Temper outbursts in Lowe syndrome: Characteristics, sequence, environmental context and comparison to Prader-Willi syndrome.
Helen Cressey, Chris Oliver, Hayley Crawford and 1 others
PMID 31144417WHAT IT FOUND
Caregivers of 17 men with Lowe syndrome described temper outbursts as frequent and severe, often triggered by unwanted demands or noise changes.
Outbursts typically involved aggression and ended with sudden calm or repair behaviours, differing from patterns seen in Prader-Willi syndrome.
Key findings
01Temper outbursts were a daily or near-daily occurrence for most participants, with physical and verbal aggression being the most common behaviours during an episode.
02The most common triggers were being asked to do something unwanted or changes in routine and expectation, with noise levels and tiredness also cited as increasing factors.
03Compared to Prader-Willi syndrome, Lowe syndrome outbursts were more likely to involve physical aggression and antisocial acts, and to end with a sudden return to normal activity as if nothing had happened.
STILL TO COME
How it was doneWhat they found
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What it does not show
The study relied entirely on caregiver reports, meaning the perspective of the individuals with Lowe syndrome was not directly captured. The sample size was very small (17 participants), which limits the statistical power and generalisability of the findings. The comparison with Prader-Willi syndrome was indirect, using data from a previous study rather than collecting new data from a matched Prader-Willi group at the same time. The interview schedule was originally designed for Prader-Willi syndrome, which may have biased the questions towards factors relevant to that condition rather than Lowe syndrome.
Declared interests
The study was funded by The Lowe Syndrome Trust, UK.
The easy way to misread this
Do not interpret the described management strategies as evidence-based interventions. The success rates reported by caregivers were subjective estimates, and the study did not test whether these strategies actually reduced outburst frequency or severity in a controlled way.