Understanding activity participation among individuals with Wolfram Syndrome.
Emily Bumpus, Tamara Hershey, Tasha Doty and 4 others
PMID 29861534WHAT IT FOUND
People with Wolfram syndrome reported a higher share of wanted or needed activities not done than comparison participants.
Adults reported walking, balance and vision loss as more limiting than diabetes.
Key findings
01Compared with people without Wolfram syndrome, children with Wolfram syndrome had a lower share of wanted or needed activities done and a higher share not done; adults with Wolfram syndrome had lower overall participation, a lower share of wanted or needed activities done, and a higher share not done.
02Among adults with Wolfram syndrome, vision loss and walking/balance problems were reported as limiting activities more than diabetes.
0310 participants with Wolfram syndrome, or 22% of the Wolfram syndrome sample, reported ever using occupational therapy services; outpatient and school settings were most common, and fine motor coordination and blindness were most often named, though diabetes management, depression, strength and other sensory issues were also addressed.
STILL TO COME
How it was doneWhat they foundWhat it means for PTsWhat it means for OTs
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What it does not show
The Wolfram syndrome survey sample was small, with a 37% response rate from the international registry. Only 20 participants with Wolfram syndrome had both survey and objective clinic data, which limited comparisons between measured symptoms and reported participation. The questionnaire modified existing occupational therapy measures for web use, and the authors say this may have made data less reliable or incomplete. Most participants were from the United States, so the results may not generalise to other healthcare systems, despite representation from 14 countries. The study is cross-sectional, so it cannot show that symptoms caused participation loss or that participation worsens as Wolfram syndrome progresses. The comparison group included people with type 1 diabetes, and the authors say further study is needed to conclude that Wolfram syndrome effects occur over and above diabetes.
Declared interests
The authors declared no conflict of interest.
The easy way to misread this
Do not conclude that occupational therapy improves activity participation in Wolfram syndrome. This was a cross-sectional questionnaire study, not a treatment trial, and only 10 participants with Wolfram syndrome reported ever using occupational therapy services.