OTCohortAmerican journal on intellectual and developmental disabilities2018

The Emergence of Effortful Control in Young Boys With Fragile X Syndrome.

Marissa Robinson, Jessica Klusek, Michele D Poe and 2 others

PMID 29480774

WHAT IT FOUND

Young boys with fragile X showed lower effortful control than typically developing boys from 18 months, and did not improve over time.

Higher autism symptoms were linked to lower effortful control, but not to slower growth.

Key findings

01Boys with fragile X syndrome had lower effortful control than typically developing boys, and their effortful control did not increase over early childhood.

02Within the fragile X group, greater autism symptom severity was associated with lower effortful control, while adaptive behavior and FMRP were not.

03The difference between the fragile X and typically developing groups widened across the ages compared, and low effortful control emerged as young as 18 months.

STILL TO COME

How it was doneWhat they foundWhat it means for OTs

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What it does not show

The typically developing comparison group was small and had only 1 to 2 repeated observations, so the shape of growth over time could have been missed. Group comparisons were limited to ages 1.5 to 5.0 years, so the findings do not speak to effortful control outside that age window. The sample was primarily Caucasian, which may limit generalizability to more diverse groups. Effortful control was based on mother report, and maternal ratings may have been biased by knowing the child had fragile X. The study did not use experimental measures of effortful control, which could have provided a different estimate of the child's abilities. FMRP was estimated from lymphocytes and included only males with a limited range, so biological associations may be imprecise. The study included children younger than 6 years and therefore does not represent the full range of childhood.

Declared interests

The supplied publication types list NIH extramural and non-U.S. government research support. No author conflict-of-interest statement is provided in the supplied text.

The easy way to misread this

Do not read the flat effortful control profile as proof that boys with fragile X cannot improve with therapy. The study did not test treatment, and group comparisons were limited to ages 1.5 to 5.0 years.

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