Longitudinal fibular deficiency: a cross-sectional study comparing lower limb function of children and young people with that of unaffected peers

Joshua W. Pate, Mark J. Hancock, Louise Tofts, Adrienne Epps, Jennifer N. Baldwin, Marnee J. McKay, Joshua Burns, Eleanor Morris, Verity Pacey

Research output: Contribution to journalArticleResearchpeer-review

Abstract

Longitudinal fibular deficiency (LFD), or fibular hemimelia, is congenital partial or complete absence of the fibula. We aimed to compare the lower limb function of children and young people with LFD to that of unaffected peers. A cross-sectional study of Australian children and young people with LFD, and of unaffected peers, was undertaken. Twenty-three (12 males) children and young people with LFD (74% of those eligible) and 213 unaffected peers, all aged 7–21 years were subject to the Knee Osteoarthritis Outcome Score (KOOS/KOOS-Child) and the Cumberland Ankle Instability Tool (CAIT/CAIT-Youth). Linear regression models compared affected children and young people to unaffected peers. Participants with LFD scored lower in both outcomes (adjusted p < 0.05). The difference between participants with LFD and unaffected peers was significantly greater among younger participants than older participants for KOOS activities and sports domain scores (adjusted p ≤ 0.01). Differences in the other KOOS domains (pain/symptoms/quality of life) and ankle function (CAIT scores) were not affected by age (adjusted p ≥ 0.08). Children and young people with LFD on average report reduced lower limb function compared to unaffected peers. Knee-related activities and sports domains appear to be worse in younger children with LFD, and scores in these domains become closer to those of unaffected peers as they become older.
LanguageEnglish
Article number45
Pages1-11
Number of pages11
JournalChildren
Volume6
Issue number3
DOIs
Publication statusPublished - 15 Mar 2019

Fingerprint

Lower Extremity
Cross-Sectional Studies
Ankle
Sports
Linear Models
Ectromelia
Fibula
Knee Osteoarthritis
Knee
Quality of Life
Pain

Bibliographical note

Copyright the Author(s) 2019. Version archived for private and non-commercial use with the permission of the author/s and according to publisher conditions. For further rights please contact the publisher.

Keywords

  • longitudinal fibular deficiency
  • lower limb function
  • children
  • young people
  • unaffected peers
  • KOOS
  • KOOS-Child
  • CAIT
  • CAIT-Youth

Cite this

Pate, Joshua W. ; Hancock, Mark J. ; Tofts, Louise ; Epps, Adrienne ; Baldwin, Jennifer N. ; McKay, Marnee J. ; Burns, Joshua ; Morris, Eleanor ; Pacey, Verity. / Longitudinal fibular deficiency : a cross-sectional study comparing lower limb function of children and young people with that of unaffected peers. In: Children. 2019 ; Vol. 6, No. 3. pp. 1-11.
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abstract = "Longitudinal fibular deficiency (LFD), or fibular hemimelia, is congenital partial or complete absence of the fibula. We aimed to compare the lower limb function of children and young people with LFD to that of unaffected peers. A cross-sectional study of Australian children and young people with LFD, and of unaffected peers, was undertaken. Twenty-three (12 males) children and young people with LFD (74{\%} of those eligible) and 213 unaffected peers, all aged 7–21 years were subject to the Knee Osteoarthritis Outcome Score (KOOS/KOOS-Child) and the Cumberland Ankle Instability Tool (CAIT/CAIT-Youth). Linear regression models compared affected children and young people to unaffected peers. Participants with LFD scored lower in both outcomes (adjusted p < 0.05). The difference between participants with LFD and unaffected peers was significantly greater among younger participants than older participants for KOOS activities and sports domain scores (adjusted p ≤ 0.01). Differences in the other KOOS domains (pain/symptoms/quality of life) and ankle function (CAIT scores) were not affected by age (adjusted p ≥ 0.08). Children and young people with LFD on average report reduced lower limb function compared to unaffected peers. Knee-related activities and sports domains appear to be worse in younger children with LFD, and scores in these domains become closer to those of unaffected peers as they become older.",
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Longitudinal fibular deficiency : a cross-sectional study comparing lower limb function of children and young people with that of unaffected peers. / Pate, Joshua W.; Hancock, Mark J.; Tofts, Louise; Epps, Adrienne; Baldwin, Jennifer N.; McKay, Marnee J.; Burns, Joshua; Morris, Eleanor; Pacey, Verity.

In: Children, Vol. 6, No. 3, 45, 15.03.2019, p. 1-11.

Research output: Contribution to journalArticleResearchpeer-review

TY - JOUR

T1 - Longitudinal fibular deficiency

T2 - Children

AU - Pate, Joshua W.

AU - Hancock, Mark J.

AU - Tofts, Louise

AU - Epps, Adrienne

AU - Baldwin, Jennifer N.

AU - McKay, Marnee J.

AU - Burns, Joshua

AU - Morris, Eleanor

AU - Pacey, Verity

N1 - Copyright the Author(s) 2019. Version archived for private and non-commercial use with the permission of the author/s and according to publisher conditions. For further rights please contact the publisher.

PY - 2019/3/15

Y1 - 2019/3/15

N2 - Longitudinal fibular deficiency (LFD), or fibular hemimelia, is congenital partial or complete absence of the fibula. We aimed to compare the lower limb function of children and young people with LFD to that of unaffected peers. A cross-sectional study of Australian children and young people with LFD, and of unaffected peers, was undertaken. Twenty-three (12 males) children and young people with LFD (74% of those eligible) and 213 unaffected peers, all aged 7–21 years were subject to the Knee Osteoarthritis Outcome Score (KOOS/KOOS-Child) and the Cumberland Ankle Instability Tool (CAIT/CAIT-Youth). Linear regression models compared affected children and young people to unaffected peers. Participants with LFD scored lower in both outcomes (adjusted p < 0.05). The difference between participants with LFD and unaffected peers was significantly greater among younger participants than older participants for KOOS activities and sports domain scores (adjusted p ≤ 0.01). Differences in the other KOOS domains (pain/symptoms/quality of life) and ankle function (CAIT scores) were not affected by age (adjusted p ≥ 0.08). Children and young people with LFD on average report reduced lower limb function compared to unaffected peers. Knee-related activities and sports domains appear to be worse in younger children with LFD, and scores in these domains become closer to those of unaffected peers as they become older.

AB - Longitudinal fibular deficiency (LFD), or fibular hemimelia, is congenital partial or complete absence of the fibula. We aimed to compare the lower limb function of children and young people with LFD to that of unaffected peers. A cross-sectional study of Australian children and young people with LFD, and of unaffected peers, was undertaken. Twenty-three (12 males) children and young people with LFD (74% of those eligible) and 213 unaffected peers, all aged 7–21 years were subject to the Knee Osteoarthritis Outcome Score (KOOS/KOOS-Child) and the Cumberland Ankle Instability Tool (CAIT/CAIT-Youth). Linear regression models compared affected children and young people to unaffected peers. Participants with LFD scored lower in both outcomes (adjusted p < 0.05). The difference between participants with LFD and unaffected peers was significantly greater among younger participants than older participants for KOOS activities and sports domain scores (adjusted p ≤ 0.01). Differences in the other KOOS domains (pain/symptoms/quality of life) and ankle function (CAIT scores) were not affected by age (adjusted p ≥ 0.08). Children and young people with LFD on average report reduced lower limb function compared to unaffected peers. Knee-related activities and sports domains appear to be worse in younger children with LFD, and scores in these domains become closer to those of unaffected peers as they become older.

KW - longitudinal fibular deficiency

KW - lower limb function

KW - children

KW - young people

KW - unaffected peers

KW - KOOS

KW - KOOS-Child

KW - CAIT

KW - CAIT-Youth

UR - http://purl.org/au-research/grants/nhmrc/1031893

U2 - 10.3390/children6030045

DO - 10.3390/children6030045

M3 - Article

VL - 6

SP - 1

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JO - Children

JF - Children

SN - 2227-9067

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ER -