Serum brain-type creatine kinase increases in children with osteogenesis imperfecta during neridronate treatment

D’Eufemia, P; Finocchiaro, R; Villani, Ciro; Zambrano, Anna; Lodato, Valentina; Palombaro, Marta; Properzi, Enrico; Celli, Mauro

doi:10.1038/pr.2014.20

Cited by 7 publications

(12 citation statements)

References 24 publications

(35 reference statements)

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“…This study included 18 children affected by OI type 1 who received 1 year neridronate treatment as described in our previous report [20]. Twelve children, five boys, and seven girls (mean age [years] ± standard deviation [SD]: 6.7 ± 1.5) continued neridronate treatment throughout the 2-year follow-up period (group A).…”

Section: Methodsmentioning

confidence: 99%

“…Recently, BP treatment was reported to result in increased CK release from rabbit osteoclasts in vitro [21]. This result explains a possible mechanism for increased serum CK in patients treated with BPs [11,20].…”

Section: Introductionmentioning

confidence: 94%

“…In 2004, we reported an increased serum CKbb level in 18 children with OI type 1 during the first year of treatment with neridronate [20]. Recently, BP treatment was reported to result in increased CK release from rabbit osteoclasts in vitro [21].…”

Section: Introductionmentioning

confidence: 97%

“…In the present study, we evaluated serum CK isoenzymes in the same 18 children used in our previous study during a further 2-year follow-up period [20]. The study included two groups: subjects who continued with neridronate treatment and subjects who discontinued neridronate treatment.…”

Section: Introductionmentioning

confidence: 99%

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Serum creatine kinase isoenzymes in children with osteogenesis imperfecta

et al. 2016

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This study provides evidence of the cumulative effect of neridronate administration in increasing serum CKbb levels and the reversible effect after its discontinuation. This approach could be employed for verifying the usefulness of serum CKbb as a biochemical marker in patients receiving prolonged bisphosphonate treatment. Moreover, the decreased serum CKmb levels suggest a systemic effect of these drugs.

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Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 94%

Section: Introductionmentioning

confidence: 97%

Section: Introductionmentioning

confidence: 99%

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Serum creatine kinase isoenzymes in children with osteogenesis imperfecta

et al. 2016

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“…We cannot exclude a multi-factorial etiopathogenesis where the exposure to bisphosphonates also plays a role. In a study conducted by D’Eufemia et al, [ 9 ] it is described that, even in the case of an interruption in the administration of neridronate, the drug remains in the circulation of patient's organism for over 2 years. In our case, this could have been the timeline in which our patient was exposed, during pregnancy, to doses of bisphosphonate that were sufficient enough to generate the clubfoot, despite the therapy interruption by part of the mother.…”

Section: Discussionmentioning

confidence: 99%

Osteogenesis imperfecta and clubfoot—a rare combination

et al. 2016

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Background:Osteogenesis imperfecta (OI) is a rare congenital genetic osteodystrophy, which has a prevalence of 1:20,000. OI is caused by the mutation of the COL1A1/COL1A2 genes, leading to a deficit of quality and/or quantity in the synthesis of procollagen-α type 1. Seven different forms of diverse clinical entity have been classified by Sillence and Glorieux, although, recently, up to 11 forms characterized by different genetic mutations have been recognized. Patients with OI suffer from extreme bone fragility and osteoporosis, which often predisposes them to frequent fractures. This paper presents the case of a child with OI type IV who, at birth, was also diagnosed with a severe clubfoot (congenital talipes equinovarus) grade III. Patient's mother also suffers from OI type IV.Methods:The treatment was started by placing femoro-podalic corrective casts, according to the Ponseti method, but some unexpected problems occurred during this treatment. When the patient was 3 months of age, we decided to correct the clubfoot before the time limit planned, performing a bilateral posteromedial surgical release.Results:Three weeks after surgery the casts were removed and replaced with bilateral Spica cast-like braces. On the 6th postoperative week, the patient began wearing Bebax corrective shoes, after 1 year ambidextrous orthopedic shoes. Now, he is 2 years old and has started to walk properly without any orthesis.Conclusion:In the presence of an orthopedic pathology associated with OI, it is recommended to manage the patient according to the underlying pathology, always considering the bone fragility associated with OI. The final surgical treatment to correct the clubfoot can be done earlier, if necessary. In our opinion, this uncommon association between OI and clubfoot is non-syndromic. This means that the two congenital diseases are not necessarily included in a singular uncommon genetic syndrome, but the clubfoot was caused by multifactorial causes, especially by both the mother's bisphosphonate drug therapy and the amniocentesis performed during her pregnancy to drain polyhydramnios. In our analysis, those environmental factors could have interacted with an already altered genetic substratum, contributing to develop this rare combination of congenital disorders.

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Future Perspectives of Bisphosphonates in Maxillofacial, Dental, and Medical Practice

Smeets

Hanken

Jung

et al. 2014

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Serum brain-type creatine kinase increases in children with osteogenesis imperfecta during neridronate treatment

Cited by 7 publications

References 24 publications

Serum creatine kinase isoenzymes in children with osteogenesis imperfecta

Serum creatine kinase isoenzymes in children with osteogenesis imperfecta

Osteogenesis imperfecta and clubfoot—a rare combination

Future Perspectives of Bisphosphonates in Maxillofacial, Dental, and Medical Practice

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