Toilet training in individuals with Angelman syndrome: A case series

Abstract

Objective: To assess if adapted versions of the response restriction toilet training protocol, based on the behavioral phenotype of Angelman syndrome (AS), were successful in fostering urinary continence in seven individuals with AS.

Method: Data were collected in AB-designs during baseline, training, generalization and follow-up. The response restriction protocol was adapted: individuals were trained in their natural environment, were prompted to void and along with improving continence, the interval between voids was prolonged and time-on-toilet decreased.

Results: During generalization five individuals had less than two accidents and one to six correct voids per day; during baseline more accidents and/or less correct voids occurred. In two participants correct voids increased, but several accidents still occurred. Three participants maintained positive results after 3–18 months.

Conclusion: Despite their intellectual and behavioral challenges, urinary continence can be acquired in AS. Several indications of voiding dysfunctions were found; further research is indicated.

• Angelman syndrome
• response restriction
• toilet training

Acknowledgments

The authors would like to thank participants, parents and caretakers for participating in this research. Gratitude goes out to Louke Peters, Mieke van der Veen and Elles Volker for their assistance during toilet training. Finally, the authors wish to thank Marije Averink and Eef Rasing for their advice.