Gait Deviations in Children with Osteogenesis Imperfecta Type I

Garman, Christina; Graf, Adam; Krzak, Joseph J.; Harris, Peter Smith Gerald

doi:10.1016/j.apmr.2017.08.222

Cited by 3 publications

(7 citation statements)

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“…The findings of reduced ankle push off power for OI group compared to healthy group [Figure 4] are aligned with that in the literature, viz., Graf et al . and Garman et al 212223. Although the number of subjects is too small to ascertain the effects of heterogeneous nature of type IV OI patients, the present results explain the gait characteristics of type IV OI subjects, which are consistent with the gait characteristics of type I OI subjects reported in the literature 2122.…”

Section: Discussionsupporting

confidence: 89%

“…Apart from the parameters reported in the Graf et al 21. and Garman et al .,22 the present study also provides the comparison of GRFs. These forces can be utilized to calculate joint reaction forces and moments at the knee and hip joints along with the calculation of muscle forces using the principles of inverse dynamics and static optimization respectively.…”

Section: Discussionmentioning

confidence: 86%

“…and Garman et al 212223. Although the number of subjects is too small to ascertain the effects of heterogeneous nature of type IV OI patients, the present results explain the gait characteristics of type IV OI subjects, which are consistent with the gait characteristics of type I OI subjects reported in the literature 2122. The information provided in the present study is important in clinical setting as it helps the clinicians, surgeons, and physiotherapists to effectively preplan the surgical intervention and rehabilitation for individual.…”

Section: Discussionmentioning

confidence: 95%

“…Reduced velocity in OI subjects also contribute to the decreased power generation as it has been reported in literature that shorter subjects have reduced velocity 24. Garman et al 22. also reported quantitative gait analysis with a large sample of 40 type I OI patients and compared them with age-matched typically developing children.…”

Section: Discussionmentioning

confidence: 96%

“…Limited studies in the literature presented quantitative knowledge on the gait in patients with OI. To our knowledge, there are specifically two studies on gait analysis of type I OI patients which are available in the literature 2122. A recent study of quantitative gait analysis for one type IV OI patient is also available in the literature 23.…”

Section: Introductionmentioning

confidence: 99%

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Kinetic and Kinematic Analysis of Gait in Type IV Osteogenesis Imperfecta Patients: A Comparative Study

Badhyal

Dhole

Gopinathan

et al. 2019

IJOO

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Background:Osteogenesis imperfecta (OI) is a genetic connective tissue disorder characterized by skeletal deformity and increased risk of fracture. Independent mobility is of concern for OI patients as it is associated with the quality of life. The present study investigates the variation of kinetic and kinematic gait parameters of type IV OI subjects and compares them with age-matched healthy subjects.Materials and Methods:Gait analysis is performed on five type IV OI patients and six age-matched normal subjects. Spatiotemporal, kinematic, and kinetic data are obtained using Helen Hayes marker placement protocol.Results:The results indicate an imprecise double-humped profile for vertical ground reaction force (GRF) with reduced ankle push off power and walking speed for OI subjects. Moreover, a comparison of vertical GRFs in OI subjects with that of healthy subjects suggests lower values for the former. The results encourage and motivate for further investigation with a bigger set of subjects.Conclusion:This information may be useful in developing a better understanding of pathological gait in type IV OI subjects, which ultimately helps the design of subject-specific implants, surgical preplanning, and rehabilitation.

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Section: Discussionsupporting

confidence: 89%

Section: Discussionmentioning

confidence: 86%

Section: Discussionmentioning

confidence: 95%

Section: Discussionmentioning

confidence: 96%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Kinetic and Kinematic Analysis of Gait in Type IV Osteogenesis Imperfecta Patients: A Comparative Study

Badhyal

Dhole

Gopinathan

et al. 2019

IJOO

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Quantitative gait analysis in children with osteogenesis imperfecta: relationship between gait deviations and clinical features

Wouters¹,

Detrembleur²,

Durnez³

et al. 2022

Acta Orthop Belg

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Osteogenesis imperfecta is a rare congenital disease of connective tissue characterized by recurrent fractures and progressive skeletal deformities which may impact on gait. The aims of this prospective study were to identify gait deviations in children with osteogenesis imperfecta compared to age-matched controls and establish relationships with clinical features. We evaluated 22 patients with different types of osteogenesis imperfecta using three-dimensional gait analysis. The incidence and location of frac- tures, fracture at birth, age at first fracture, use of intramedullary rodding and number of surgical in- terventions in the lower extremities, bone mineral density, hypermobility and number of injections of bisphosphonates were recorded for each patient. Step length was lower in the osteogenesis imperfecta group compared with the control group. Kinematics showed that sagittal pelvic and transversal hip range of motion were higher in the osteogenesis imperfecta group, whereas sagittal knee range of motion during swing phase was reduced. Regarding kinetics, hip flexion moment and hip negative power peak were significantly decreased in the osteogenesis imperfecta group. Mechanical and energetic parameters were considered as normal. The principal component analysis revealed that the bone mineral density was increased in children who had received more in- jections of bisphosphonates and these had also less deficit in kinematic parameters. Main modifications in gait parameters were observed in spatiotemporal, kinematic and kinetic data. More studies are necessary to allow stratification of severity of the osteogenesis imperfecta disease, help improve its challenging multidisciplinary treatment and ob- jectively assess treatment outcomes.

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Tendon and motor phenotypes in the Crtap-/- mouse model of recessive osteogenesis imperfecta

Grol

Haelterman

Lim

et al. 2021

eLife

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Osteogenesis imperfecta (OI) is characterized by short stature, skeletal deformities, low bone mass, and motor deficits. A subset of OI patients also present with joint hypermobility; however, the role of tendon dysfunction in OI pathogenesis is largely unknown. Using the Crtap-/- mouse model of severe, recessive OI, we found that mutant Achilles and patellar tendons were thinner and weaker with increased collagen cross-links and reduced collagen fibril size at 1- and 4-months compared to wildtype. Patellar tendons from Crtap-/- mice also had altered numbers of CD146+CD200+ and CD146-CD200+ progenitor-like cells at skeletal maturity. RNA-seq analysis of Achilles and patellar tendons from 1-month Crtap-/- mice revealed dysregulation in matrix and tendon marker gene expression concomitant with predicted alterations in TGF-b, inflammatory, and metabolic signaling. At 4-months, Crtap-/- mice showed increased aSMA, MMP2, and phospho-NFkB in the patellar tendon consistent with excess matrix remodeling and tissue inflammation. Finally, a series of behavioral tests showed severe motor impairments and reduced grip strength in 4-month Crtap-/- mice – a phenotype that correlates with the tendon pathology.

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Gait Deviations in Children with Osteogenesis Imperfecta Type I

Cited by 3 publications

References 0 publications

Kinetic and Kinematic Analysis of Gait in Type IV Osteogenesis Imperfecta Patients: A Comparative Study

Kinetic and Kinematic Analysis of Gait in Type IV Osteogenesis Imperfecta Patients: A Comparative Study

Quantitative gait analysis in children with osteogenesis imperfecta: relationship between gait deviations and clinical features

Tendon and motor phenotypes in the Crtap-/- mouse model of recessive osteogenesis imperfecta

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