The when, why, and how of using glycopyrronium to diminish drooling in children with neurodevelopmental disabilities: Implications for clinical practice.

BACKGROUND: Despite the established efficacy of glycopyrronium bromide in reducing drooling among children with neurodevelopmental disabilities, evidence on its impact on the daily lives of children and parents and effectiveness in a real-world setting are scarce, especially among long-term users. This study explored timing and duration of glycopyrronium treatment, effect and impact on daily life, and occurrence of side effects to inform clinical practice. METHODS: This was a retrospective cohort study at a national referral centre for drooling, including 61 children with nonprogressive neurodevelopmental disabilities, treated with glycopyrronium for anterior and/or posterior drooling between 2011 and 2021. Data were obtained from medical records and supplemented by structured telephone interviews with parents. RESULTS: Anterior drooling severity decreased in 82% of the included children. Changes in the impact of drooling on burden of care, social interaction, and self-esteem were reported in 55%, 31%, and 36%, respectively. Side effects were noted for 71% of cases, yet only 36% of parents deemed these as outweighing the positive impact of treatment. A substantial majority (77%) of the included children were long-term users (≥6 months). Among these, 38% of parents reported decreasing effectiveness and 27% noticed more prominent side effects over time. CONCLUSIONS: Glycopyrronium demonstrated potential in mitigating the impact of drooling on daily life, although variations were observed in the specific aspects and extent of improvement. The real-world context of our study provides important insights for refining clinical practices, emphasizing the need for balanced consideration of treatment benefits and potential side effects to facilitate shared decision-making.

Comparing the evidence for botulinum neurotoxin injections in paediatric anterior drooling: a scoping review.

Paediatric anterior drooling has a major impact on the daily lives of children and caregivers. Intraglandular botulinum neurotoxin type-A (BoNT-A) injections are considered an effective treatment to diminish drooling. However, there is no international consensus on which major salivary glands should be injected to obtain optimal treatment effect while minimizing the risk of side effects. This scoping review aimed to explore the evidence for submandibular BoNT-A injections and concurrent submandibular and parotid (i.e. four-gland) injections, respectively, and assess whether outcomes could be compared across studies to improve decision making regarding the optimal initial BoNT-A treatment approach for paediatric anterior drooling. PubMed, Embase, and Web of Science were searched to identify relevant studies (until October 1, 2023) on submandibular or four-gland BoNT-A injections for the treatment of anterior drooling in children with neurodevelopmental disabilities. Similarities and differences in treatment, patient, outcome, and follow-up characteristics were assessed. Twenty-eight papers were identified; 7 reporting on submandibular injections and 21 on four-gland injections. No major differences in treatment procedures or timing of follow-up were found. However, patient characteristics were poorly reported, there was great variety in outcome measurement, and the assessment of side effects was not clearly described.   Conclusion: This review highlights heterogeneity in outcome measures and patient population descriptors among studies on paediatric BoNT-A injections, limiting the ability to compare treatment effectiveness between submandibular and four-gland injections. These findings emphasize the need for more extensive and uniform reporting of patient characteristics and the implementation of a core outcome measurement set to allow for comparison of results between studies and facilitate the optimization of clinical practice guidelines. What is Known: • There is no international consensus on which salivary glands to initially inject with BoNT-A to treat paediatric drooling. What is New: • Concluding on the optimal initial BoNT-A treatment based on literature is currently infeasible. There is considerable heterogeneity in outcome measures used to quantify anterior drooling.and clinical characteristics of children treated with intraglandular BoNT-A are generally insufficiently reported. • Consensus-based sets of outcome measures and patient characteristics should be developed and implemented.

Surgery versus botulinum neurotoxin A to reduce drooling and improve daily life for children with neurodevelopmental disabilities: a randomized controlled trial.

AIM: To compare the effect of bilateral submandibular duct ligation and botulinum neurotoxin A (BoNT-A) on drooling severity and its impact on daily life and care in children and adolescents with moderate-to-severe drooling. METHOD: This was a randomized, interventional, controlled trial in which 53 children and adolescents (31 males, 22 females, mean age 11y, range 8-22y, SD 2y 10mo) with cerebral palsy (58.5%) or other non-progressive developmental disorders (41.5%) were randomized to BoNT-A (n=26) or bilateral submandibular duct ligation (n=27). A parent questionnaire on the severity of drooling in specific positions and daily activities and the impact of drooling on daily life and care was filled out at baseline and 8 and 32 weeks posttreatment. RESULTS: Both BoNT-A and bilateral submandibular duct ligation had a positive effect on daily care, damage to electronic equipment and/or furniture, social interactions, and self-esteem. However, bilateral submandibular duct ligation had a significant greater and longer-lasting short- (8wks) and medium-term (32wks) effect on daily care, reducing damage to electronic devices, and improving social interactions and satisfaction with life in general. INTERPRETATION: This randomized controlled trial confirms reduced drooling by both BoNT-A and bilateral submandibular duct ligation, but provides new evidence on improved well-being through a reduction in drooling. Even though there is a greater risk of complications and morbidity after bilateral submandibular duct ligation, compared to BoNT-A there was a significantly greater and longer-lasting positive effect on most outcomes. What this paper adds Bilateral botulinum neurotoxin A (BoNT-A) and submandibular duct ligation had a positive effect on the well-being of individuals with moderate-to-severe drooling. Bilateral submandibular duct ligation had a greater effect on the impact of drooling during daily care than BoNT-A. Bilateral submandibular duct ligation reduced damage to electronic devices and improved social interactions and satisfaction with life.

Therapy-related stress in parents of children with a physical disability: a specific concept within the construct of parental stress.

PURPOSE: The aim of this article is to conceptualise the phenomenon of therapy-related parental stress in parents of children with a physical disability. METHODS: Three models related to parental stress were reviewed, i.e., general parental stress, burden of caregiving in parents of children with physical disabilities, and experiences of these parents with their child's therapy. RESULTS: The proposed definition of therapy-related parental stress is "the subjective stress and subsequent changes of functioning and health experienced by a parent of a child with a physical disability in response to paramedical therapies (i.e., physical, occupational, and/or speech and language therapy)". A theoretical model is proposed to describe the process of therapy-related parental stress. Available questionnaires will most likely not be valid and responsive to capture the (changes in) stress parents experience related to therapy their child receives. CONCLUSIONS: This article provides a first definition of therapy-related parental stress and a theoretical model to visualise the processes with regard to this topic. Empirical testing of the presented components and their coherence is needed to confirm or improve the model. A questionnaire that specifically measures the concept of therapy-related parental stress is needed, along with evaluating therapy-related parental stress in clinical practice and research.Indications for rehabilitationIt is imperative to recognise paramedical therapies (i.e., physical, occupational, and speech and language therapy) as a potential stressor for parents of children with a physical disability, both in paediatric rehabilitation practice and research.A definition and theoretical model of therapy-related parental stress is proposed and the need for measures to evaluate this phenomenon is argued.The authors provide a starting point for the evidence base of the concept of therapy-related parental stress.

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

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.

Outpatient Treatment Based on Self-Management Strategies for Chronic Drooling in Two Children.

Drooling is a distressing condition, which is often caused by reduced oral motor control associated with a neurological disorder. It has significant medical, practical and psychosocial impact on children or youth and their families. Therefore, treatment is necessary. Although behavioural therapy for drooling shows promising results, it is generally time- and cost-intensive. For this reason, alternative ways to provide behavioural treatment for chronic drooling need to be explored. In a pair of case studies, the feasibility and potential of an outpatient variant of a behavioural treatment programme for drooling based on self-management strategies was researched with two children with oral motor difficulties. In a three week programme, these children were taught to perform a self-management routine in order to achieve saliva control during regular visits to the child rehabilitation centre. In addition, their parents and teachers were taught to prompt the self-management routine and instructed to provide additional practice at home and at school. In doing so, they were offered support by means of telehealth and personal contact. At the end of the treatment programme, both children showed a significant decrease in drooling severity. Their parents and teachers were satisfied with the treatment effect. Although the present treatment programme showed promising results, further adaptions are necessary to make the treatment programme more widely accessible.

The impact of submandibular duct relocation on drooling and the well-being of children with neurodevelopmental disabilities.

OBJECTIVE: The aim of this study was to evaluate the impact of a reduction in drooling after bilateral submandibular duct relocation (SMDR) with sublingual gland excision on daily life and care, as well as social and emotional consequences in children and adolescents with neurodevelopmental disabilities. METHODS: This prospective cohort study included 72 children and adolescents (46 males, 26 females) with moderate to severe drooling, and their caregivers. Mean age at the time of surgery was 15 years 2 months (SD 4y 3mo). Fifty-two children were diagnosed with cerebral palsy and 20 had other non-progressive developmental disabilities. A caregiver questionnaire to document the impact of drooling on daily care and economic consequences, social interaction and emotional development and self-esteem was administered before, and 8 and 32 weeks after surgery. RESULTS: Following bilateral SMDR the mean Visual Analogue Scale (VAS, 0-100) scores demonstrated a significant (p < 0.001) reduction in the severity of drooling from 81 at baseline to 28 and 36 after 8 and 32 weeks, respectively. This was accompanied by a decrease in the amount of daily care required and reduced economic consequences. In addition, an increase in social contact with other children and adults was reported by caregivers after surgery. CONCLUSION: Bilateral SMDR with sublingual gland excision provides a significant positive reduction in daily care of children suffering from drooling. Caregivers also report positive changes in their child's social interaction and sense of self-esteem.

Food preferences in young Dutch children and recommendations for feeding intervention in developmental disabilities.

Total and chronic food refusal (i.e., the refusal of all types of food during a prolonged period) in young children with developmental disabilities can be treated effectively using a combination of environmental interventions. However, no guidelines for the selection of food items to offer the child in these interventions are available. The aim of the present study was to assess the preferences for specific food items of young Dutch nondisabled children (N=254) in order to enable trainers to select food items that maximize success of feeding interventions. Results indicate that 54 out of 107 food items were found to be preferred. The mean appreciation scores of boys and girls did not differ significantly for these preferred food items, except for raisins and brown bread. Also, there were no differences between the distinguished age-groups, except for peach. Recommendations for the selection of food items within feeding intervention for total and chronic food refusal in young children with developmental disabilities are given.

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.

A descriptive analysis of studies on behavioural treatment of drooling (1970-2005).

A descriptive analysis was conducted on studies on the behavioural treatment of drooling (published between 1970 and 2005). The 17 articles that met the inclusion criteria described 53 participants (mean age 14y 7mo, [SD 4y 9mo]; range 6-28y). Sex of 87% of the participants was reported: 28 male, 18 female. For 60% of the participants the degree of learning disability was reported, varying from severe/profound (n=24, 75%), moderate (n=4, 13%), to mild (n=2, 6%), while two participants (6%) had no learning disabilities. Forty-two participants (79%) were diagnosed with cerebral palsy. Behavioural procedures included instruction, positive and negative reinforcement, overcorrection and restitution, verbal and automatic cueing, and/or self-management. Effective behavioural procedures are reported in children with and without learning disability and/or motor impairment. Even participants with profound learning disability may benefit from behavioural intervention. However, the evidence base in terms of number of studies in this area is limited. Fifteen studies used a single participant design; two studies implemented an experimental-comparison group design. Some of these studies were poorly designed and methodological flaws were identified. Therefore, conclusions about efficacy of behaviour therapy for drooling and/or best practice cannot be drawn, although our analysis suggests that this approach is promising. However, future research on this topic is needed. After years of research focused on medical treatment, the option of behavioural treatment to reduce drooling should be reconsidered in relation to the medical management of this problem.

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.

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

UNLABELLED: 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.