The muscle-bone relationship in X-linked hypophosphatemic rickets

J Clin Endocrinol Metab. 2013 May;98(5):E990-5. doi: 10.1210/jc.2012-4146. Epub 2013 Mar 22.

Abstract

Context: We recently found that patients with X-linked hypophosphatemic rickets (XLH) have a muscle function deficit in the lower extremities. As muscle force and bone mass are usually closely related, we hypothesized that patients with XLH could also have a bone mass deficit in the lower extremities.

Objective: The study objective was to assess the muscle-bone relationship in the lower extremities of patients with XLH.

Setting: The study was carried out in the outpatients department of a pediatric orthopedic hospital.

Patients and other participants: Thirty individuals with XLH (6 to 60 y; 9 male patients) and 30 age- and gender-matched controls participated.

Main outcome measures: Calf muscle size and density as well as tibia bone mass and geometry were assessed by peripheral quantitative computed tomography. Muscle function was evaluated as peak force in the multiple 2-legged hopping test.

Results: Muscle force was significantly lower in XLH patients than in controls but muscle cross-sectional area did not differ (after adjustment for tibia length). External bone size, expressed as total bone cross-sectional area, was higher in the XLH group than in controls. The XLH cohort also had statistically significantly higher bone mineral content.

Conclusions: Patients with XLH have increased bone mass and size at the distal tibia despite muscle function deficits.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Bone Density
  • Bone Resorption / diagnostic imaging
  • Bone Resorption / etiology*
  • Bone and Bones / diagnostic imaging
  • Bone and Bones / pathology*
  • Child
  • Cohort Studies
  • Familial Hypophosphatemic Rickets / metabolism
  • Familial Hypophosphatemic Rickets / pathology
  • Familial Hypophosphatemic Rickets / physiopathology*
  • Female
  • Genetic Diseases, X-Linked*
  • Humans
  • Leg
  • Male
  • Middle Aged
  • Models, Biological
  • Muscle Development
  • Muscle Weakness / diagnosis
  • Muscle Weakness / diagnostic imaging
  • Muscle Weakness / etiology*
  • Muscle, Skeletal / diagnostic imaging
  • Muscle, Skeletal / pathology
  • Muscle, Skeletal / physiopathology*
  • Osteogenesis
  • Predictive Value of Tests
  • Tibia
  • Tomography, X-Ray Computed
  • Young Adult