

Authors:  Erik A Imel; Ziyue Liu; Dena Acton; Melissa Coffman; Netsanet Gebregziabher; Yan Tong; Michael J Econs
Journal:  J Bone Miner Res       Date:  2019-05-13       Impact factor: 6.741

2.  Diagnosis of osteopetrosis in bilateral congenital aural atresia: Turning point in treatment strategy.

Authors:  Ritu Verma; Manisha Jana; Ashu Seith Bhalla; Arvind Kumar; Rakesh Kumar
Journal:  World J Clin Pediatr       Date:  2016-05-08

3.  Acidification of the osteoclastic resorption compartment provides insight into the coupling of bone formation to bone resorption.

Authors:  Morten A Karsdal; Kim Henriksen; Mette G Sørensen; Jeppe Gram; Sophie Schaller; Morten H Dziegiel; Anne-Marie Heegaard; Palle Christophersen; Thomas J Martin; Claus Christiansen; Jens Bollerslev
Journal:  Am J Pathol       Date:  2005-02       Impact factor: 4.307

4.  Serum creatine kinase isoenzymes in children with osteogenesis imperfecta.

Authors:  P D'Eufemia; R Finocchiaro; A Zambrano; V Lodato; L Celli; S Finocchiaro; P Persiani; A Turchetti; M Celli
Journal:  Osteoporos Int       Date:  2016-08-25       Impact factor: 4.507

Review 5.  Advances in osteoclast biology resulting from the study of osteopetrotic mutations.

Authors:  T Segovia-Silvestre; A V Neutzsky-Wulff; M G Sorensen; C Christiansen; J Bollerslev; M A Karsdal; K Henriksen
Journal:  Hum Genet       Date:  2008-11-06       Impact factor: 4.132

Review 6.  Osteopetrosis.

Authors:  Zornitza Stark; Ravi Savarirayan
Journal:  Orphanet J Rare Dis       Date:  2009-02-20       Impact factor: 4.123

7.  Characterization of osteoclasts from patients harboring a G215R mutation in ClC-7 causing autosomal dominant osteopetrosis type II.

Authors:  Kim Henriksen; Jeppe Gram; Sophie Schaller; Bjarne H Dahl; Morten H Dziegiel; Jens Bollerslev; Morten A Karsdal
Journal:  Am J Pathol       Date:  2004-05       Impact factor: 4.307

8.  Characteristics of ClC7 Cl- channels and their inhibition in mutant (G215R) associated with autosomal dominant osteopetrosis type II in native osteoclasts and hClcn7 gene-expressing cells.

Authors:  Hiroshi Kajiya; Fujio Okamoto; Kimiko Ohgi; Akihiro Nakao; Hidefumi Fukushima; Koji Okabe
Journal:  Pflugers Arch       Date:  2009-06-19       Impact factor: 3.657

9.  Dissociation of bone resorption and bone formation in adult mice with a non-functional V-ATPase in osteoclasts leads to increased bone strength.

Authors:  Kim Henriksen; Carmen Flores; Jesper S Thomsen; Anne-Marie Brüel; Christian S Thudium; Anita V Neutzsky-Wulff; Geerling E J Langenbach; Natalie Sims; Maria Askmyr; Thomas J Martin; Vincent Everts; Morten A Karsdal; Johan Richter
Journal:  PLoS One       Date:  2011-11-07       Impact factor: 3.240

10.  A novel missense mutation in the CLCN7 gene linked to benign autosomal dominant osteopetrosis: a case series.

Authors:  Ban Mousa Rashid; Nawshirwan Gafoor Rashid; Ansgar Schulz; Georgia Lahr; Beston Faiek Nore
Journal:  J Med Case Rep       Date:  2013-01-09
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