Peter H. Jongerius

Effect of Botulinum Toxin in the Treatment of Drooling: A Controlled Clinical Trial

Objective. To investigate the clinical effectiveness of botulinum neurotoxin type A (BoNT) to reduce drooling in children with cerebral palsy (CP). Methods. A controlled clinical trial was performed in which the results of single-dose BoNT injections in the submandibular glands were compared with treatment with scopolamine. Forty-five children who had CP and experienced severe drooling were enrolled. Drooling severity was measured at baseline, during application of scopolamine, and at different intervals after BoNT injections up to 24 weeks, using the Drooling Quotient (DQ), the Teacher Drooling Scale (TDS), and Visual Analog Scales (VAS). Results. Drooling was reduced during scopolamine application as well as after BoNT injections. Compared with baseline, the mean DQ showed a significant decrease throughout the study. Greatest reductions were achieved 2 to 8 weeks after BoNT injection. No significant differences were found between scopolamine measurements and those up to 24 months after BoNT injection. Using VAS, parents recorded the effect on drooling in which significant differences were found between baseline VAS score and all follow-up assessments. According to our definition of “success to therapy,” demanding a 2-point decrease on the TDS, 61.5% of patients responded to BoNT injections. Analysis of the DQ demonstrated a response rate of 53% of the patients to scopolamine and 48.7% to BoNT until 24 weeks after BoNT injections, the actual duration of this study. As a reaction to scopolamine, 71.1% of the patients had moderate to severe side effects. Only nonsevere, incidental side effects were reported after BoNT injections. Conclusions. During scopolamine application as well as after intraglandular BoNT injections, a clinically relevant reduction in drooling was achieved in children with CP, demonstrating maximum effect 2 to 8 weeks after injections. This is the first controlled clinical trial that confirmed a significant effect of BoNT injections in the treatment of drooling. General anesthesia was needed for all children. BoNT injections show fewer and less serious side effects than transdermal scopolamine treatment.

Effectiveness of modified constraint-induced movement therapy in children with unilateral spastic cerebral palsy: a randomized controlled trial.

Background. In children with unilateral spastic cerebral palsy (CP), there is only limited evidence for the effectiveness of modified constraint-induced movement therapy (mCIMT). Objective. To investigate whether 6 weeks of mCIMT followed by 2 weeks of bimanual task-specific training (mCIMT-BiT) in children with unilateral spastic CP improves the spontaneous use of the affected limb in both qualitative and quantitative terms more than usual care (UC) of the same duration. Methods. Children with unilateral spastic CP with Manual Ability Classification System (MACS) scores I, II, or III and aged 2.5 to 8 years were recruited and randomly allocated to either the mCIMT-BiT group (three 3-hour sessions per week: 6 weeks of mCIMT, followed by 2 weeks of task-specific training in goal-directed bimanual play and self-care activities) or to 1.5 hours of more general physical or occupational weekly plus encouragement to use the affected hand for the UC group. Primary outcome measures were the Assisting Hand Assessment and the ABILHAND-Kids. Secondary outcomes were the Melbourne Assessment of Unilateral Upper Limb Function, the Canadian Occupational Performance Measure, and the Goal Attainment Scale. Results. Twenty-eight children were allocated to mCIMT-BiT and 24 to UC. Except for the Melbourne, all primary and secondary outcome measures demonstrated significant improvements in the mCIMT-BiT group. Conclusion. mCIMT followed by task-specific training of goal-directed bimanual play and self-care activities is an effective intervention to improve the spontaneous use of the more affected upper limb in children with relatively good baseline upper extremity function.

The treatment of drooling by ultrasound-guided intraglandular injections of botulinum toxin type A into the salivary glands.

Objective The aim of the study was to present the background, procedure, and technique of bilateral ultrasound‐guided, single‐dose injections of botulinum toxin type A (BTX) into the salivary glands in patients with severe drooling.

Botulinum toxin A: a new option for treatment of drooling in children with cerebral palsy. Presentation of a case series.

Abstract Drooling beyond the age of 4 years is pathological, particularly if it occurs in children with neurological and developmental impairment and disability. Considering the therapeutic spectrum of botulinum toxin A and in view of the innervation of the salivary glands, we postulated that intraglandular injections into the submandibular glands with botulinum toxin A could reduce the secretion of saliva and consequently decrease drooling. Three patients with cerebral palsy and severe drooling were selected and evaluated over a 4-month period. Under ultrasound guidance, one dose of botulinum toxin A was injected bilaterally into the submandibular glands. Saliva secretion was measured at baseline and repeated four times during the following 4 months. In the three patients, maximal salivary flow rate of the sublingual and submandibular glands was reduced by 51% to 63%. The time of the maximal effect differed among the three children. The parents reported a satisfactory reduction of drooling throughout the whole study period. No objectionable disturbances of oral functions were observed. There was mild transient thickening of saliva in one of the patients. Conclusion The application of botulinum toxin A to the submandibular gland is a promising technique to reduce salivary flow rate and probably an alternative in the treatment of drooling in children with cerebral palsy.

A systematic review for evidence of efficacy of anticholinergic drugs to treat drooling.

Drooling frequently occurs in children with multiple handicaps; application of anticholinergic drugs is a potential strategy to treat drooling. A computer aided search of original studies concerning the treatment of drooling was carried out. The methodological and statistical integrity of the identified studies were assessed with previously defined criteria. The articles were weighed for their separate contribution to the evidence. The search resulted in 64 reports, of which seven studies passed the screening and were subjected to further assessment and discussion by three referees. Because of the small number of reports and the methodological restriction within the studies, no meta-analysis could be performed. No general conclusion could be made about the efficacy of anticholinergic drugs in treatment of drooling in children with multiple handicaps. There was some evidence that three anticholinergic drugs (benztropine, glycopyrrolate, and benzhexol hydrochloride) are effective in the treatment of drooling, but it could not be concluded that one drug is preferable.

Clinical practice: swallowing problems in cerebral palsy.

Cerebral palsy (CP) is the most common physical disability in early childhood. The worldwide prevalence of CP is approximately 2–2.5 per 1,000 live births. It has been clinically defined as a group of motor, cognitive, and perceptive impairments secondary to a non-progressive defect or lesion of the developing brain. Children with CP can have swallowing problems with severe drooling as one of the consequences. Malnutrition and recurrent aspiration pneumonia can increase the risk of morbidity and mortality. Early attention should be given to dysphagia and excessive drooling and their substantial contribution to the burden of a child with CP and his/her family. This review displays the important functional and anatomical issues related to swallowing problems in children with CP based on relevant literature and expert opinion. Furthermore, based on our experience, we describe a plan for approach of investigation and treatment of swallowing problems in cerebral palsy.

Social interaction and self-esteem of children with cerebral palsy after treatment for severe drooling

The impact of salivary flow reduction following medication (scopolamine and botulinum neurotoxin) on social interaction and emotional development (self-esteem) was evaluated in a group of 45 children with cerebral palsy who suffered from severe drooling. The children ranged in age from 3 to 16 years (median 9.1 years); 28 were male, 17 female. A questionnaire to document the impact of drooling on social interaction and self-esteem for both the children and their parents was developed and administered during the use of scopolamine and up to 24 weeks after intraglandular botulinum neurotoxin in the submandibular glands. The reduction of drooling was related to increased social contacts with peers. In addition, parents perceived that the impact of drooling on the level of the child’s satisfaction on physical appearance, relations within the extended family, and life in general increased. Although medication led to (temporary) positive changes, many social and emotional consequences remained unchanged. Conclusion: Interventions to treat drooling should not only be evaluated using measurements of drooling, but the consequences on social interaction and self-esteem should also be assessed.

Drooling in children with cerebral palsy : effect of salivary flow reduction on daily life and care

The purpose of this study was to investigate the effect of salivary flow reduction on daily life and provision of care in children with cerebral palsy (CP). Parents of children with CP were asked to fill in a questionnaire on the impact of drooling on the daily life of their children and their families and the data were then analyzed. Forty‐five children with severe drooling (28 males, 17 females; mean age 9y 5mo [SD 3y 7mo]; range 3 to 16y) were monitored before and after receiving medication (scopolamine and botulinum toxin) to reduce salivary flow. Type of CP included hypotonia (n=1), spastic paresis (n=27), and mixed motor disorders with spastic and dyskinetic paresis (n=17). Eight children were independently ambulant and 37 children were wheelchair users. Thirty‐four children had learning disability* with a developmental age of below 6 years. Six participants dropped out of the study; data on 39 children were analyzed. Results showed that anticholinergic agents effectively reduced salivary flow. Drooling diminished substantially and this was accompanied by a significant reduction in care needs, making daily care less demanding. The amount of reported damage to communication devices and computers decreased. In addition to the evaluation of primary variables, such as the salivary flow rate, investigation of impact of drooling on daily life provides useful information about the outcome of treatment for reduction in drooling.

Assessment of Salivary Flow Rate: Biologic Variation and Measure Error†

Objective: To investigate the applicability of the swab method in the measurement of salivary flow rate in multiple‐handicap drooling children. To quantify the measurement error of the procedure and the biologic variation in the population.

Behavioral Treatment of Drooling A Methodological Critique of the Literature With Clinical Guidelines and Suggestions for Future Research

Many children with mental retardation and developmental disabilities suffer from the consequences of chronic drooling. Behavioral treatment for drooling should be considered before other, more intrusive treatments such as medication and surgery are implemented. However, empirical studies on behavioral procedures are scarce. This article reviews 19 behavioral studies published since 1970. Treatment procedures are (a) instruction, prompting, and positive reinforcement; (b) negative social reinforcement and declarative procedures; (c) cueing techniques; and (d) self-management procedures. Although these procedures yield positive results, critical examination of experimental methodology of the studies reveals several methodological shortcomings. Guidelines for clinical use of behavioral treatment for drooling are presented, and recommendations are given for future research in this area.