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Cranial base pathology in pediatric osteogenesis imperfecta patients treated with bisphosphonates
- Source :
- Journal of Neurosurgery: Pediatrics. 15:313-320
- Publication Year :
- 2015
- Publisher :
- Journal of Neurosurgery Publishing Group (JNSPG), 2015.
-
Abstract
- OBJECT Cranial base pathology is a serious complication of osteogenesis imperfecta (OI). Our aim was to analyze whether bisphosphonate treatment, used to improve bone strength, could also prevent the development of craniocervical junction pathology (basilar impression, basilar invagination, or platybasia) in children with OI. METHODS In this single-center retrospective study the authors analyzed the skull base morphology from lateral skull radiographs and midsagittal MR images (total of 94 images), obtained between the ages of 0 and 25 years in 39 bisphosphonate-treated OI patients. The results were compared with age-matched normative values and with findings in 70 OI patients who were not treated with bisphosphonates. In addition to cross-sectional data, longitudinal data were available from 22 patients with an average follow-up period of 7.6 years. The patients, who had OI types I, III, IV, VI, and VII, had been treated with zoledronic acid, pamidronate, or risedronate for 3.2 years on average. RESULTS Altogether 33% of the 39 bisphosphonate-treated patients had at least 1 cranial base anomaly, platybasia being the most prevalent diagnosis (28%). Logistic regression analysis suggested a higher risk of basilar impression or invagination in patients with severe OI (OR 22.04) and/or older age at initiation of bisphosphonate treatment (OR 1.45), whereas a decreased risk was associated with longer duration of treatment (OR 0.28). No significant associations between age, height, or cumulative bisphosphonate dose and the risk for cranial base anomaly were detected. In longitudinal evaluation, Kaplan-Meier curves suggested delayed development of cranial base pathology in patients treated with bisphosphonates but the differences from the untreated group were not statistically significant. CONCLUSIONS These findings indicate that cranial base pathology may develop despite bisphosphonate treatment. Early initiation of bisphosphonate treatment may delay development of craniocervical junction pathology. Careful followup of cranial base morphology is warranted, particularly in patients with severe OI.
- Subjects :
- Male
medicine.medical_specialty
Pathology
Adolescent
Bone disease
Radiography
Pamidronate
Dentistry
030209 endocrinology & metabolism
Basilar invagination
Kaplan-Meier Estimate
Zoledronic Acid
Drug Administration Schedule
03 medical and health sciences
0302 clinical medicine
Platybasia
Humans
Medicine
Child
Retrospective Studies
Skull Base
Bone Density Conservation Agents
Diphosphonates
business.industry
Imidazoles
Infant
Etidronic Acid
General Medicine
Osteogenesis Imperfecta
medicine.disease
Surgery
Skull
Logistic Models
medicine.anatomical_structure
Zoledronic acid
Osteogenesis imperfecta
Child, Preschool
Female
business
Complication
Risedronic Acid
030217 neurology & neurosurgery
Follow-Up Studies
medicine.drug
Subjects
Details
- ISSN :
- 19330715 and 19330707
- Volume :
- 15
- Database :
- OpenAIRE
- Journal :
- Journal of Neurosurgery: Pediatrics
- Accession number :
- edsair.doi.dedup.....6709964c11c68a07d438a934d6716877