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.

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.

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.

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.

What could predict effectiveness of Botulinum Toxin to treat drooling: a search for evidence of discriminatory factors on the level of body functions or structures.

BACKGROUND: The treatment of drooling is important to families that experience the daily impact and research to elucidate clinical factors that play a role in the outcome of drooling treatment should be encouraged. AIM: To define clinical factors that influence therapy outcome of submandibular Botulinum Toxin (BoNT-A) injections for drooling. METHODS: Prospectively collected data of 128 children with cerebral palsy were evaluated; 80 spastic and 48 dyskinetic movement disorder, mostly Gross Motor Function Classification System III and higher; over 70% had an IQ <70. In addition, 23 fully ambulant children with exclusively intellectual disability were treated for drooling by ultrasound-guided injections of BoNT-A into the submandibular glands. Salivary flow rates and drooling quotients were measured at baseline and at 8 weeks after injection. Extensive information about the oral motor performance was gathered. Successful clinical response was defined as a 50% reduction of the baseline Drooling Quotient; 85 children were responsive to BoNT-A and 66 children unresponsive. RESULTS: Five nominated clinical factors that possibly could influence saliva reduction (head position, lip seal, voluntary control over the tongue, control of voluntary movement functions, and mental age) did not influence the responsiveness to BoNT-A. INTERPRETATION: Other variables need to be considered to predict the outcome of BoNT-A treatment. This article describes the first attempt to reveal the contribution of body functions and structures to the outcome of BoNT-A submandibular injections.

Does motor performance matter in botulinum toxin efficacy for drooling?

The aim of this study was to define factors that influence therapy outcome of submandibular botulinum toxin injections for drooling in children with cerebral palsy or mental disability. We postulated that differences in response may be explained by the variation of dysfunctions in the various cerebral palsy subtypes. Prospectively collected data were evaluated of 80 spastic and 48 dyskinetic children, of whom 70% had an IQ of <70. In addition, the data of 23 fully ambulant children with mental disability only were examined. Flow and Drooling Quotient were assessed at baseline and at 8 weeks after injection. After treatment, both the Drooling Quotient and submandibular flow decreased in all children. Morbidity associated with the procedure was limited. Ninety-three children responded to botulinum. Decrease of submandibular flow in these children was associated with reduction of parotid flow. In those who did not respond to therapy, spread across all 3 diagnostic classifications, parotid flow increased after injection. Response failure is characterized by increased parotid flow after injection; however, the precise role of parotid flow in therapy failure remains unclear. We cannot predict who will respond to botulinum toxin to treat drooling.

Efficacy and duration of botulinum toxin treatment for drooling in 131 children.

OBJECTIVE: To address the efficacy of botulinum toxin and the duration of its effect when used on a large scale for the treatment of drooling in children with neurological disorders. DESIGN: Prospective cohort study. SETTING: Academic multidisciplinary drooling clinic. PATIENTS: A total of 131 children diagnosed as having cerebral palsy or another nonprogressive neurological disorder and who also have moderate to severe drooling. INTERVENTION: Injection of botulinum toxin to the submandibular glands. MAIN OUTCOME MEASURES: Direct observational drooling quotient (DQ) (0-100) and caretaker visual analog scale (VAS) scores (0-100). RESULTS: A clinically notable response was found in 46.6% of children, reflected in a significant mean reduction in DQ from a baseline of 29 to 15 after 2 months and 19 after 8 months (P < .001). The mean VAS score decreased from 80 at baseline to 53 after 2 months and increased to 66 after 8 months (P < .001). Kaplan-Meier analysis showed that patients who initially responded to treatment experienced relapse after a median of 22 weeks (interquartile range, 20-33 weeks). CONCLUSIONS: Our study provides further support for botulinum toxin's efficacy for treatment of drooling in approximately half of patients for a median of 22 weeks. Further optimization of patient selection should be an area of attention in future studies.

Botulinum toxin versus submandibular duct relocation for severe drooling.

AIM: Botulinum neurotoxin type A (BoNT-A) has been described as an effective intervention for drooling and is being increasingly adopted. However, its effectiveness compared with established treatments is still unknown. We undertook a within-participants observational study to examine this. METHOD: An historic cohort was formed of 19 children and young adults (10 males, nine females) with severe drooling who underwent BoNT-A injections followed by surgical re-routing of the submandibular duct at least 6 months later. Mean age at time of admission was 11 years 5 months (range 5-17 y) and mean age at the time of surgery was 14 years (range 6-23 y). Fifteen children were diagnosed with bilateral cerebral palsy (CP), three with unilateral CP, and one with non-progressive developmental delay. Gross Motor Function Classification System levels were the following: level I, n=1; level II, n=2; level III, n=7; level IV, n=6; and level V, n=3). The primary outcome was the drooling quotient, which was assessed before each intervention and 8 and 32 weeks thereafter. A multivariate analysis of variance of repeated measures was performed, with the measurement points as the within-participant variables. RESULTS: The drooling quotient was reduced to a greater extent after surgery than after BoNT-A administration (p=0.001). Compared with a baseline value of 28, the mean drooling quotient 8 weeks after surgery was 10, and 32 weeks after surgery was 4 (p<0.001). Among the group treated with BoNT-A, the drooling quotient showed a significant reduction from a baseline value of 30 to 18 after 8 weeks (p=0.02), and a continued but diminished effect after 32 weeks (drooling quotient 22; p=0.05). INTERPRETATION: Both interventions are effective, but surgery provides a larger and longer-lasting effect.