Interdisciplinary assessment and treatment of paediatric drooling: two decades of experience by the Nijmegen saliva control team reflected in a stepwise algorithm.

Anterior and posterior drooling are prevalent comorbidities in children with neurodevelopmental disabilities. Considering the heterogeneity of the patient population and the multifactorial aetiology of drooling, an interdisciplinary and individualised treatment approach is indispensable. However, no tool for stepwise decision-making in the treatment of paediatric drooling has been developed previously. Within the Radboudumc Amalia Children's Hospital, care for children with anterior and/or posterior drooling secondary to neurodevelopmental disabilities is coordinated by a saliva control team with healthcare professionals from six disciplines. In alignment with international literature, published guidelines, and evidence gained from two decades of experience and research by our team, this paper proposes an algorithm reflecting the assessment and treatment approach applied in our clinic. First, directions are provided to decide on the necessity of saliva control treatment, taking type of drooling, the child's age, and the severity and impact of drooling into account. Second, the algorithm offers guidance on the choice between available treatment options, highlighting the importance of accounting for child characteristics and child and caregiver preferences in clinical (shared) decision-making. CONCLUSIONS: With this algorithm, we aim to emphasise the importance of repeated stepwise decision-making in the assessment and treatment of drooling in children during their childhood, encouraging healthcare professionals to apply a holistic approach. WHAT IS KNOWN: • Children with anterior or posterior drooling secondary to neurodevelopmental disabilities comprise a heterogeneous group, necessitating an individualised treatment approach. • No stepwise decision-making tool is available for the treatment of paediatric drooling. WHAT IS NEW: • Deciding on the necessity of saliva control treatment should be a conscious process, based on type of drooling, age, and drooling severity and impact. • Type of drooling, age, cognition, oral motor skills, self-awareness, posture, diagnosis, and child/caregiver preferences need to be considered to decide on the optimal treatment.

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.

The Non-speech Oral Movement Assessment Children (NOMAC).

INTRODUCTION: Examination of oral movements is often part of an assessment undertaken by a speech and language therapist (SLT). Until now there have been no specific instruments or tests with reference values for typically developing children in Dutch that exclusively evaluate non-speech oral movements in young children. Therefore, a non-speech oral-motor observation list was designed to attempt to bridge this gap: The Non-speech Oral Movement Assessment Children (NOMAC). The aim of this study was to evaluate the psychometric properties of the NOMAC in terms of inter-rater reliability and its' construct validity. In addition, we aimed to collect reference values for the non-speech oral movements in children. METHODS: Data from typically developing Dutch children aged 2 to 8 years were collected. Inter-rater reliability was studied by estimating the intra-class correlation coefficient (ICC). Construct validity was investigated by assessing the effect of age group and gender on the mean execution score per item (general linear model). To present normative data the percentage of the children performing a normal oral - motor execution was calculated. RESULTS: The study includes a total of 318 children, divided into 9 age groups. The inter-rater reliability shows a [sufficient] to [good] ICC for most items. A significant effect of the factor age group for almost all items was seen, confirming a robust construct validity. Normative data are presented with the percentage of the children performing a normal oral movement execution. CONCLUSION: Non-speech oral movements can be assessed with the NOMAC in children between 2 to 8 years old and can be compared with values obtained from a normative group. It should be used as part of a clinical feeding and speech assessment. Despite the fact that current insights indicate that oral- motor training has no value for improving mastication, swallowing and speech, it is important to know the status of non-speech oral motor capabilities. With this assessment a complete profile of the child's oral-motor abilities can be achieved, supportive for clinical decision making in SLT.

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.

Predicting non-response to intraglandular botulinum neurotoxin A injections for drooling in children with neurodevelopmental disabilities.

AIM: To develop robust multivariable prediction models for non-response to (1) submandibular botulinum neurotoxin A (BoNT-A) injections and (2) concurrent submandibular and parotid (four-gland) injections, to guide treatment decisions for drooling in children with neurodevelopmental disabilities, including cerebral palsy. METHOD: This was a retrospective cohort study including 262 children (155 males/107 females, median age 7 years 11 months [IQR 5 years 1 month], range 4 years 0 months - 17 years 11 months) receiving submandibular injections and 74 children (52 males/22 females, median age 7 years 7 months [IQR 4 years 3 months], range 4 years 9 months - 18 years 8 months) receiving four-gland injections. Multivariable logistic regression analyses were used to estimate associations between candidate predictors and non-response 8 weeks after injection. RESULTS: Ninety-six children (37%) were non-responders to submandibular injections, for which developmental age was the strongest predictor (adjusted odds ratio [aOR] 2.13; 95% confidence interval [CI] 1.02-4.45 for developmental age <4 years or 4-6 years with IQ <70). Other characteristics that showed a trend towards an increased risk of non-response were diagnosis, sex, and head position. Thirty-four children (46%) were non-responders to four-gland injections, for which tongue protrusion (aOR 3.10; 95% CI 1.14-8.43) seemed most predictive, whereas multiple preceding submandibular injections (aOR 0.34; 95% CI 0.10-1.16) showed a trend towards being protective. Predictors were, however, unstable across different definitions of non-response and both models (i.e. submandibular and four-gland) had insufficient discriminative ability. INTERPRETATION: Potential predictors of non-response to BoNT-A injections were identified. Nevertheless, the developed prediction models seemed inadequate for guidance of treatment decisions. WHAT THIS PAPER ADDS: Developmental age seemed most predictive of non-response to submandibular botulinum neurotoxin A injections. Non-response to concurrent submandibular and parotid injections was best predicted by tongue protrusion and number of previous injections. Multivariable prediction models including these clinical characteristics were unable to discriminate well. Predictors differed when non-response was defined using alternative outcome measures.

Long-term effects of submandibular gland excision on drooling in children with neurodevelopmental disorders: A cross-sectional study.

AIM: Submandibular gland excision (SMGE) is suitable for the management of drooling in patients with non-progressive neurodisabilities. We aimed to investigate the long-term effects of SMGE. METHOD: Patients who had SMGE between 2007 and 2018 were included. Main outcomes were a Visual Analogue Scale (VAS), Drooling Severity (DS), and Drooling Frequency (DF) collected at baseline, 8 weeks, 32 weeks and with a median of 313 weeks after SMGE (long-term). Secondary outcomes were satisfaction with the procedure, Drooling Quotient (DQ) and adverse events (AEs). RESULTS: We included thirty-five patients in the long-term analysis with a mean age of 14.5 years. A baseline VAS score of 80.4 was found, which improved on the long-term (mean difference -21.8, t(26) = 4.636, p < 0.0005)). DS and DF decreased significantly at the long-term compared to baseline (Z = -4.361, p < 0.0001 for DS, Z = -3.065, p = 0.002 for DF). Twenty-three out of 35 (66%) patients would recommend the procedure to peers. INTERPRETATION: This study indicates a long-term stable effect on drooling after SMGE in patients with anterior drooling. Recurrence of drooling occurs due to unknown reasons, nevertheless most caregivers and/or patients are still satisfied and would recommend the procedure to others.

Dysphagia limit in children with cerebral palsy aged 4 to 12 years.

AIM: To assess the dysphagia limit in children with cerebral palsy (CP) according to Eating and Drinking Ability Classification System (EDACS) level, sex, and age compared to typically developing children. METHOD: Seventy-seven children with CP (54 males, 23 females; mean age 7y 6mo, SD 2y 2mo, age range 4-12y) were assessed with the Maximum Volume Water Swallow Test. Median dysphagia limit in the CP group was compared with data of typically developing children. RESULTS: The dysphagia limit of children with CP differed significantly (p<0.001) from typically developing children. The latter showed a threefold higher median dysphagia limit (22mL) compared to children with CP in EDACS level I (7mL). The higher the EDACS level, the lower the dysphagia limit in children with CP. EDACS level explained 55% of the variance in the dysphagia limit of the CP group. INTERPRETATION: Where children with CP in EDACS levels IV and V showed that their capacity met the level of their performance, children in EDACS level I had the ability to perform a maximum capacity task, but still had a threefold lower median dysphagia limit than typically developing children. Establishment of the dysphagia limit should be part of general swallowing assessment in children with CP.

The medium to long-term effects of two-duct ligation for excessive drooling in neurodisabilities, a cross-sectional study.

OBJECTIVES: This study aims to evaluate the medium to long-term (1-12 years) effectiveness of two-duct ligation (2-DL) in patients with nonprogressive neurodisabilities. METHODS: Main outcomes included a Visual Analogue Scale (VAS), Drooling Severity (DS) and Drooling Frequency (DF), collected at baseline, 32 weeks postintervention and 1-12 years postintervention. Secondary outcomes were adverse events (AEs), and satisfaction. RESULTS: Forty-two patients were analyzed (mean age 17 years, mean baseline VAS 82). VAS decreased significantly from baseline to long-term (n = 30. Mean difference -36.5, CI -47.0 to -26.0, p ≤ 0.001; -26.1, CI -36.2 to -15.9, p ≤ 0.001). However, long-term VAS significantly increased compared to VAS at 32 weeks (+10.4, CI 1.0-19.8, p = 0.031). Out of 42 patients, 64% would recommend 2-DL to peers. CONCLUSION: There is a significant subjective 2-DL effect on drooling severity in the medium to long-term as reported by patients and caregivers, but there is also a certain degree of recurrence in this time span, and 33% of the patients required subsequent treatment. However, the majority of patients and/or caregivers would recommend 2-DL to peers.

Speech-language profiles in the context of cognitive and adaptive functioning in SATB2-associated syndrome.

SATB2-associated syndrome (SAS) is a neurodevelopmental disorder caused by heterozygous pathogenic variants in the SATB2 gene, and is typically characterized by intellectual disability and severely impaired communication skills. The goal of this study was to contribute to the understanding of speech and language impairments in SAS, in the context of general developmental skills and cognitive and adaptive functioning. We performed detailed oral motor, speech and language profiling in combination with neuropsychological assessments in 23 individuals with a molecularly confirmed SAS diagnosis: 11 primarily verbal individuals and 12 primarily nonverbal individuals, independent of their ages. All individuals had severe receptive language delays. For all verbal individuals, we were able to define underlying speech conditions. While childhood apraxia of speech was most prevalent, oral motor problems appeared frequent as well and were more present in the nonverbal group than in the verbal group. For seven individuals, age-appropriate Wechsler indices could be derived, showing that the level of intellectual functioning of these individuals varied from moderate-mild ID to mild ID-borderline intellectual functioning. Assessments of adaptive functioning with the Vineland Screener showed relatively high scores on the domain "daily functioning" and relatively low scores on the domain "communication" in most individuals. Altogether, this study provides a detailed delineation of oral motor, speech and language skills and neuropsychological functioning in individuals with SAS, and can provide families and caregivers with information to guide diagnosis, management and treatment approaches.

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.

Posterior drooling in children with cerebral palsy and other neurodevelopmental disorders.

AIM: To evaluate the effect of botulinum neurotoxin A (BoNT-A) injections, submandibular gland excision (SMGE), and bilateral submandibular duct ligation (2DL) for the control of posterior drooling in children with neurological impairment. METHOD: In a retrospective cohort, children with neurological impairment (e.g. cerebral palsy) treated between 2000 and 2016 were identified. Mean age at time of surgery was 9 years (range 1-21y). The primary outcome was posterior drooling severity by a visual analogue scale (VAS; 0-10) at baseline, 8-weeks, and 32-weeks follow-up. The secondary outcome was lower respiratory tract infections during the follow-up period. RESULTS: Ninety-two patients (out of 475; 47 males, 45 females) were identified. They were undergoing three different treatments: BoNT-A (n=63), SMGE (n=16), and 2DL (n=13). A significant reduction in VAS over time was observed in the total group of 92 patients. After SMGE, VAS decreased significantly from 6.82 (SD 3.40) at baseline to 2.29 (SD 1.93) at 8 weeks, and 2.17 (SD 2.58) at 32 weeks (F[2.34]=11.618, p<0.001). There was no significant decrease after both BoNT-A and 2-DL. INTERPRETATION: Posterior drooling is an unfamiliar, potentially life-threatening condition that is treatable with medication, BoNT-A injections, or surgery. Although all treatments reduced signs and symptoms of posterior drooling, there is a greater effect after SMGE compared to BoNT-A and 2-DL. What this paper adds Submandibular gland excision has better results for posterior drooling than botulinum toxin A or submandibular duct ligation.

Repeated onabotulinum neurotoxin A injections for drooling in children with neurodisability.

AIM: To evaluate the effect of repeated onabotulinum neurotoxin A injections for the treatment of drooling in children with neurodisabilities. METHOD: This was a retrospective cohort study, in which the first, second, and third onabotulinum neurotoxin A injection were compared within children treated between 2000 and 2020. Primary outcomes included drooling quotient, visual analogue scale (VAS), and treatment success defined as ≥50% reduction in drooling quotient and/or VAS 8 weeks after treatment. Each outcome was obtained at baseline and 8 weeks posttreatment. RESULTS: Seventy-seven children were included (mean age at first injection: 8y 3mo, SD 3y 7mo, range 3-17y; 44 males, 33 females; 51.9% with cerebral palsy, 45.5% wheelchair-bound). The objective (drooling quotient) and subjective (VAS) effect after the second injection was lower compared to the first injection. The third injection showed less objective and significantly less subjective effect compared to the first injection. An overall success rate of 74.0%, 41.6%, and 45.8% were found for the first, second, and third injection respectively. INTERPRETATION: Although onabotulinum neurotoxin A remained effective throughout the entire treatment course, there is less effect of subsequent onabotulinum neurotoxin A injections compared to the first. Although there might be a loss of effect after repeated injections, there is continued improvement for most children. What this paper adds Repeated injections show a diminished treatment effect after the second injection. A continued improvement is seen in most patients.

Submandibular duct ligation after botulinum neurotoxin A treatment of drooling in children with cerebral palsy.

AIM: To assess: (1) the effect on drooling of bilateral submandibular duct ligation as surgical therapy after the administration of submandibular botulinum neurotoxin A (BoNT-A) for excessive drooling and (2) the predictive value of treatment success with BoNT-A on treatment success after bilateral submandibular duct ligation. METHOD: This was a within-participant retrospective observational study in which 29 children with severe drooling (15 males, 14 females) received BoNT-A treatment at a mean age of 9 years 6 months (SD 2y 5mo), followed by bilateral submandibular duct ligation at a mean age of 10 years 11 months (SD 2y 4mo). Fifteen children were diagnosed with cerebral palsy (CP), with 12 children classified in Gross Motor Function Classification System levels IV and V. The 14 children without CP had non-progressive developmental disorders. The primary drooling severity outcomes were the Visual Analogue Scale (VAS; subjective assessment) and drooling quotient (objective assessment). Measurements were taken before each intervention and again at 8 and 32 weeks. RESULTS: The VAS was significantly lower after bilateral submandibular duct ligation at follow-up compared to BoNT-A treatment (mean difference -33, p≤0.001; 95% confidence interval [CI]=-43.3 to -22.9). The mean drooling quotient did not significantly differ between BoNT-A treatment and bilateral submandibular duct ligation at follow-up (3.3, p=0.457; 95% CI=-4.35 to 9.62) or between 8 and 32 weeks (4.7, p=0.188; 95% CI=-2.31 to 11.65). INTERPRETATION: BoNT-A treatment and bilateral submandibular duct ligation are both effective treatment modalities for drooling. At 32-week follow-up, subjective drooling severity after bilateral submandibular duct ligation was significantly lower compared to previous BoNT-A injections in participants. However, treatment success with BoNT-A is no precursor to achieving success with bilateral submandibular duct ligation. WHAT THIS PAPER ADDS: Bilateral submandibular duct ligation is an effective therapy for drooling after treatment with botulinum neurotoxin A (BoNT-A). Treatment success with BoNT-A is not a predictor of successful therapy with bilateral submandibular duct ligation.

An evaluation of predictors for success of two-duct ligation for drooling in neurodisabilities.

BACKGROUND: Drooling is dependent on various clinical variables. However, while drooling proves refractory to two-duct ligation in 40% of patients, predictors for treatment success are sparse and to date there is little evidence why some respond well while others are non-responders. We aim to find predictors for treatment success and study the effectiveness of two-duct ligation for drooling in neurodisabilities. METHODS: Fifty-four patients with moderate to severe drooling who had undergone two-duct ligation were screened for inclusion. Four patients were excluded due to missing or unreliable primary outcomes. The average age at the time of surgery was 12 years. Predictors were evaluated for treatment success which was defined as ≥ 50% visual analog scale for severity of drooling and/or drooling quotient reduction from baseline. Treatment effect was measured after 8 and 32 weeks compared to baseline. RESULTS: Age (more mature), adequate posture (no anteflexion), and normal speech are predictors for treatment success. Compared to baseline, drooling quotient was significantly lower at 8 (difference 18.6%, 95% confidence interval 12.3-24.9%) and 32 weeks (difference 10.1%, 95% confidence interval 3.9-16.4%). Compared to baseline, visual analog scale was significantly lower at 8 (difference 45.0, 95% confidence interval 37.0-52.9) and 32 weeks (difference 32.9, 95% confidence interval 25.0-40.7). CONCLUSIONS: Age, adequate posture, and a normal speech are predictors for treatment success, are easily determined pre-operatively, and help the clinician providing patient-specific probability of treatment success. There is a significant subjective and objective decrease of drooling after two-duct ligation.

Changes in severity and impact of drooling after submandibular gland botulinum neurotoxin A injections in children with neurodevelopmental disabilities.

AIMS: To examine changes in objective and subjective drooling severity measures after submandibular botulinum neurotoxin A injection in children with neurodevelopmental disabilities, explore their relationship, and evaluate if clinically relevant responses relate to changes in the impact of drooling. METHOD: This longitudinal, observational cohort study involved 160 children (92 males, 68 females; 3-17y, mean 9y 1mo, SD 3y 6mo) treated between 2000 and 2012 at the Radboud University Medical Center, Nijmegen, the Netherlands. Repeated measures analysis of variance was used to compare the 5-minute Drooling Quotient (DQ5) and Visual Analogue Scale (VAS) for drooling severity pretreatment and posttreatment, and Pearson's rho to assess their association. A parent questionnaire was used to assess drooling impact in responders (defined as ≥50% reduction in DQ5 and/or ≥2 SD reduction in VAS for drooling severity 8wks postintervention) and non-responders. RESULTS: One hundred and twelve children (70%) were responders. Their mean VAS for drooling severity and DQ5 scores were significantly lower 32 weeks postintervention compared to baseline. At baseline, the VAS for drooling severity-DQ5 relationship was 'weak' (rs =0.15, p=0.060), whereas it was 'fair' at 8 weeks (rs =0.43, p=0.000) and 32 weeks (rs =0.30, p=0.000). For responders, a significant change was found regarding the impact of drooling on daily care and social interactions at 8 weeks after intervention; most of these effects were maintained at 32 weeks. INTERPRETATION: A clinically relevant response based on a combination of objective and subjective measures of drooling severity was accompanied by positive changes regarding the impact of drooling on daily care and social interactions. WHAT THIS PAPER ADDS: Botulinum neurotoxin A injection into the submandibular glands reduced drooling severity in 70% of children with neurodevelopmental disabilities. Objective (5-minute Drooling Quotient) and subjective (Visual Analogue Scale for drooling severity) measures correlated 8 and 32 weeks after treatment. Objective and subjective measures complemented each other when changes in drooling severity were assessed. Reduced drooling severity was accompanied by positive changes with regard to the impact of drooling.

Unsuccessful submandibular duct surgery for anterior drooling: Surgical failure or parotid gland salivation?

OBJECTIVES: To evaluate if drooling recurrence after surgery of the submandibular ducts is due to surgical failure or other variables. METHODS: Historic cohort with prospective collected data of all patients with severe drooling who underwent unsuccessful submandibular duct surgery with subsequent re-intervention between 2003 and 2018. A reference cohort was used for comparison of clinical variables. RESULTS: Six males and 4 females were included (cerebral palsy n = 8, neurodevelopmental disorders n = 2). All patients underwent submandibular gland surgery as a primary intervention (duct ligation n = 8, submandibular duct relocation n = 2) followed by re-intervention (submandibular gland excision n = 7, parotid duct ligation n = 3). One patient underwent tertiary surgery (parotid duct ligation after re-intervention by submandibular gland excision). Three patients were successful after re-intervention. No difference was found between both re-intervention techniques. There was significantly more severe dental malocclusion (50% vs. 21%, P value = 0.047) and severe speech disorders (80% vs. 42%, P value = 0.042) in the current cohort when compared to the reference cohort. CONCLUSION: Recurrence of drooling surgery is most likely not caused by surgical failure of the primary intervention, because re-intervention (submandibular gland excision) did not lead to more success. Dysarthria and dental malocclusion might negatively influence treatment outcome.

Submandibular gland botulinum neurotoxin A injection for predicting the outcome of submandibular duct relocation in drooling: a retrospective cohort study.

AIM: This study evaluated whether the effect of submandibular gland botulinum neurotoxin A (BoNT-A) injection can predict the outcome of submandibular duct relocation with sublingual gland excision (SMDR) in children with drooling. Furthermore, we compared the effectiveness of both procedures. METHOD: A retrospective cohort study was performed in 42 children and adolescents (25 males, 17 females; mean [SD] age at BoNT-A injection 11y [4], range 4-20y; mean [SD] age at SMDR 15y [4], range 7-23y) with cerebral palsy or another non-progressive developmental disability who had undergone both BoNT-A injection and SMDR for drooling. Main outcomes were the drooling quotient and the visual analogue scale (VAS) on drooling severity at 8 weeks and 32 weeks follow-up. RESULTS: Failure or success of previous BoNT-A injections had no influence on success of consecutive SMDR. Relative change in main outcomes showed no significant relation between BoNT-A injection and SMDR for any follow-up measurement. After 8 weeks, SMDR was more successful than BoNT-A injection in diminishing VAS (VAS 80.0% vs 54.3%; drooling quotient 56.2% vs 51.0%). After 32 weeks, both drooling quotient (64.3% vs 29.5%) and VAS (75.7% vs 37.1%) showed significantly higher proportions of success for SMDR. INTERPRETATION: The effect of submandibular BoNT-A injection does not predict subsequent SMDR success in drooling. Furthermore, SMDR has a larger and longer-lasting positive effect on drooling than BoNT-A injections. WHAT THIS PAPER ADDS: Submandibular botulinum neurotoxin A (BoNT-A) injection effect does not predict submandibular duct relocation with sublingual gland excision outcome. Submandibular duct relocation is more effective and more permanent than BoNT-A injection.

INYECCIÓN DE NEUROTOXINA BOTULÍNICA A EN LA GLÁNDULA SUBMANDIBULAR PARA PREDECIR EL RESULTADO DE LA REUBICACIÓN DEL CONDUCTO SUBMANDIBULAR EN BABEO: UN ESTUDIO DE COHORTE RETROSPECTIVO: OBJETIVO: Este estudio evaluó si el efecto de la inyección de neurotoxina A botulínica submandibular (BoNT-A) puede predecir el resultado de la reubicación del conducto submandibular con escisión de la glándula sublingual (SMDR) en niños con babeo. Además, comparamos la efectividad de ambos procedimientos. MÉTODO: Se realizó un estudio de cohorte retrospectivo en 42 niños y adolescentes (25 varones, 17 mujeres; edad media [DE] en la inyección de BoNT-A 11 años [4], rango 4-20 años; edad media [SD] a SMDR 15 años [4] rango 7-23 años) con parálisis cerebral u otra discapacidad del desarrollo no progresiva que se haya sometido a una inyección de BoNT-A y SMDR para babear. Los resultados principales fueron el cociente de babeo y la escala analógica visual (VAS) en la severidad del babeo a las 8 semanas y 32 semanas de seguimiento. RESULTADOS: El fracaso o el éxito de las inyecciones anteriores de BoNT-A no tuvo influencia en el éxito de la SMDR consecutiva. El cambio relativo en los resultados principales no mostró una relación significativa entre la inyección de BoNT-A y SMDR para cualquier medición de seguimiento. Después de 8 semanas, SMDR fue más exitoso que la inyección de BoNT-A en la VAS disminuida (VAS 80,0% vs 54,3%; cociente de babeo 56,2% vs 51,0%). Después de 32 semanas, tanto el cociente de babeo (64,3% vs 29,5%) como el VAS (75,7% vs 37,1%) mostraron proporciones significativamente más altas de éxito para SMDR. INTERPRETACIÓN: El efecto de la inyección submandibular de BoNT-A no predice el éxito posterior de SMDR en el babeo. Además, el SMDR tiene un efecto positivo mayor y más duradero en el babeo que las inyecciones de BoNT-A.

INJEÇÃO DE NEUROTOXINA BOTULÍNICA NA GLÂNDULA SUBMANDIBULAR PARA PREDIÇÃO DO RESULTADO DA RELOCACÃO DO DUCTO SUBMANDIBULAR NA SIALORRÉIA: UM ESTUDO DE COORTE RETROSPECTIVO: OBJETIVO: Este estudo avaliou se o efeito da neurotoxina botulínica A (NTBo-A) na glândula submandibular pode predizer o resultado da relocação do ducto submandibular com excisão sublingual da glândula (RDSM) em crianças com sialorréia. Ainda, comparamos a efetividade de ambos os procedimentos. MÉTODO: Um estudo de coorte retrospectivo foi realizado em 42 crianças e adolescentes (25 do sexo masculino, 17 do sexo feminino; idade média [DP] no momento da injeção de NTBo-A 11a[4], variação de 4-20a; idade média [DP] no momento da RDSM 15a [4], variação de 7-23a) com paralisia cerebral ou outra desordem não-progressiva do desenvolvimento que passaram por injeção de NTBo-A e RDSM para sialorréia. Os principais desfechos foram o quociente de sialorréia e a escala visual análoga (EVA) sobre a severidade da sialorréia no acompanhamento de 8 e 32 semanas. RESULTADOS: A falha ou sucesso da NTBo-A prévia não teve influência no sucesso da RDSM consecutiva. A mudança relativa nos principais desfechos não mostrou nenhuma relação significativa entre a injeção de NTBo-A e a RDSM para nenhuma das medidas no acompanhamento. Após 8 semanas, a RDSM foi mais bem sucedida do que a NTBo-A na redução da EVA (EVA 80,0% vs 54,3%; quociente de sialorréia 56,2% vs 51,0%). Após 32 semanas, tanto o quociente de sialorréia (64,3% vs 29,5%) quanto a EVA (75,7% vs 37,1%) mostram proporções significativamente mais altas de sucesso para a RDSM. INTERPRETAÇÃO: O efeito da injeção submandibular de NTBo-A não prediz o sucesso da RDSM subsequente na sialorréia. Ainda, a RDSM tem efeito maior e mais duradouro na sialorréia do que injeções de NTBo-A.

Randomized controlled trial comparing botulinum vs surgery for drooling in neurodisabilities.

OBJECTIVE: To compare the effect of submandibular duct ligation (2-DL) and submandibular botulinum neurotoxin type A (BoNT-A) for drooling in children and adolescents with neurodevelopmental disabilities. METHODS: A randomized, interventional, controlled, and partly single-blinded study was performed in which submandibular BoNT-A was compared with 2-DL to treat excessive drooling. Main outcomes included a Visual Analog Scale (VAS), drooling quotient (DQ), drooling severity (DS) scale and drooling frequency (DF) scale. Each was obtained at baseline, and 8 and 32 weeks post treatment. RESULTS: Fifty-seven patients (mean age: 11 years, mean baseline VAS score 7.9, mean baseline DQ 27.3%) were randomized to the 2-DL or BoNT-A group. Four patients were excluded from analyses, leaving 53 patients for intention-to-treat analyses. Response to treatment, defined as a ≥50% reduction in DQ or VAS score, was higher for 2-DL after 32 weeks (63.0% vs 26.9%, p = 0.008). Both VAS score (24.5, p < 0.001) and DQ (-9.3%, p = 0.022) were significantly lower at follow-up after 2-DL vs BoNT-A. The total number of adverse events (p = 0.088, 40.7% vs 19.2%) and postoperative complaints was higher (p < 0.001, mean 9.6 vs 3.6 days) for 2-DL than for BoNT-A. CONCLUSION: The 2-DL procedure is a more effective treatment for drooling than botulinum toxin, but carries a slightly greater risk of complications and morbidity. TRIALREGISTERNL IDENTIFIER: NTR3537. CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that for children and adolescents with neurodevelopmental disabilities and severe drooling, 2-DL compared to a one-time intraglandular BoNT-A injection is more effective at reducing drooling at 32 weeks.

Effectiveness of submandibular duct relocation in 91 children with excessive drooling: A prospective cohort study.

OBJECTIVE: To evaluate the effectiveness of submandibular duct relocation (SMDR) in drooling children with neurological disorders. DESIGN: Prospective cohort study. SETTING: Academic Outpatient Saliva Control Clinic. PARTICIPANTS: Ninety-one children suffering from moderate to severe drooling. MAIN OUTCOME MEASURES: Direct observational drooling quotient (DQ; 0-100) and caretaker Visual Analogue Scale (VAS; 0-100). Secondary outcome measures were drooling severity (DS) and frequency rating scales. RESULTS: The DQ at baseline, 8 and 32 weeks postoperatively was 26.4, 12.3 and 10.8, respectively. VAS score decreased from 80.1 at baseline to 28.3 and 37.0 at 8 and 32 weeks after surgery. Median DS at baseline, 8 and 32 weeks was 5, 3 and 4, whereas the drooling frequency median scores were 4, 2 and 2, respectively. Five children required prolonged intubation due to transient floor of the mouth swelling, two of whom developed a ventilator-associated pneumonia. Another child developed atelectasis with postoperative pneumonia. Two more children needed tube feeding because of postoperative eating difficulties for 3 days or suprapubic catheterisation for urinary retention. Children aged 12 years or older (OR = 3.41; P = 0.03) and those with adequate stability and position of the head (OR = 2.84; P = 0.09) appeared to benefit most from treatment. CONCLUSIONS: Submandibular duct relocation combined with excision of the sublingual glands appears to be relatively safe and effective in diminishing visible drooling in children with neurological disorders, particularly in children aged 12 years and older and those without a forward head posture.

Reliability, construct validity and usability of the Eating and Drinking Ability Classification System (EDACS) among Dutch children with Cerebral Palsy.

PURPOSE: To assess the interrater reliability, construct validity and usability of the Eating and Drinking Ability Classification System (EDACS) among Dutch children with Cerebral Palsy (CP) when used by speech and language therapists (SLTs) familiar and unfamiliar with the child's eating and drinking performance and parents. METHODS: Translation was undertaken using the method of Eremenco. Agreement between SLTs and parents when using EDACS was determined by intraclass correlation coefficient (ICC) and linear weighted Kappa (κW). Associations with other functional classification systems including the Dysphagia Management Staging Scale (DMSS) were investigated to determine construct validity by Kendall's tau-b. RESULTS: Thirty-one SLTs classified 149 children (67 girls; mean 10 y, SD 4 y, range 3-21 y) with EDACS. Pairs of SLTs showed good agreement ([ICC] = 0.84; 95% confidence interval [CI] 0.79-0.88; [κW] = 0.71). Eighty-one parents showed good agreement with SLTs (n= 31) as well (ICC = 0.80; 95% CI 0.71-0.87; κW= 0.61). There was a significant and strong positive correlation of EDACS with DMSS (Kendall's tau-b 0.81) supporting its construct validity. Usability of EDACS was generally good. CONCLUSION: The Dutch version of EDACS is reliable and valid, and can be used easily by (familiar and unfamiliar) SLTs and parents of children with CP. Parents and professionals showed a high level of consistency when classifying eating and drinking abilities. EDACS enables uniform and efficient communication about safety and efficiency of functional eating and drinking ability in clinical and research contexts.