Intravenous (iv) bisphosphonates are widely used to treat the skeletal manifestations of osteogenesis imperfecta (OI). Obtaining peripheral iv access in pediatric patients with OI is often difficult and traumatic. Although this may be mitigated with surgically placed iv ports (port-a-caths), surgeons may be hesitant to perform this procedure on these children because of the lack of safety data. This study aims to gain better insight into the safety and efficacy of port-a-cath use in this population and identify risk factors for port-acath complications. In the present study, we conducted a retrospective cohort analysis of patient characteristics and the incidence of port-a-cath-related complications in children with OI. Fifty-three port-a-caths were placed in 29 children (21 males and 8 females). Of the 29 patients, most are OI type III (n = 18), followed by type I (n = 4), type IV (n = 4), and type V (n = 3). At the time of initial port-acath placement, the median age was 52 months (10-191 months), and the median weight was 7.9 kg (5.1-41.1 kg). Most patients (n = 20) weighed less than 10 kg during initial placement. Weight correlated significantly with OI type (p = 0.048), sex (p = 0.03), and vessel used (p = 0.02). Median initial port-a-cath longevity was 43 months (1-113 months), and we found no significant difference in port-a-cath longevity between sexes, OI types, or vessels used. Most patients (n = 19) required multiple port-a-cath placements. There is a significant difference (p = 0.02) between the number of placements and OI type, with type IV having more than type III. Port-a-cath removal was almost always due to mechanical complications (n = 19) but also for infection (n = 1) and malposition (n = 1). Eight patients still had their initial port-a-caths in place at the conclusion of this study. These findings indicate that complications associated with port-a-cath placement are mild and can be used to safely deliver iv bisphosphonates to pediatric OI patients.
INTRODUCTION A wide spectrum of diseases and disorders compromise bone mineral density (BMD) and bone microstructure, thus increasing the risk of bone damage and fracture. Clinically, bone imaging via dual energy X‐ray absorptiometry (DXA) and qualitative computed tomography (CT) allows for non‐destructive three‐dimensional structural analysis, but translational bone and mineral science often demands methodology that is beyond the capabilities of in vivo imaging. Human subject, transilial bone biopsy is a gold standard in translational bone and mineral science, allowing for quantitative micro‐CT and histomorphometry analyses. These analyses, however, require consent to elective surgical collection of a transilial bone biopsy, a taxing process that includes recruiting and retaining human research subjects. The purpose of this study was to review and demonstrate the use of cadaveric transilial bone analyses as an effective and economic approach to studying bone health. METHODS The NIH National Library of Medicine electronic database was used to perform a systematic literature review according to the following cadaveric, BMD, and bone microstructure keywords: (human cadaver) AND (bone) AND (mineral density) AND (micro‐CT) OR (histomorphometry). Resulting citations were analyzed for effective and economic methodology for studying bone health. Additionally, quantitative micro‐CT and histomorphometry were performed on a cadaveric transilial bone biopsy to demonstrate protocol efficacies. 1) Superficial‐lateral gluteal muscles were reflected for open field access the gluteal surface of the iliac ala. A transilial biopsy containing both the external and internal iliac cortices was collected using an 8mm trephine at the standard human subject site, approximately 2 cm posterior and inferior to the anterior‐superior iliac spine. 2) Biopsies were subjected to compact cone‐beam tomography (micro‐CT‐40, Scanco Medical AG, Bassersdorf, Switzerland). Using 16um resolution, 3D isotropic images were collected with an integration of 250 milli‐seconds. 3) Biopsies were subjected to gradual dehydration in graded ethanol and acetone and subsequently embedded in methyl methacrylate. Serial sections were obtained with an otorizedmicrotome, stained, and mounted on glass slides. RESULTS The systematic literature review yielded seventy‐eight citations demonstrating the utilization of human cadaveric bone to analyze bone mineral density (BMD) and bone microstructure. Twelve citations were excluded due to analysis of non‐human specimens, reporting of a case study, or manuscript access limitations. Protocol efficacies were confirmed with quantification of bone volume, connectivity density, structure model index, trabecular number, trabecular thickness, and trabecular separation via quantitative micro‐CT and histomorphometry analysis of cadaveric transilial biopsies. CONCLUSION Cadaveric transilial bone biopsy analysis via quantitative micro‐CT and histomorphometry provides an effective and economic approach to studying diseases and disorders ...
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