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Purpose <p>Accurate measurement is essential for tracking changes in clinical outcomes. Individuals with Smith-Magenis syndrome (SMS) exhibit challenging and unique food-related behaviors. We sought to determine the best tool to capture their unique food-related behaviors.</p> Methods <p>We conducted focus groups with caregivers of individuals with SMS to evaluate two commonly used questionnaires for food-related behaviors– the Food Related Problems Questionnaire (FRPQ) and the Hyperphagia Questionnaire for Clinical Trials (HQ-CT). Based on caregiver input and clinical expertise, we adapted these existing measures into a new tool: the SMS-FRPQ. We then validated this instrument for internal consistency and concurrent validity using online responses from 125 caregivers.</p> Results <p>Caregivers (<i>n =</i> 24) indicated neither the FRPQ or HQ-CT fully captured their child’s food-related behaviors; however, the newly developed SMS-FRPQ was deemed comprehensive by a new group of caregivers (<i>n =</i> 19). The SMS-FRPQ demonstrated strong internal consistency, with a Cronbach’s alpha of α = 0.87 [0.84–0.90]. Factor analysis indicated a three-factor model was a good fit (comparative fit index (CFI) = 0.9, Tucker-Lewis index (TLI) = 0.88). The three factors each showed good internal consistency: Desire for Food, α = 0.80 [0.74–0.85]; Takes Food, α = 0.87 [0.83–0.90]; Satiety Impairment, α = 0.83 [0.77–0.87]. The SMS-FRPQ showed higher internal reliability than the FRPQ. Concurrent validity was supported through alignment with similar items from the HQ-CT or Behavioral Problems Inventory (BPI-01).</p> Conclusions <p>The SMS-FRPQ has 14 items across 3 different factors and 5 additional clinically relevant items. This validated tool may be useful for tracking food-related behavior outcomes in clinical trials for this high-risk population.</p>

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Investigating food-related behaviors in Smith-Magenis syndrome: tailoring a questionnaire for a rare disease

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年4月29日

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  • Citrine Elatrash,
  • Theresa A. Wilson,
  • Alexis C. Wood,
  • Sarah H. Elsea,
  • Stephanie Sisley

摘要

Purpose

Accurate measurement is essential for tracking changes in clinical outcomes. Individuals with Smith-Magenis syndrome (SMS) exhibit challenging and unique food-related behaviors. We sought to determine the best tool to capture their unique food-related behaviors.

Methods

We conducted focus groups with caregivers of individuals with SMS to evaluate two commonly used questionnaires for food-related behaviors– the Food Related Problems Questionnaire (FRPQ) and the Hyperphagia Questionnaire for Clinical Trials (HQ-CT). Based on caregiver input and clinical expertise, we adapted these existing measures into a new tool: the SMS-FRPQ. We then validated this instrument for internal consistency and concurrent validity using online responses from 125 caregivers.

Results

Caregivers (n = 24) indicated neither the FRPQ or HQ-CT fully captured their child’s food-related behaviors; however, the newly developed SMS-FRPQ was deemed comprehensive by a new group of caregivers (n = 19). The SMS-FRPQ demonstrated strong internal consistency, with a Cronbach’s alpha of α = 0.87 [0.84–0.90]. Factor analysis indicated a three-factor model was a good fit (comparative fit index (CFI) = 0.9, Tucker-Lewis index (TLI) = 0.88). The three factors each showed good internal consistency: Desire for Food, α = 0.80 [0.74–0.85]; Takes Food, α = 0.87 [0.83–0.90]; Satiety Impairment, α = 0.83 [0.77–0.87]. The SMS-FRPQ showed higher internal reliability than the FRPQ. Concurrent validity was supported through alignment with similar items from the HQ-CT or Behavioral Problems Inventory (BPI-01).

Conclusions

The SMS-FRPQ has 14 items across 3 different factors and 5 additional clinically relevant items. This validated tool may be useful for tracking food-related behavior outcomes in clinical trials for this high-risk population.