Sialorrhea in children with cerebral palsy<sup>,</sup>

Dias, Bruno Leonardo Scofano; Fernandes, Alexandre Ribeiro; Maia Filho, Heber de Souza

doi:10.1016/j.jped.2016.03.006

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Review articles • J. Pediatr. (Rio J.) 92 (6) • Nov-Dec 2016 • https://doi.org/10.1016/j.jped.2016.03.006 copy

Sialorrhea in children with cerebral palsy^☆ ☆ Please cite this article as: Scofano Dias BL, Fernandes AR, Maia Filho HS. Sialorrhea in children with cerebral palsy. J Pediatr (Rio J). 2016;92:549-58. ^,^☆☆ ☆☆ Study carried out at Rede SARAH de Hospitais de Reabilitação, Rio de Janeiro, RJ, Brazil.

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

Objective:

To review the literature on sialorrhea in children with cerebral palsy.

Source of data:

Non-systematic review using the keywords "sialorrhea" and "child" carried out in the PubMed^®, LILACS^®, and SciELO^® databases during July 2015. A total of 458 articles were obtained, of which 158 were analyzed as they were associated with sialorrhea in children; 70 had content related to sialorrhea in cerebral palsy or the assessment and treatment of sialorrhea in other neurological disorders, which were also assessed.

Data synthesis:

The prevalence of sialorrhea is between 10% and 58% in cerebral palsy and has clinical and social consequences. It is caused by oral motor dysfunction, dysphagia, and intraoral sensitivity disorder. The severity and impact of sialorrhea are assessed through objective or subjective methods. Several types of therapeutic management are described: training of sensory awareness and oral motor skills, drug therapy, botulinum toxin injection, and surgical treatment.

Conclusions:

The most effective treatment that addresses the cause of sialorrhea in children with cerebral palsy is training of sensory awareness and oral motor skills, performed by a speech therapist. Botulinum toxin injection and the use of anticholinergics have a transient effect and are adjuvant to speech therapy; they should be considered in cases of moderate to severe sialorrhea or respiratory complications. Atropine sulfate is inexpensive and appears to have good clinical response combined with good safety profile. The use of trihexyphenidyl for the treatment of sialorrhea can be considered in dyskinetic forms of cerebral palsy or in selected cases.

KEYWORDS
Sialorrhea; Cerebral palsy; Child

Technique	Method	Description
Drooling quotient (DQ)²¹21 Heine RG, Catto-Smith AG, Reddihough DS. Effect of antireflux medication on salivary drooling in children with cerebral palsy. Dev Med Child Neurol. 1996;38:1030-6.^,³⁴34 Raap D. Drool control: long-term follow-up. Dev Med Child Neurol. 1980;22:448-53.	Saliva collection and use of its own formula for quantification	At every 15 s, in a 15-minute period (60 observations) the presence or absence of sialorrhea was observed. DQ {%} = 100 × the number of episodes of sialorrhea/60 observations
Sochaniwskyj's technique¹²12 Silvestre-Rangil J, Silvestre FJ, Puente-Sandoval A, Requeni-Bernal J, Simó-Ruiz JM. Clinical-therapeutic management of drooling: review and update. Med Oral Patol Oral Cir Bucal. 2011;16:e763-6.^,³⁵35 Sochaniwskyj AE. Drool quantification: noninvasive technique. Arch Phys Med Rehabil. 1982;63:605-7.	Saliva collection and use of its own formula for quantification	Collection of saliva that leaked through the mouth and reached the chin, using a glass, for a 30-minute period
Thomas-Stonnel and Greenberg scale¹²12 Silvestre-Rangil J, Silvestre FJ, Puente-Sandoval A, Requeni-Bernal J, Simó-Ruiz JM. Clinical-therapeutic management of drooling: review and update. Med Oral Patol Oral Cir Bucal. 2011;16:e763-6.^,²⁶26 Thomas-Stonell N, Greenberg J. Three treatment approaches and clinical factors in the reduction of drooling. Dysphagia. 1988;3:73-8.	Direct observation of the examiner quantified through a severity scale	1 - Dry lips (no sialorrhea); 2 - Wet lips (mild sialorrhea); 3 - Wet lips and chin (moderate sialorrhea); 4 - Wet clothing around the neck (severe sialorrhea); 5 - Wet clothing, hands, and objects (profuse sialorrhea)
Others³¹31 van Hulst K, Lindeboom R, van der Burg J, Jongerius P. Accurate assessment of drooling severity with the 5-minute drooling quotient in children with developmental disabilities. Dev Med Child Neurol. 2012;54:1121-6.	Measuring the weight of the container used for direct collection of saliva	Use of collection units, towels, and diapers or dental cotton rolls

Therapeutic modalities	Advantages	Disadvantages
Training of sensory awareness and oral motor skills	Treats the underlying cause; Long-lasting effect	Depends on the intellectual capacity; Requires access to regular therapy and a trained professional
Botulinum toxin use	High efficacy; Safe	Transient effect; Requires equipment and trained multidisciplinary staff; Requires good clinical condition for sedation or anesthesia; Does not treat the underlying cause
Surgical treatment	Definitive efficacy	Definitive side effects; Requires general anesthesia; Demands equipment and trained surgical team; Does not treat the underlying cause.
Pharmacological treatment with anticholinergics	Proven effectiveness; Self-administered (by the patient or caregiver); Does not require sedation or anesthesia; Transient side effects	Frequently anticholinergic effects (vomiting, diarrhea, irritability, changes in mood and insomnia); Does not treat the underlying cause.

Author and year	Drug	Associated pathology	Ages	n	Study design	Results	Side effects
Mier et al.⁵⁶56 Mier RJ, Bachrach SJ, Lajin RC, Barker T, Childs J, Moran M. Treatment of sialorrhea with glycopyrrolate: a double-blind, dose-ranging study. Arch Pediatr Adolesc Med. 2000;154:1214-8.	Glycopyrrolate	CP and other neurological conditions	4-19 years	39	DB, PC	Glycopyrrolate 0.10 mg/kg/dose is effective for the control of sialorrhea	Incidence of 20% of SE, enough to withdraw the drug
Zeller et al.⁵⁵55 Zeller RS, Davidson J, Lee HM, Cavanaugh PF. Safety and efficacy of glycopyrrolate oral solution for management of pathologic drooling in pediatric patients with cerebral palsy and other neurologic conditions. Ther Clin Risk Manag. 2012;8:25-32.	Glycopyrrolate	CP and other neurological conditions	3-18 years	137	Uncontrolled clinical trial	Satisfactory response in 40.3% of patients in the 4th week and 52.3% in the 24th week, with a peak of 56.7% in the 16th week	Intestinal constipation (20.4%), vomiting (17.5%), diarrhea (17.5%), pyrexia (14.6%), xerostomia (10.9%), flushing (10.9%), nasal congestion (10. 9%)
Mato et al.⁵⁰50 Mato A, Limeres J, Tomas I, Munoz M, Abuin C, Feijoo JF, et al. Management of drooling in disabled patients with scopolamine patches. Br J Clin Pharmacol. 2010;69:684-8.	Scopolamine TD	CP and other neurological conditions	12-58 years	30	Prospective, randomized, DB, PC	Significant improvement (p < 0.005) in the scopolamine group	Incidence of 23% of SE (four patients with more severe SE and three with milder SE)
Camp-Bruno et al.⁵¹51 Camp-Bruno JA, Winsberg BG, Green-Parsons AR, Abrams JP. Efficacy of benztropine therapy for drooling. Dev Med Child Neurol. 1989;31:309-19.	Benztropine	CP	4-44 years (14 children/adolescents and 6 adults)	20	DB, PC	Significant reduction in sialorrhea when compared to placebo	Incidence of 11% of more severe SE resolved in 24-48 h after drug withdrawal
Carranza-del Rio et al.⁵⁷57 Carranza-del Rio J, Clegg NJ, Moore A, Delgado MR. Use of trihexyphenidyl in children with cerebral palsy. Pediatr Neurol. 2011;44:202-6.	Trihexyphenidyl	CP	1-18 years	70	Retrospective	Most patients (96%) reported some improvement in sialorrhea	SE were found in 69.3% of patients: intestinal constipation (43), urinary retention (19), xerostomia (seven), blurred vision (five), increase in involuntary movements (four) reduction in seizure control (four), hallucinations (two)
De Simone et al.⁷⁰70 De Simone GG, Eisenchlas JH, Junin M, Pereyra F, Brizuela R. Atropine drops for drooling: a randomized controlled trial. Palliat Med. 2006;20:665-71.	Atropine sulfate SL	Upper digestive tract cancer	48-87 years	22	Prospective, randomized, DB, PC	The study failed to demonstrate effectiveness of atropine sulfate compared to placebo	Only one patient (xerostomia)

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[1] * Corresponding author. E-mail:brunoscofano91@gmail.com (B.L. Scofano Dias).