Understanding the nature and consequences of intellectual and developmental disabilities is challenging, especially when the condition is rare, affected individuals are geographically dispersed, and/or resource constraints limit large-scale studies involving direct assessment. Surveys provide an alternative methodology for gathering information but must be carefully designed and interpreted in light of obvious limitations. In this paper we discuss the potential of surveys in understanding a disabling condition; delineate characteristics of successful survey research; describe a survey of families of individuals with fragile X syndrome; and synthesize major findings. The survey has provided new information about the nature and consequences of fragile X syndrome in a cost-effective fashion, suggesting that survey methodology has a useful place in creating new knowledge about intellectual and developmental disabilities.
"In addition to the standardized measures, survey respondents were asked to provide information regarding several areas of caregiving burden [Bailey et al., 2012b]. Many of these items were replicated from a previous survey of caregivers of individuals with FXS [Bailey et al., 2010]. Prior to data collection, we conducted cognitive testing to make sure the items were written clearly, the response options were not too broad or narrow, and the overall format of the survey was understandable. "
[Show abstract][Hide abstract] ABSTRACT: To identify the most common neurological and behavioral symptoms treated by medications in individuals with fragile X syndrome (FXS), factors associated with treatment variability, and difficulty in swallowing a pill.
A total of 1019 caregivers provided information about 1064 sons and 299 daughters with FXS in a US national survey. Caregivers reported (a) current use of medications for attention, anxiety, hyperactivity, mood swings, anger, depression, seizures, self-injury, or sleep; (b) perceived efficacy; and (c) difficulty in swallowing a pill.
Sixty-one percent of males and 38% of females were currently taking medication for at least 1 symptom. The most common symptoms were anxiety, attention, and hyperactivity. Treatments for attention and hyperactivity were common in childhood but declined substantially after the age of 18 years; anxiety treatment remained high in adults. Children perceived to be more impaired and children diagnosed or treated for autism were more likely to be taking medications. Caregivers considered most medications somewhat effective, but less than one-third rated current medication as "a lot" effective. Many children had difficulty swallowing a pill, but only 11% of adult males and 2% of adult females had a lot of difficulty.
Symptom-based medication use is common in FXS, although response is incomplete and there is clearly an unmet need for medications with improved efficacy. The persistent use of medications to treat anxiety, mood, and behavior problems throughout adolescence and into the adult years suggests important outcomes when evaluating the efficacy of new medications.
Journal of developmental and behavioral pediatrics: JDBP 11/2011; 33(1):62-9. DOI:10.1097/DBP.0b013e318236c0e1 · 2.13 Impact Factor
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