Comparing botulinum toxin and 4-duct ligation for Sialorrhea in children - A systematic review.

INTRODUCTION: Sialorrhea or drooling can result in physical and psychosocial complications, such as aspiration and social isolation. Treatment options include botulinum toxin into the salivary glands and 4-duct ligation (i.e., simultaneous ligation of the bilateral parotid and submandibular ducts). This systematic review aimed to compare the efficacy and complication rates of botulinum toxin and 4-duct ligation for the treatment of drooling in children. METHODS: Following PRISMA guidelines, PubMed, Embase, Web of Science, and Cochrane Library were searched from inception through June 17, 2021 for studies examining the efficacy of botulinum toxin or 4-duct ligation for drooling in children. Data were summarized by pooled counts, percentages, and means. Complication rates were compared by a chi-squared test. RESULTS: A total of 22 studies (n = 606) examining botulinum toxin and 5 studies (n = 124) examining 4-duct ligation were included. From 12 botulinum toxin studies (n = 211), mean drooling frequency and severity scores was 7.5 at baseline. Mean difference from baseline was -2.6 (n = 92) at 4 weeks follow-up, -2.1 at 8 weeks (n = 41), -2.1 at 12 weeks (n = 56), and - 2.1 at 16 weeks (n = 58). From 4 4-duct ligation studies (n = 103), mean baseline drooling frequency and severity score was 8.4. Mean difference was -3.7 at mean follow-up of 35.6 months (n = 103). Eighteen botulinum studies (n = 343) recorded 53 (15.5 %) complications, including thickened saliva (n = 9), dysphagia (n = 4), and cheek abscesses (n = 4). Four 4-duct ligation studies (n = 108) recorded 25 (23.1 %) complications, including parotid gland swelling (n = 4), aspiration pneumonia (n = 3), and oxygen desaturation (n = 3). There was no statistically significant difference in complication rates between botulinum toxin and four-duct ligation (p = 0.065). CONCLUSION: Botulinum toxin injection and 4-duct ligation are both effective in improving sialorrhea in children and have comparable complication rates.

Pulmonary and Clinical Outcomes After Bilateral Submandibular Gland Excision and Parotid Duct Ligation for Refractory Sialorrhea.

Importance: Refractory sialorrhea in children can result in pulmonary aspiration and irreversible lung damage. Despite many studies devoted to the surgical treatment of sialorrhea, there is a paucity of objective outcome measures after surgery, especially with regard to pulmonary health. Objectives: To assess whether bilateral submandibular gland excision and bilateral parotid duct ligation ("DROOL" procedure) is associated with reduced pulmonary inflammation in bronchoalveolar lavage (BAL) samples after surgery and to assess patient factors associated with improvement after surgery. Design, Setting, and Participants: This retrospective case series included all 112 patients undergoing the DROOL procedure at a single tertiary care pediatric children's hospital from January 1, 2012, to December 31, 2021. Statistical analysis was performed from March 30 to June 10, 2023, and August 20 to September 23, 2023. Exposure: DROOL procedure for refractory sialorrhea. Main Outcomes and Measures: Degree of pulmonary inflammation (neutrophil percentage) according to BAL cytologic findings and overall bronchoscopy findings up to 12 months before and after the DROOL procedure. Secondary outcomes included number of annual hospitalizations, caregiver report of function before and after the procedure, and need for revision procedures and/or additional operations for secretion management. Results: A total of 112 patients (median age, 3.4 years [IQR, 2.0-7.1 years]; 65 boys [58.0%]) underwent DROOL procedures and had both preoperative and postoperative BAL samples during the study period. Patients demonstrated objective improvement in pulmonary inflammation after surgery, with the median polymorphonuclear neutrophil percentage decreasing from 65.0% (IQR, 14.0%-86.0%) before the surgery to 32.5% (IQR, 3.0%-76.5%) after the surgery (median difference in percentage points, -9.0 [95% CI, -20.0 to 0.0]). Prior to the DROOL procedure, 34 patients (30.4%; 95% CI, 21.8%-38.9%) were hospitalized 2 or more times annually for respiratory illness, which decreased to 10.1% (11 of 109; 95% CI, 4.4%-15.7%) after surgery (3 patients did not have hospitalization data available following surgery). Most caretakers (73 [65.2%]) reported improved secretion management after the procedure. Conclusions and Relevance: This study suggests that patients with impaired secretion management who underwent a DROOL procedure demonstrated improvement in pulmonary inflammation and a reduction in hospitalizations after surgery. Caretakers were also likely to report subjective improvement in secretion management and quality of life. Additional research is necessary to guide optimal timing and patient selection for this procedure.

[Clinical effect of free posterior interosseous artery perforator flap carrying superficial vein for reconstructing severe perioral scar hyperplasia and contracture].

Objective: To observe the clinical effect of free posterior interosseous artery perforator flap carrying superficial vein for reconstructing severe perioral scar hyperplasia and contracture. Methods: The retrospective observational study method was used. From August 2019 to March 2023, 11 patients with severe perioral scar hyperplasia and contracture after severe facial burns who met the inclusion criteria were admitted to General Hospital of TISCO (the Sixth Hospital of Shanxi Medical University). All patients were male and aged 23 to 56 years, with an average age of 31.3 years. After the perioral scar was removed and released, the wound area was 3.0 cm×2.0 cm to 10.5 cm×2.0 cm. The free posterior interosseous artery perforator flap carrying superficial vein was used to repair the wound, and the flap incision area was 3.5 cm×2.5 cm to 11.0 cm×2.5 cm. Among them, 6 patients required repair of wounds after resecting scar around ipsilateral upper and lower lips, and the lobular treatment of the flap was conducted. The wound in the flap donor area was directly sutured. After surgery, the survival of the flap and the occurrence of vascular crisis were observed. During follow-up after surgery, the appearance, texture, and color of the flap, the appearance of the flap donor area, and improvements of crooked mouth, drooling, limited mouth opening, and lip valgus in patients were observed. Results: All the flaps in patients completely survived after surgery, with no occurrence of vascular crisis. During follow-up of 6 to 36 months after surgery, the flap was not significantly bloated, was soft in texture, and had a similar color to that of the normal facial skin. Only linear scars were left in the flap donor area, and crooked mouth, drooling, limited mouth opening, and lip valgus in patients were significantly improved. Conclusions: The free posterior interosseous artery perforator flap carrying superficial vein can reconstruct severe perioral scar hyperplasia and contracture, with low incidence of postoperative flap vascular crisis, and the lobular treatment of flaps can repair wounds around unilateral upper and lower lips at the same time. After surgery, the appearance and function of the perioral area are significantly improved. The flap is a good choice for repairing small area of severe perioral scar hyperplasia and contracture.

Cirugía de desfuncionalización de glándulas salivales mayores en sialorrea masiva / Major salivary gland defunctionalization surgery in massive sialorrhea

Introducción: La sialorrea es la pérdida involuntaria de saliva de la boca, ya sea debido a la producción excesiva de saliva o disminución de la frecuencia de deglución. Se habla de sialorrea patológica cuando persiste más allá de los 4 años de edad. Además de las implicaciones sociales, cambios de ropa frecuentes, puede provocar neumonías por aspiración y deshidratación. El manejo de la sialorrea requiere una evaluación completa con un enfoque de equipo multidisciplinario para el tratamiento, que incluye terapias no farmacológicas, farmacológicas y quirúrgicas. Objetivo: Presentar resultados quirúrgicos y farmacológicos en el tratamiento de sialorrea masiva. Material y Método: Se realizó revisión de historias clínicas de 7 pacientes portadores de sialorrea masiva. Todos los pacientes incluidos fueron refractarios a tratamiento médico. El diagnóstico fue obtenido por un equipo multidisciplinario. Se les realizó desfuncionalización quirúrgica y farmacológica de glándulas salivales. Se les aplicó Escala de Severidad (DSS) y escala de frecuencia (DFS), previo a cirugía y posterior a procedimiento hasta el año. Resultados: Mejoría clínica subjetiva posterior a desfuncionalización quirúrgica con disminución de DSS y DFS. Disminución promedio de baberos a 10/día. Conclusión: Los resultados obtenidos son buenos, si se consideran las escalas DSS, DFS y el número de baberos al día, que son mediciones tanto subjetivas y objetivas respectivamente.

Introduction: Massive Sialorrhea is the involuntary loss of saliva from the mouth, either due to excessive saliva production or decreased swallowing frequency. We speak of pathological sialorrhea when it persists beyond 4 years old. In addition to the social implications and frequent clothing changes. It can cause aspiration pneumonia and dehydration. Treatment for sialorrhea requires a comprehensive evaluation with a multidisciplinary team approach. Including non-pharmacological, pharmacological, and surgical therapies. Aim: Presentation of the results of surgical defunctionalization of the salivary glands plus injection of Botulinum Toxin in the treatment of massive sialorrhea. Material and Method: A review of the clinical records of 7 patients with massive sialorrhea was carried out. All included patients were refractory to medical treatment. The diagnosis was obtained by a multidisciplinary team. Surgical and pharmacological dysfunctionalization of salivary glands was performed. Severity Scale (DSS) and Frequency Scale (DFS) were applied before surgery and after the procedure up to a year. Results: Subjective clinical improvement after surgical defunctionalization with decreased SHD and DFS. Average decrease in bibs to 10/day. Conclusion: The evaluated strategy presented similar benefits with respect to the literature. The SHD and DFS scales and the number of bibs per day are both subjective and objective measurements, respectively, and allow the clinical improvement and quality of life of patients undergoing surgery to be evaluated individually.

Comprehensive management of anterior drooling: An International Pediatric Otolaryngology Group (IPOG) consensus statement.

OBJECTIVE: To provide guidance for the comprehensive management of children referred for anterior drooling. The mission of the International Pediatric Otolaryngology Group (IPOG) is to develop expertise-based recommendations for the management of pediatric otolaryngologic disorders with the goal of improving patient care. METHODS: Survey of expert opinion by the members of the International Pediatric Otolaryngology Group (IPOG). The recommendations are derived from current expert consensus and critical review of the literature. RESULTS: Consensus recommendations include initial care and approach recommendations for health care providers who commonly evaluate children with drooling. This includes evaluation and treatment considerations for commonly debated issues in drooling management, initial work-up of children referred for anterior drooling, treatment recommendations, indications and contra-indications for rehabilitation, medical, and surgical management, as well as pros and cons of different surgical procedures in the hands of drooling management experts. CONCLUSION: Anterior drooling consensus recommendations are aimed at improving patient-centered care in children referred for sialorrhea.

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.

Salivary gland surgery and nonviral respiratory-related hospitalizations in children with neurodevelopmental impairment.

OBJECTIVES: Neurodevelopmentally impaired (NI) children with chronic sialorrhea are at elevated risk for aspiration and respiratory tract infections. Direct resection or ligation ("DROOL") of the submandibular glands (SMG) with parotid duct ligation are surgical interventions intended to decrease salivary output. The objective of this study is to determine the impact of DROOL surgery on the incidence of nonviral respiratory-related (NVR) post-procedure hospital encounters including emergency department visits and admissions. METHODS: Retrospective case series of NVR related outcomes after DROOL surgery in children performed at a single institution, tertiary referral center. RESULTS: A total of 35 gastrostomy tube-dependent patients (60% male, average age 8.2 [SD 6.0] years) with NI underwent DROOL surgery (86% SMG excision). Pre- and post-surgical follow-up time was 3.6 and 3.2 years, respectively. Presurgical and postsurgical NVR hospital encounters occurred in 28 (80%) and 14 (40%) patients, respectively (p < 0.01). Mean (SD) postoperative NVR hospital encounters occurred less frequently when compared to presurgical period (0.4 [0.6] vs. 1.0 [1.2] per year, p < 0.01) with average change of -0.7 encounters per year (SD 1.4, 95% CI -1.0 to -0.2). Patients with encounters within a year preceding DROOL (OR 4.9, p = 0.04, 95% CI 1.1-22.8), or those with at least 3 preoperative encounters (OR 8.0, p = 0.01, 95% CI 1.6-40.3) were significantly associated with a postsurgical NVR event. Fewer patients used anti-sialorrhea medication postoperatively compared to preoperatively (60% vs. 17%, p < 0.01). No patient developed surgical site complications requiring operative interventions. CONCLUSIONS: DROOL surgery for chronic sialorrhea in patients with NI was associated with decreased hospitalization and ED visits for NVR respiratory events post-procedurally. Sialorrhea may be an actionable source of recurrent respiratory illnesses requiring hospitalizations.

Effectiveness and safety of botulinum toxin in comparison with surgery for drooling in paediatric patients with neurological disorders: a systematic review.

Different therapeutic methods for chronic drooling in paediatric patients with neurological problems have been described in the scientific literature. However, there is no consensus on the ideal strategy of treatment. The aim of this study was to compare botulinum toxin injection therapy and surgical modalities to control drooling in paediatric patients with neurological disorders. A systematic literature search was conducted on nine electronic databases for publications until April 2020. Six articles were included with a total sample of 209 patients, 67.4% (n = 141) of whom had cerebral palsy. All studies used injections of botulinum toxin type A with application to the submandibular and/or parotid salivary glands. The surgical treatments were duct ligation in the parotid and/or submandibular salivary glands, duct relocation in the submandibular salivary glands, and glandular excision of the submandibular and sublingual salivary glands. There were complications in only 16.1% (n = 27) of the sample (11 cases due to botulinum toxin application and 16 due to surgery). Drooling control was assessed by objective and subjective measures. Although surgical procedures presented a higher risk of adverse effects than botulinum toxin type A in all the studies and measurements performed, they presented larger and longer-lasting positive effects on drooling. We suggest bilateral submandibular duct relocation with bilateral sublingual gland excision or isolated bilateral submandibular duct ligation, which were the surgical techniques with the largest samples in this review. Nevertheless, further studies are necessary to compare samples with botulinum toxin type A and surgical treatment.

The Use of Vastus Lateralis Muscle Flap for Functional Lip Reconstruction.

BACKGROUND: Total or near-total lip defects poses a serious challenge to a reconstructive surgeon with static procedures not providing desired functional and aesthetic outcomes. The dynamic lip reconstructive methods using functional muscles have become a current issue in recent years showing admirable results. This study present 3 cases of successful total and near-total lip reconstruction are presented using vastus lateralis muscle. METHODS: This is a retrospective analysis of a consecutive series of 3 patients (2 men and 1 woman) aged 44 to 56 years (mean) who had resection of extensive squamous cell carcinoma 2 had both upper and lower lip involved and 1 had lower lip involvement only. After resection patients had near total full thickness lower lip defect and 40% upper lip defect. The innervated vastus lateralis muscle free flap was transferred to the lip and end-to-end vascular anastomosis on the facial artery and end-to-side to internal jaguar vein was performed. The marginal mandibular branch of the facial nerve was used for nerve coaptation. The inner and outer surfaces of the flaps were grafted with a thick-split-thickness skin graft. Drooling rating scale and patient and observer scar assessment scale as well as electromyography were performed to evaluate oral competency and aesthetic outcome. RESULTS: All patients underwent single stage near-total lower lip and 2 had part of upper lip reconstruction successfully and survived the surgical operation. One patient lost the skin graft and was managed consecutively and muscle granulated and healed. Two patients underwent radiation therapy and 1 died before starting radiation due to other unnatural causes. The 2 patients achieved perfect oral sphincter competence without drooling and at 9months postoperative demonstrated successful reinnervation of the vastus lateralis muscle. CONCLUSIONS: This study demonstrates that lip reconstruction using an innervated vastus lateralis muscle free flap is a reliable method, providing a functional lip.

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.

US-guided percutaneous microwave ablation (MWA) of submandibular gland: A new minimal invasive and effective treatment for refractory sialorrhea and treatment response evaluation with contrast-enhanced imaging techniques.

A 33 years' old male complained of excessive salivation with frequent swallowing and spitting, which resulted in communication disturbance, reduced quality of life, and social embarrassment for 19 years. He had been diagnosed as sialorrhea and submandibular gland hyperfunction by stomatologist, then had unilateral submandibular gland resection 13 years ago, but the symptom relief was not satisfactory. After that, he had been treated with glycopyrrolate for less than a year, which was withdrawn because of the short duration of symptomatic control after each tablet take-in and intolerable side effects. With the wish to receive a new treatment with long term effectiveness, low re-operation risk and normal preserved saliva secretion function, the patient was subject to MWA for the right submandibular gland. After systematic clinical evaluation, US-guided percutaneous MWA was successfully performed with an uneventful post-operative course. The volume of the right submandibular gland and ablated area were measured precisely by an ablation planning software system with automatic volume measurement function based on three-dimensional reconstruction of the pre-operative and post-operative enhanced magnetic resonance imaging (MRI) raw data. Finally, the ablated volume was calculated as 62.2% of the whole right submandibular gland. The patient was discharged 1 day after the operation, with symptoms relieved significantly, the mean value of whole saliva flow rate (SFR) decreased from 11 ml to 7.5 ml per 15 minutes. During the follow up by phone three months after operation, the patient reported that the treatment effect was satisfactory, whereas the SFR value became stable as 7 ml per 15 minutes, drooling frequency and drooling severity (DFDS) score decreased from 6 to 5, drooling impact scale (DIS) score decreased from 43 to 26. US-guided percutaneous MWA of submandibular gland seems to be an alternative, minimal invasive, and effective treatment for refractory sialorrhea.We described a patient with refractory sialorrhea treated successfully with ultrasound (US) guided percutaneous microwave ablation (MWA).

Modified Stair-Step Flap With Turnover Flap for Drooling Correction After Lower Lip Composite Defect Reconstruction.

BACKGROUND: Oral commissure and buccal complex defects are commonly seen after cancer ablation. Free flap reconstruction can offer adequate soft tissue volume and outer skin lining. However, oral incompetence often occurs when an oral commissure has defects, particularly when the patient receives postoperative radiotherapy. The purpose of this article was to describe our method of the modified stair-step technique and improve the oral competence. METHODS: This study involves 22 patients who had partial lower lip and/or upper lip defects and underwent flap reconstruction and/or postoperative radiotherapy resulting in oral incontinence. Fourteen patients had been treated with an anterolateral thigh flap, 7 patients had been treated with a radial forearm flap, and 1 patient had been treated with a fibular osteocutaneous flap. Our modified stair-step commissuroplasty was done in all cases, and a debulking procedure was done in each second operation. RESULTS: Acceptable oral continence and contour were achieved in 19 patients. Some patients still had drooling after their second operation, but it was considerably less than before. CONCLUSIONS: This technique is an easy procedure for revision of lower lip deformities after oral commissure reconstruction.

Surgical Management of Sialorrhea: A Systematic Review and Meta-analysis.

OBJECTIVE: Chronic sialorrhea commonly occurs in patients with neurodevelopmental disorders. While conservative management can provide sufficient symptom control, surgical intervention is often required. One of the most common procedures utilized is submandibular gland excision (SMGE), with or without parotid duct ligation or rerouting (PDL or PDR). This study aims to compare these surgical approaches and their outcomes. DATA SOURCES: PubMed, Web of Science, and Embase. REVIEW METHODS: This systematic review includes studies of patients with chronic sialorrhea treated with SMGE alone or SMGE plus PDR or PDL and reports on postintervention outcomes and complications. Two independent investigators assessed study eligibility, rated quality, and extracted data for analysis. A random effects model was used for meta-analysis of pooled data. RESULTS: Of 3186 studies identified, 21 met inclusion criteria, with 708 patients: 103 underwent SMGE alone (15%); 299 (42%), SMGE and PDL; and 306 (43%), SMGE plus PDR. Overall, a majority of patients had significant improvement, with very good to excellent control of symptoms after surgery: SMGE, 82% (95% CI, 73%-89%); SMGE and PDL, 79% (95% CI, 73%-85%); and SMGE and PDR, 85% (95% CI, 75%-92%). Importantly, there was no significant difference in outcomes with the addition of PDL or PDR. Reported complications included sialocele, parotitis, dental caries, and dry mouth. CONCLUSION: Our systematic review identified consistent positive outcomes with SMGE for patients with chronic sialorrhea but no additional benefit when PDR or PDL was performed as a concurrent procedure.

Subtotal functional sialoadenectomy vs four-duct ligation for the treatment of drooling in neurologically impaired children: Long-term follow-up.

OBJECTIVES: The best surgical option to treat drooling in neurodisabilities is still under debate. The aim of this study was to describe the technique of subtotal functional sialoadenectomy (SFS) (ie four-duct ligation (4-DL) together with bilateral sublingual gland excision) and its long-term outcomes, in comparison with 4-DL. DESIGN: Retrospective observational cohort study. SETTING: Unit of Pediatric Surgery of Bambino Gesù Children's Hospital (Rome). PARTICIPANTS: Seventy-five patients surgically treated for drooling between 2002 and 2012, with at least five years of follow-up, divided into two groups: 4-DL group (19 patients) underwent four-duct ligation, and SFS group (56 patients) underwent subtotal functional sialoadenectomy. MAIN OUTCOME MEASURES: Primary end points were the evaluation of drooling improvement after surgery (parameters: Drooling Severity and Frequency Scale, DSFS; no of bibs/day; no of shirts/day; no of pneumonia/year; use of antidrooling drugs) and the comparison between two different surgical techniques. RESULTS: Median age at surgery was 10 years (1-35). Long-term outcomes showed significant improvement in DSFS and in no of shirts/day in both groups. Significantly better results were found in the SFS group than in the 4-DL group as far as DSFS (P value .045), no of bibs/day (P value .041), no of shirts/day (P value .032) are concerned. Reoperation rate for recurrence was 42% in the 4-DL group and 0% in the SFS group (P value < .0001). Six patients (8%; 2 in the 4-DL group and 4 in the SFS group) experienced perioperative complications, while 4 patients (5%; 2 in the 4-DL group and 2 in the SFS group) recorded long-term complications, with no difference between groups neither need for surgical treatment. No surgery-related mortality was recorded. CONCLUSIONS: In our experience, subtotal functional sialoadenectomy ensured significantly greater long-term effects than four-duct ligation for drooling treatment in neurologically impaired child, with equal complication rate.

Primary sialorrhea and its surgical treatment with denervation of the submandibular glands in combination with sublingual gland excision.

OBJECTIVES: The purpose of this study was to evaluate the effectiveness of ablating postganglionic parasympathetic fibers of the submandibular ganglion in combination with sublingual gland excision in the treatment of primary sialorrhea. MATERIALS AND METHODS: Healthy volunteers were initially chosen to determine a basic saliva flow rate value by saliva collection. Next, unstimulated and stimulated saliva flow rates, and a visual analog scale index for sialorrhea were recorded in all the patients pre- and postoperatively. In addition, the generalized anxiety disorder scale was used to assess preoperative anxiety symptoms. The follow-up durations ranged from 24 to 36 months after surgery. RESULTS: A total of 10 patients were included in this study. Preoperatively, the mean unstimulated saliva flow rate for the 10 patients was 11.26 ± 4.19 ml/10 min, the stimulated saliva flow rate was 16.76 ± 3.49 ml/6 min, and the visual analog scale index was 66.29 ± 14.86. Postoperatively, the mean unstimulated and stimulated saliva flow rates were significantly reduced to 5.99 ± 1.33 ml/10 min (p = 0.001) and 13.28 ± 1.86 ml/6 min (p = 0.013), respectively, and the visual analog scale index was 25.41 ± 5.6 (p < 0.001). No complications were found after operation. CONCLUSION: This study demonstrates that ablation of postganglionic parasympathetic fibers of the submandibular ganglion in combination with sublingual gland excision by an intraoral approach is a simple, safe, and efficient approach for treating primary sialorrhea.

Cost-effectiveness of botulinum neurotoxin A versus surgery for drooling: a randomized clinical trial.

AIM: This study compared the cost-effectiveness of botulinum neurotoxin A (BoNT-A) injections with two-duct ligation of the submandibular glands as treatment for severe drooling after one treatment cycle. METHOD: The study was part of a larger, partly single-blinded, randomized clinical trial (trialregister.nl identifier NTR3537). Data were collected between 2012 and 2017. Evaluation was at 32 weeks after one treatment cycle. Fifty-seven patients with cerebral palsy or other neurological, non-progressive disorders and severe drooling classified as having a drooling frequency ≥3 or a drooling severity ≥2, in whom conservative treatment was deemed ineffective, were randomized to treatment by BoNT-A or two-duct ligation. An incremental cost-effectiveness ratio (ICER) was calculated using the success rates as the measure of benefit. Treatment success was defined as a decrease ≥50% from baseline to 32 weeks in the subjective visual analogue scale for the severity of drooling or the objective drooling quotient. RESULTS: Fifty-three patients were analysed (22 females, 31 males; mean age 11y, range 8-22y). Average costs for one treatment cycle, which included one BoNT-A injection, were €1929 (standard error 62) for BoNT-A and €3155 (standard error 99) for two-duct ligation. Treatment success was in favour of two-duct ligation (63% vs 27%; number needed to treat 3). The ICER was €34 per 1% gain in treatment success in favour of two-duct ligation versus BoNT-A. INTERPRETATION: The additional cost of two-duct ligation is to some extent offset by a larger treatment success rate compared with BoNT-A. WHAT THIS PAPER ADDS: Botulinum neurotoxin A (BoNT-A) is less expensive per percentage of success than two-duct ligation. The additional cost of two-duct ligation over BoNT-A is offset by greater treatment success.

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.