Anterior sialorrhoea, or “drooling,” is the involuntary loss of saliva due to an inability to manage oral secretions. In cerebral palsy (CP), drooling results from oral motor dysfunction, significantly impacting the quality of life of both patients and their caregivers. Despite its physical and psychological repercussions, the clinical approach to sialorrhoea remains poorly defined.
ObjectivesThis document aims to provide a guideline for the evaluation and management of sialorrhoea in children with CP, based on the best available evidence and expert consensus. Additionally, it seeks to identify gaps in the literature and propose future research directions.
MethodologyA working group was formed, composed of paediatric neurologists from the Spanish Society of Paediatric Neurology (SENEP). A systematic review of the literature was conducted in databases such as MEDLINE, PubMed, and Cochrane, prioritising randomised clinical trials and systematic reviews. Clinical evidence was included when higher-level evidence was unavailable, and recommendations were formulated based on expert consensus.
Consensus and DiscussionThe management of sialorrhoea should be multidisciplinary, including thorough clinical evaluations and assessment scales for severity and impact. Treatment should follow a stepwise approach, starting with non-pharmacological strategies (oromotor therapy, postural correction) and, if necessary, pharmacological interventions with anticholinergics (glycopyrrolate, trihexyphenidyl) or botulinum toxin injection. Surgical options and radiotherapy are reserved for refractory cases.
ConclusionSialorrhoea is a clinically relevant issue in children with CP, requiring an individualised approach based on the best available evidence. This consensus provides a useful guideline for clinical practice and highlights the need for further studies to optimise treatment.
La sialorrea anterior, «babeo», es la pérdida involuntaria de saliva debido a una incapacidad para manejar las secreciones orales. En la parálisis cerebral (PC), el «babeo» es consecuencia de una disfunción motora oral que afecta significativamente la calidad de vida de los pacientes y sus cuidadores. A pesar de sus repercusiones físicas y psicológicas, el abordaje clínico de la sialorrea sigue sin estar bien definido.
ObjetivosEste documento tiene como objetivo proporcionar una guía para la evaluación y manejo de la sialorrea en niños con PC, basándose en la mejor evidencia disponible y en el consenso de expertos. Además, busca identificar lagunas en la literatura y proponer futuras líneas de investigación.
MetodologíaSe constituyó un grupo de trabajo conformado por neuropediatras de la Sociedad Española de Neurología Pediátrica (SENEP). Se realizó una revisión sistemática de la literatura en bases de datos como Medline, PubMed y Cochrane, priorizando ensayos clínicos aleatorizados y revisiones sistemáticas. Se incluyó evidencia clínica cuando no existía evidencia de mayor nivel, y las recomendaciones fueron formuladas según el consenso de expertos.
Consenso y discusiónEl manejo de la sialorrea debe ser multidisciplinar incluyendo evaluaciones clínicas exhaustivas y escalas de valoración, gravedad e impacto. El tratamiento debe seguir un enfoque escalonado, comenzando por estrategias no farmacológicas (terapia oromotora, corrección postural) y, si es necesario, intervenciones farmacológicas con anticolinérgicos (glicopirronio, trihexifenidilo) o infiltración de toxina botulínica. Las opciones quirúrgicas y la radioterapia quedan reservadas para casos refractarios.
ConclusiónLa sialorrea es un problema clínico relevante en niños con PC, que requiere un abordaje individualizado basado en la mejor evidencia disponible. Este consenso proporciona una guía útil para la práctica clínica y destaca la necesidad de más estudios para optimizar el tratamiento.
Anterior sialorrhoea, also known as ptyalism or drooling, is defined as the involuntary loss of saliva and oral content due to the inability to properly manage oral secretions.1 Although this phenomenon is physiological in children younger than 2 years, its persistence beyond the age of 4 years is considered pathological and is usually associated with neuromuscular disorders, intellectual disability, or orofacial structural alterations.2,3 In the majority of paediatric cases, sialorrhoea is not a primary entity but rather secondary to an underlying condition.
From a clinical perspective, sialorrhoea may be classified as anterior, when observed in the physical examination, or posterior, when it is not easily identified; the latter form usually presents in patients with severe oropharyngeal dysphagia, with the subsequent risk of aspiration. In some cases, both types may coexist in the same individual.
In pathophysiological terms, aetiology of sialorrhoea is multifactorial and includes anatomical abnormalities, sensory dysfunction, hypersecretion of saliva, and neuromuscular disorders.4 In the context of cerebral palsy (CP), sialorrhoea has been established to be a consequence not of hypersalivation but rather of an oral motor dysfunction secondary to an alteration in the oral and pharyngeal phases of swallowing, which hinders adequate control and removal of secretions.5,6 However, in cases of CP with dyskinetic manifestations, abnormal involuntary movements may stimulate the activity of the parotid glands, increasing saliva production.7
Despite its clinical impact and repercussion on the quality of life of patients and caregivers, sialorrhoea is still an underdiagnosed symptom that is insufficiently treated in the paediatric population with neurological disease.8 Considering that the optimal approach for its management is yet to be clearly defined,9,10 it is essential to define assessment and treatment strategies based on the best available evidence. The scarcity of reliable studies in this field11 underscores the need to refer to expert consensus documents as key tools in clinical decision-making, enabling the drafting of recommendations based both on scientific evidence and on the accumulated clinical expertise.
EpidemiologyLittle research has addressed the prevalence of sialorrhoea, and the available data present considerable heterogeneity due to differences in the study design, patient selection criteria, and the methodology used for data collection and analysis. Although the available literature does not allow us to draw definitive conclusions, one-third of patients with CP are estimated to present some degree of sialorrhoea.12
Aim of the consensus statementThis consensus statement is aimed at providing healthcare professionals with a guideline based on the best evidence possible for the assessment, intervention, and treatment of sialorrhoea in children with PC. Furthermore, it identifies some current knowledge gaps that may guide future lines of research in this field.
Despite the existence of consensus guidelines addressing several aspects of the management of CP, there is no updated document in Spain that includes the most recent advances in the treatment of associated comorbidities, such as sialorrhoea. The lack of a standardised protocol hinders clinical decision-making and highlights the need for clear recommendations adapted to clinical practice.
Therefore, the main aim of this consensus document is to establish a framework for the diagnosis and therapeutic management of anterior sialorrhoea in paediatric patients with CP. This document is intended to become a useful tool for healthcare professionals in clinical decision-making, facilitating comprehensive evaluation and selection of therapeutic strategies based on scientific evidence and clinical experience.
MethodsA working group was established to draft this consensus document, including paediatric neurologists from several hospitals, who belong to the cerebral palsy working group of the Spanish Society of Paediatric Neurology and are experienced in the diagnosis and treatment of sialorrhoea in patients with cerebral palsy.
We conducted a systematic literature review in the MEDLINE, PubMed, and Cochrane databases, using the following strategy: ((guidelines [MeSH Terms]) OR consensus document [MeSH Terms]) AND cerebral palsy (guidelines [MeSH Terms]) AND sialorrhoea. To issue recommendations, we prioritised studies with the highest possible level of evidence, giving preference to randomised controlled trials (RCT) and systematic reviews. In the absence of such level of evidence, we included observational studies and data based on clinical practice.
Where the literature did not provide conclusive evidence, we included expert opinions, which are duly identified as such in the document and which must be interpreted according to clinical judgement. Furthermore, we issue recommendations based on consensus by the expert group, with the aim of facilitating decision-making in everyday clinical practice (Fig. 1).
DiagnosisClinical assessment
As drooling is a multifactorial problem, a series of evaluations are required; ideally, these should be performed by a multidisciplinary team, preferentially led by the physician responsible for the patient (Fig. 2).13–17
Initial clinical assessment18
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Comprehensive clinical history taking that includes medical and psychosocial aspects of the patient, as well as the patient’s motivation and capacity to participate in the diagnosis and treatment of sialorrhoea.
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Review of the prescribed drugs: antiseizure medications, benzodiazepines, and antipsychotics.
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Comprehensive physical and neurological examination that includes:
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Awareness, cranial nerves, general motor skills, muscle tone, and posture.
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Orofacial examination: respiratory pattern (mouth, nose, etc), oral closure, occlusion, and lip seal. Oral sensorimotor pattern (sensitivity, tone, tongue position). Management of secretions. Oral hygiene and health.
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Language (dysarthria, dyspraxia, etc) and communication skills.
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Assessment of the oropharyngeal stage of swallowing during eating and drinking (swallowing safety and effectiveness).
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Assessment by a paediatric gastroenterologist to evaluate nutritional and hydration status; ruling out the presence of gastro-oesophageal reflux, which, when severe, may be associated with overstimulation of the salivary glands.
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Assessment by the otorhinolaryngology department to rule out obstruction of the upper respiratory tract.
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Assessment by a rehabilitation physician.
For comprehensive assessment of patients with sialorrhoea, it is essential to determine the frequency, intensity, and impact of drooling on the quality of life of the patients and their families. Although several objective methods have been developed to quantify anterior sialorrhoea in children with developmental disorders, no standardised and validated assessment system has been developed to date that comprehensively addresses all clinical and functional aspects of drooling in this population.19,20
Given this limitation, several subjective questionnaires have been developed and validated in recent years to record both patients’ and caregivers’ perception. These scales have been shown to be useful in assessing the severity and functional impact of sialorrhoea, and in monitoring the response to different therapeutic interventions. Their use in clinical practice enables us to adopt a more structured, systematic approach in the assessment of these patients, facilitating therapeutic decision-making on an individual basis.
Several useful scales are available for clinical practice (Fig. 3):
- 1.
The drooling severity scale (Thomas-Stonell and Greenberg scale),21 which enables complete assessment of the frequency and intensity of sialorrhoea in the patient and facilitates monitoring of treatment response.
- 2.
The Drooling Rating Scale,22 designed to assess drooling intensity in everyday activities. It includes ten items, classified in a 1 to 10 point-scale, which reflects the changes in drooling perceived by the person filling out the questionnaire.
- 3.
The Sialorrhea Rating Scale, a visual scale used by our group, which classifies drooling into 5 levels (Fig. 4).
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The Drooling Impact Scale,22 which quantifies the short- and medium-term response and benefits of treatments in controlling sialorrhoea.
The therapeutic approach for patients with sialorrhoea should be based on the clinical and functional impact of drooling, considering its repercussion on the quality of life of patients and their families. The main criterion to start an intervention is the presence of significant impact in the physical, social, and emotional spheres, whether reported by the patient themself or by caregivers.23
Before starting any treatment, it is essential to perform an exhaustive baseline examination, following the previously described clinical schema, and to include objective and subjective measurement tools that enable quantification of the severity and impact of sialorrhoea. The application of validated scales facilitates monitoring of treatment response and individualised decision-making.
Follow-up must be performed every 3 to 6 months, with the aim of assessing the effectiveness of the intervention, persistence of treatment response, and the need to adjust or switch treatment, particularly in those cases in which a pharmacological strategy had been adopted (Table 1).
Pharmacological treatment and infiltration of botulinum toxin A in sialorrhoea.
| Drug | Initial dose | Titration/maximum dose | Adverse reactions | Contraindications |
|---|---|---|---|---|
| Glycopyrronium bromide | 0.02 mg/kg/day | Weekly increase: 0.02 mg/kg/dose until: 0.1 mg/kg/dose | Constipation | Myasthenia gravis |
| Every 6-8 hours | Irritability | Glaucoma | ||
| Somnolence | Kidney disease | |||
| Tachycardia | Intestinal/urinary obstruction | |||
| Dry mouth | ||||
| Vomiting | ||||
| Nasal congestion | ||||
| Trihexyphenidyl (Artane®) | 0.1-0.2 mg/kg/day every 8-12 h | Increase of 10%–20% every 1−2 weeks | Urinary retention | Glaucoma |
| Mean: 0.5 mg/kg/day | Blurred vision | Intestinal/urinary obstruction | ||
| Dry mouth | ||||
| Irritability | ||||
| Transdermal scopolamine (Scopoderm® TTS 1.5 mg) | First dose of ¼ patch | Progressive weekly up-titration until reaching the complete dose. | Irritability | Myasthenia gravis |
| Change every 3 days. | Hyperthermia | Ulcerative colitis | ||
| Skin reaction | Glaucoma | |||
| Behavioural change | ||||
| Ophthalmic atropine (Colircusi Atropina® 0.5%-1%) | 1-2 drops | – | – | Limited use in paediatric patients due to lack of evidence |
| 3 times/day Sublingual | ||||
| Incobotulinumtoxin A (Xeomin®) | 75-100 U* | Every 4-6 months | Pain at injection site | – |
| Haematoma | ||||
| Dysphagia | ||||
| Dry mouth |
There is currently no standardised treatment protocol for the management of sialorrhoea in children with CP, as no strategy had been shown to be universally effective in all patients. For this reason, therapeutic management should be individualised, considering the clinical characteristics of each patient and the functional impact of drooling on their quality of life.
The main targets of treatment include reduction of salivary flow, improved social interaction and hygiene, as well as a reduction in the risk of respiratory and skin infections. Therefore, we propose a treatment algorithm based on a stepwise approach, which progresses from less invasive to those more complex interventions.
This algorithm focuses on 4 different therapeutic options, which follow a hierarchical approach, moving from less invasive to more invasive treatments24:
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Non-pharmacological treatment
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Pharmacological treatment
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Surgical treatment
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Radiotherapy.
In general terms, the initial management of sialorrhoea may prioritise non-pharmacological measures, combined with minimally invasive pharmacological treatments if necessary. Such invasive options as surgical interventions and radiotherapy are reserved for those cases not showing appropriate response to conventional treatments (Figs. 5 and 6).
Non-pharmacological treatment24
Non-pharmacological approaches are the first-line treatment and should particularly be considered in patients with mild or moderate sialorrhoea. To ensure effectiveness, candidates for treatment should have sufficient cognitive status, disability compatible with treatment application, and the ability to follow basic instructions.
Treatment strategies include oromotor rehabilitation, which may be monitored by a rehabilitation physician or speech therapist with experience in neurodevelopmental disorders. The main aims of rehabilitation include:
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Improving head control.
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Toning of perioral muscles.
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Optimising lip seal.
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Correcting dental malocclusions (eg, anterior open bite).
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Improving tongue position both at rest and during mastication and swallowing.
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Stimulating the swallowing reflex.
A minimum duration of 6 months is recommended before considering a change in treatment strategy.
Pharmacological treatmenta. Oral drugs
To date, there is no clear consensus on the most effective drug nor on the optimal doses in the paediatric population. However, anticholinergic agents represent the most widely used pharmacological option, given their blocking of cholinergic muscarinic receptors, which reduces production of saliva.25–27
First-line drugs:
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Glycopyrronium bromide (GPB) oral solution 1 mg/5 mL (150 mL, Pharma International®)27,28: level of evidence B. It should be considered the first-line treatment option. Although it is covered by the Spanish National Health System, it must be requested as a foreign drug in Spain.
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Initial dose: 0.02 mg/kg, 3-4 times/day.
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Progressive up-titration: +0.02 mg/kg/dose each week until reaching a maximum dose of 0.1 mg/kg/dose.
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Adverse effects: constipation, irritability, somnolence or insomnia, visual alterations, tachycardia, dry mouth, nasal congestion, vomiting, and cutaneous flushing.
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Contraindications: myasthenia gravis, severe ulcerative colitis, narrow-angle glaucoma, end-stage kidney disease, intestinal or urinary obstruction.
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Trihexyphenidyl (2 mg and 5 mg tablets, Artane®)
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Off-label administration in children (3 months-17 years).
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Initial dose: 0.1−0.2 mg/kg/day, every 8−12 h.
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Progressive up-titration: 10%–20% increase every 1-2 weeks.
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Mean dose: 0.5 mg/kg/day (2-3 mg/dose, 2-3 times/day).
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Adverse effects: urinary retention, irritability, blurred vision, xeroderma.
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Indications: it is especially useful in patients with sialorrhoea associated with dyskinetic symptoms.
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Contraindications: narrow-angle glaucoma, gastrointestinal or genitourinary tract obstruction.
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Second-line drugs:
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Transdermal scopolamine (Scopoderm® TTS 1.5 mg)
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Level of evidence: B.
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Titration: starting with ¼ patch, with weekly increases in the amount applied until reaching the complete dose.
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Adverse effects: irritability, somnolence, hyperthermia, skin reactions.
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Contraindications: glaucoma, obstructive uropathy, obstructive intestinal symptoms.
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The use of such other pharmacological options as benztropine mesylate (Cogentin®) and sublingual ophthalmic atropine (Colircusi Atropina® 0.5%-1%) is limited due to the lack of evidence in paediatric populations or adverse effects.
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Intraglandular injection of botulinum toxin A has shown effectiveness in reducing sialorrhoea in children with neurological disorders (level of evidence A). Its action mechanism is based on the inhibition of acetylcholine release in cholinergic nerve terminals, thus reducing production of saliva.
The available options include:
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OnabotulinumtoxinA (Botox®, Allergan Pharmaceuticals)
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AbobotulinumtoxinA (Dysport®, Biopharm Ltd.)
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IncobotulinumtoxinA (Xeomin®, Merz Pharmaceuticals GmbH): this is the only form specifically indicated for chronic sialorrhoea in children.32
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Recommended dose33:
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U of Xeomin® divided between the bilateral parotid gland and submandibular gland.
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Recommended dilution: 100 U Xeomin® in 2 mL of physiological saline, equally distributed between the parotid glands (2-4 sites) and submandibular glands (1-2 sites).
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Frequency of administration: every 3-6 months, using ultrasound-guided infiltration.
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Adverse effects: dry mouth, mastication and swallowing alterations.
Surgical interventions are reserved for severe and refractory cases. Options include:
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Surgical excision of the submandibular or parotid gland (risk of lesion to the facial nerve).
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Bilateral salivary duct ligation (parotid or submandibular route).
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Redirection of the submandibular duct (risk of aspiration in posterior sialorrhoea).
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Gland radiofrequency or ablation with sclerotherapy.
Radiotherapy is the last resort in extremely severe cases that are refractory to conventional treatment.
Conclusions and expert recommendationsSialorrhoea is a frequent manifestation in children with neurodevelopmental disorders, with an estimated prevalence of up to 30% among patients with CP. Its impact extends beyond clinical aspects, significantly affecting the quality of life of patients and their families. Anterior sialorrhoea may cause skin irritation, local infections, and impairment of self-esteem, whereas posterior sialorrhoea is associated with a high risk of aspiration and recurrent respiratory infections, representing a relevant cause of morbidity and mortality.37
Clinical management of sialorrhoea should be performed by a multidisciplinary team to include assessments by paediatric neurologists, rehabilitation physicians, speech therapists, otorhinolaryngologists, and other specialists, depending on the case. Therapeutic decision-making should be based on an exhaustive, individualised assessment, considering the age of the patient, the severity and functional impact of drooling, and the tolerance to the different treatment strategies. To achieve this, the use of objective and subjective scales is fundamental in quantifying severity and in monitoring of treatment response.
Treatment should follow a hierarchical and stepwise approach, prioritising non-pharmacological strategies that include posture correction interventions, oromotor rehabilitation, and biofunctional therapy, aimed at improving oral motor control. These measures represent the only approach that directly targets the aetiopathogenesis of drooling, and should therefore be considered the first line of treatment. However, their effectiveness may be limited, requiring the use of complementary pharmacological treatment.
Anticholinergic drugs have been shown to be effective in reducing the production of saliva, although their use is conditioned by the onset of systemic adverse effects, including cognitive/behavioural alterations, insomnia, gastrointestinal disorders, and urinary retention. Among these, glycopyrronium bromide is the first-line drug due to its effectiveness and better tolerability profile, as its quaternary amine structure limits its transport across the blood-brain barrier, thus reducing adverse effects at the central level.
In cases in which pharmacological treatment is ineffective or poorly tolerated, the infiltration of botulinum toxin type A to the submandibular and parotid glands represents an alternative (level of evidence A). Its administration temporarily inhibits saliva secretion, with minimal adverse effects; it should be guided by ultrasound to ensure safety and effectiveness.
Finally, surgical interventions and radiotherapy are reserved for patients with severe and refractory sialorrhoea, when previous strategies have failed. Such options should be considered with caution, due to their irreversible nature and possible associated risks, including neurological damage, severe dry mouth, and dysphagia.
In conclusion, the management of sialorrhoea in children with cerebral palsy requires an individualised, multidisciplinary approach, based on the best available evidence. Selection of the therapeutic strategy may be based on symptom severity, the impact on the quality of life of the patient, and the risk-benefit balance of each intervention. Despite advances in treatment, there are still limitations to the available evidence, underscoring the need for new research aimed at optimising therapeutic strategies and establishing more precise and standardised management protocols.
None.
