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At 1 year post-operatively, the mean was 5. When looking at the changes in the GMFM crawling C , standing D , and walking and running E domains from baseline to 1 year, statistically significant improvements are observed.

For the total GMFM scores, there was a The PEDI mean-scaled scores improved in the mobility domain from We did not analyze the data at the 2-, 3-, and 4-year intervals due to the small sample size. Figures 1 , 2 , and 3 demonstrate that the gains made in ambulation, gross motor function, mobility, and self-care at 1 year are at least maintained at 4 years post-operatively. Table 3 Factors significantly associated with change from pre-operative to 12 months.

Previous reports indicated that FD rodding of the femur in children with OI involves a lower complication rate as compared to the Bailey—Dubow rod [ 25 ]. FD rodding was also reported to significantly improve both the global quality of life and walking in nine patients 14 rods , and presented a safe and useful implant for the treatment of lower limb deformities in patients with OI [ 26 ]. The objectives of the current study were to examine the functional outcomes of children with OI at 1 year following initial FD femoral rodding to the femora, and to determine which factors are predictive of change in gross motor function, ambulation, and functional performance.

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The functional outcomes at 1 year following initial FD femoral rodding in children with OI have not yet been reported in the literature. Ninety degrees of knee flexion is needed to climb and descend stairs alternately. Our experience suggests that the absence of a knee arthrotomy in the FD rodding system favors this high degree of AROM 5 weeks after surgery.

Does not take full weight on feet; does not walk on a routine basis [ 30 ]. At this stage, the child is able to walk more than 15—20 feet outside the home, but usually uses a wheelchair or stroller for community distances or in congested areas. As for gross motor function, total and individual domain scores increased from pre-operatively to 1 year.

The greatest gains were observed in the crawling C and standing D domains of the GMFM, where we would anticipate changes from baseline to 1 year. The goal of the FD femoral rodding is to facilitate standing and ambulation with a lower risk of fracture. It would be expected that the greatest improvement would be in the standing domain, especially for the children with type III OI, as these children do not always achieve independent ambulation.

The mean total GMFM score at 1 year is All items on the GMFM could usually be accomplished by a 5-year-old with normal motor abilities [ 37 ]. The gains in the mobility scores of the PEDI may reflect the impact that the FD rodding had on the correction of the lower limb deformity in our sample of children with OI, thus, resulting in better weight-bearing and ambulation and, consequently, in better performance in daily mobility activities, such as transferring to a car or taking a bath and climbing partial or full flights of stairs.

Gains were also observed in the self-care domain; however, these gains may be attributable to developmental achievements, as well as increased skill performance following FD femoral rodding. It has been our experience that children with OI who have been rodded in the lower extremities participate in more activities than prior to the surgery. As a result, they increase their risk for more falls and possible fractures.

It is reassuring to observe that the gains made at 1 year are maintained up until 4 years. The younger the child, the higher the incidence of re-rodding due to growth. Even in the older children who were not able to walk prior to surgery, their level of ambulation, gross motor skills, and independence in self-care improved.

In 11 children aged 4. It is our conclusion that these gains were primarily due to the surgical intervention, as children aged 4. We looked at the factors which were associated with change at 1 year.

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Our findings indicate that increasing age and weight is associated with lower scores in the self-care areas. Our clinical experience has demonstrated that children with physical limitations and increased weight are at higher risk for lower performance in daily life activities. We conclude that children with OI should be rodded as soon as they meet the criteria established. In our study, the children from South America and the Middle East would have benefited from earlier rodding.

At the time of surgery, their scores in the crawling C domain were lower. Our sample included children from local communities younger children , as well as children from abroad older children , with younger children probably having better prospects for developmental outcome.

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This may have attenuated the improvements for all patients at 12 months. The sample size of this study precluded analyses at the 2-, 3-, and 4-year time points, as many children came from abroad and returned on a needs basis. This study did not include a control group as withholding surgery for appropriate candidates would be deemed unethical. The outcome measures used in this study did not demonstrate a ceiling effect, especially in children with more severe OI types. Multisite studies with a longitudinal follow-up would strengthen our findings. The findings of this study are generalizable to children with OI who have undergone bisphosphonate treatment prior to intramedullary femoral rodding using the FD rod.

This study represents an examination of the functional outcomes 1 year post-initial Fassier—Duval FD rodding to the femora in children with osteogenesis imperfecta OI receiving bisphosphonates. The current study revealed that initial FD femoral rodding resulted in benefits in ambulation, gross motor function, self-care, and mobility for children with OI beyond physiological expectations due to developmental growth.

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The gains obtained persist for up to 4 years. This study provides useful information for clinicians on the post-operative course for children with OI. It is our opinion that FD rodding is a procedure which can improve the overall mobility in children with OI with significant femoral deformities. Special thanks go to the entire medical, nursing, and rehabilitation team at the Shriners Hospital for Children for their devoted care of children with osteogenesis imperfecta. Downloaded times. Original Clinical Article. Table 2 Comparisons between pre-operative and month outcomes Outcome measure: mean SD [ n ] Pre-operative 12 month Mean change between pre-operative and 12 months FAQ 2.

References 1. Glorieux FH. Osteogenesis imperfecta. Best Pract Res Clin Rheumatol ; — Curr Orthop ; — Genetic heterogeneity in osteogenesis imperfecta. J Med Genet ; — Rauch F, Glorieux FH. Lancet ; — Type V osteogenesis imperfecta: a new form of brittle bone disease. J Bone Miner Res ; — Osteogenesis imperfecta type VI: a form of brittle bone disease with a mineralization defect. J Bone Miner Res ; —38 Osteogenesis imperfecta type VII maps to the short arm of chromosome 3.

Bone ; —25 Pamidronate in children with osteogenesis imperfecta: histomorphometric effects of long-term therapy. J Clin Endocrinol Metab ; — Enright W, Noonan K. Bone plating in patients with type III osteogenesis imperfecta: results and complications.

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