Date of Award

January 2015

Document Type

Open Access Thesis

Degree Name

Master of Public Health (MPH)

Department

School of Public Health

First Advisor

Robert Dubrow

Abstract

Context: Treatment of X-linked hypophosphatemia (XLH) with active vitamin D metabolites and phosphate can partially correct skeletal deformities. It is unclear if therapy influences the occurrence of two major long-term morbidities in XLH: enthesopathy and dental disease.

Objective: To investigate the relationship between treatment and enthesopathy and dental disease in adult XLH patients.

Design: Observational and cross-sectional.

Setting: Academic medical center’s hospital research unit.

Participants: 52 XLH patients aged >= 18 at time of study.

Interventions: None

Main outcome measures: Number of enthesopathy sites identified by radiographic skeletal survey and dental disease severity (>5 or <=5 dental abscesses), identified historically.

Methods: Associations between proportion of adult life and total life with treatment and number of enthesopathy sites were assessed using multiple linear regression, while associations between these exposure variables and dental disease severity were assessed using multiple logistic regression. All models were adjusted for confounding factors.

Results: Neither proportion of adult nor total life with treatment was a significant predictor of extent of enthesopathy. In contrast, both of these treatment variables were significant predictors of dental disease severity (multivariate-adjusted global p-value =0.0080 and =0.0010 respectively). Participants treated 0% of adulthood were more likely to have severe dental disease than those treated 100% of adulthood (adjusted OR 25 [95% CI 1.2-520]). As proportion of total life with treatment increased, the odds of having severe dental disease decreased (multivariate-adjusted p-value for trend=0.015).

Conclusions: Treatment in adulthood may not promote or prevent enthesopathy; however it may be associated with lower risk of experiencing severe dental disease.

Comments

This is an Open Access Thesis.

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