P.H. Jongerius

Botulinum toxin effect on salivary flow rate in children with cerebral palsy

Objective: To investigate the effectiveness of botulinum neurotoxin (BoNT) type A in reducing salivary flow rate in children with cerebral palsy (CP) with severe drooling. Methods: During a controlled clinical trial, single-dose BoNT injections into the submandibular salivary glands were compared with scopolamine treatment. Forty-five school-aged children were included. Salivary flow rates from all major glands were obtained at baseline and compared with measurements during the interventions. Basic statistics consisted of analysis of difference scores. Results: Compared with baseline, the mean decrease in submandibular flow was 25% during scopolamine and 42% following BoNT injections. The difference scores were significant with maximum reductions 2, 4, and 8 weeks following BoNT. Of all children, 95% responded during scopolamine. Response rates for BoNT were significantly lower and varied from 69% at 2 weeks to 49% at 24 weeks after injection (the end of the study). Four patients discontinued scopolamine therapy because of side effects. Only incidentally mild side effects were reported from BoNT. Conclusions: Intraglandular BoNT injections significantly reduce salivary flow rate in the majority of drooling CP children, demonstrating high response rates up to 24 weeks. The procedure is simple to perform, effective, and safe when ultrasound guidance is used. The anticholinergic effect of BoNT exceeds that of scopolamine. As anticholinergic drugs are frequently contraindicated because of side effects, BoNT injections offer an alternative in the treatment of drooling.

The treatment of posterior drooling by botulinum toxin in a child with cerebral palsy.

Drooling is the unintentional loss of saliva from the mouth, known as anterior drooling (1). However, anterior drooling has to be distinguished from posterior drooling which refers to saliva that is spilled over the tongue through the faucial isthmus. Under physiological conditions this initiates the pharyngeal phase of swallowing, during which the larynx closes to protect the airways followed by relaxation of the upper esophageal sphincter. Whenever the trigger to swallow is impaired or missing, pooled saliva may lead to posterior drooling mostly apparent from an alarming congested breathing, coughing, gagging, vomiting, and at times aspiration into the trachea. Unrecognized and silent pneumonia can occur (2). The risk of posterior drooling can be enhanced by the fact that many disabled children are taken care of in a supine position for a substantial part of the day. Many children with cerebral palsy (CP) suffer from gastroesophageal reflux (3). In healthy subjects, exposure of the distal esophagus to acid results in an immediate increase of saliva secretion. The possible function of this is that swallowed saliva plays a role in the defense of esophageal mucosa to acid–induced injuries. Reflux in children with CP causes stimulation of pH-sensitive receptors in the mucosa of the distal esophagus which activates the esophageal-salivary reflex leading to an increase of salivary flow rate (3,4). This may exacerbate anterior and posterior drooling. Botulinum neurotoxin (BoNT) injections in the salivary glands in the treatment of anterior drooling demonstrate promising efficacy (5–8). This case report summarizes the results of repeated bilateral single-dose BoNT injections into the submandibular glands in a patient with CP with severe drooling, aspiration, and recurrent pneumonia. The primary treatment intention was to decrease salivary flow in an effort to reduce anterior as well as posterior drooling.

Self-management treatment of drooling: a case series.

Behavioral treatment of drooling is advocated widely, but evidence of its effectiveness is lacking. In a center-based case-series study, 10 participants with severe drooling were taught self-management skills to reduce drooling. Following treatment, all participants remained dry for intervals of 30-60 min, while being engaged in daily activities. Generalization to the classroom occurred in each participant. For three participants, maintenance of treatment effect was established at 6 and 24 weeks. Seven participants failed to maintain self-management skills at follow-up. Although the self-management procedure showed promising results, further adaptations are required to improve efficacy, generalization, and maintenance.

Negative effects of submandibular botulinum neurotoxin A injections on oral motor function in children with drooling due to central nervous system disorders

The aims of this study were: (1) to determine the incidence and nature of adverse effects on oral motor function after first injections of botulinum neurotoxin A (BoNT‐A) in submandibular glands for excessive drooling in children with central nervous system disorders; and (2) to identify independent predictors of these adverse effects.

Generalization and maintenance of a self-management program for drooling in children with neurodevelopmental disabilities: A second case series

ABSTRACT In this case series (n = 10) with a non-concurrent multiple baseline design, a self-management program was shown to be effective during inpatient training in eight participants with oral-motor problems and normal intelligence or mild intellectual disabilities. They were taught to perform a self-management routine and to remain dry for increasing time intervals. In addition, the program provided differential (self-) reinforcement of swallowing, controlling and wiping behavior, explicit formulation of motivational factors, instruction and feedback for parents and teachers, and continued practice after discharge. Generalization of the initial effect to the school setting was demonstrated in seven participants. Follow-up assessments demonstrated maintenance of positive results in four children up to 24 weeks, and for two other participants until 6 weeks after discharge. These results appeared to be relevant to parents and teachers. Teaching parents and teachers to implement the procedure enables them to support the child without professional supervision in the case of relapse.

Development of the Drooling Infants and Preschoolers Scale (DRIPS) and reference charts for monitoring saliva control in children aged 0–4 years

OBJECTIVESnTo develop and validate a parent questionnaire to quantify drooling severity and frequency in young children (the Drooling Infants and Preschoolers Scale - the DRIPS). To investigate development of saliva control in typically developing young children in the age of 0-4 years. To construct sex-specific reference charts presenting percentile curves for drooling plotted for age to monitor the development of saliva control in infancy and preschool age.nnnSTUDY DESIGNnThe DRIPS was developed consisting of 20 items to identify severity and frequency of drooling during meaningful daily activities. Factor analysis was performed to test construct validity. A piecewise logistic regression was followed by a piecewise linear regression to construct sex-specific reference charts.nnnRESULTSnWe obtained 652 completed questionnaires from parents of typically developing children. The factor analysis revealed four discriminating components: drooling during Activities, Feeding, Non nutritive sucking, and Sleep. To illustrate the development of saliva control, eight sex-specific reference curves were constructed to plot the scores of the DRIPS by age group, at the 15th, 50th, 85th and 97th percentile. About 3-15% of the preschoolers in our cohort did not acquire full saliva control at the age of 4 years.nnnCONCLUSIONSnWith the DRIPS it is possible to validly compare and visualize the development of saliva control in an individual infant or preschooler and allow clinicians to timely initiate individually targeted interventions if children outperform.