Download Management of Sialorrhea in Children with Cerebral

AMERICAN JOURNAL OF CLINICAL NEUROLOGY
REVIEW ARTICLE
Management of Sialorrhea in Children with Cerebral Palsy
Ali Alrefai and Samah K Aburahma
Affiliation: Department of Neuroscience, Jordan University of Science and Technology (JUST), Irbid, Jordan
A B S T R A C T
Drooling, the overflowing of saliva from the mouth, is mainly due to neurological disorder and, less frequently, to hypersalivation. Drooling
is prevalent among children with cerebral palsy (CP) and has a negative impact on their social and physical wellbeing. Treatment options
include oral medications, chemodenervation, and submandibular duct relocation. Although no treatment option has proven to be ideal, an
optimum approach needs to be tailored to the needs of the child. This article provides an overview of the different treatment approaches and
significant research findings.
Keywords: drooling, cerebral palsy, botulinum toxin
Correspondence: Ali Alrefai, Department of Neuroscience, Jordan University of Science and Technology (JUST), PO Box 3030, Irbid, Jordan
22110. Tel: (962)-2720-0600, Ext 40708; Fax: (962)-2720-0621; e-mail: [email protected]
sublingual salivary glands produce about 70% of saliva in the
resting state, although when stimulated, the parotid glands
provide most of the saliva. The submandibular glands
produce a high-viscosity fluid, whereas the parotids produce
watery saliva. Saliva serves a number of important functions,
including: (1) providing a protective effect from tooth decay
and the gingival tissues from inflammation; (2) acting as a
lubricant for swallowing and a solvent for facilitating taste;
(3) providing an antibacterial action in the mouth; and (4)
promoting protein and carbohydrate breakdown.
INTRODUCTION
Sialorrhea (drooling) is the unintentional loss of saliva and
other oral contents from the mouth. It is a normal
phenomenon of infancy that subsides in early childhood,
usually by 15–18 months, as a consequence of physiological
maturity of oral motor function [1].
Drooling children frequently have irritated facial skin, foul
odor, and in cold weather the dampness from saliva is
chilling. Their dehydration experience can be a recurrent
problem from chronic fluid loss. They may also damage
books, toys, computers, and other communication aids [2].
Salivary secretion is regulated by a reflex arc. The afferent
part is mainly activated by stimulation of chemoreceptors
located in the taste buds and mechanoreceptors located in the
periodontal ligament. The afferent arc is mediated through
cranial nerves V, VII, IX, and X, which carry impulses to the
salivary nuclei in the medulla oblongata [4]. The efferent part
of the reflex is mainly parasympathetic. Cranial nerve VII
provides control of the submandibular, sublingual, and minor
glands, whereas cranial nerve IX controls the parotid glands
[4]. The flow of saliva is enhanced by sympathetic innervation, which promotes contraction of muscle fibers around the
salivary ducts [5].
Drooling affects the social and physical wellbeing of
children with cerebral palsy (CP), and may represent
problems for caregivers. The unsightly nature of drooling,
speech spray, and a cough or sneeze can lead to social
avoidance [2].
Reported treatment options have included behavioral
modification therapy, oral or topical anticholinergic medications, surgical excision of salivary glands or duct relocation,
and chemodenervation with botulinum toxin. Despite the
abundance of reports on the efficacy and safety profiles of
each treatment option, definitive conclusions are difficult to
draw, given the heterogeneous nature of the patient populations studied and the different outcome measures used in the
various studies. In this review, an analysis of outcome
measures commonly used for assessing response to treatment
and a study of recent reports on the various therapeutic
options available will be presented.
ETIOLOGY AND EPIDEMIOLOGY OF
DROOLING
Hypersalivation, which is excessive saliva production, is not
synonymous with drooling. Drooling in children with CP is
usually not due to hypersalivation; in fact, in most cases, the
volume of saliva produced is normal [6]. It was found that
there was no statistical difference in the rate of salivary flow,
the buffering capacity, and the concentration of sodium and
potassium between children with CP who drool and
unaffected age-matched children [6]. Drooling occurs as a
result of central or peripheral etiologies. Of the central
BASIC PHYSIOLOGY
Healthy subjects secrete variable amounts of saliva, averaging 0.5–1.5 L/day. The parotid and submandibular glands
produce about 90% of saliva [3]. The submandibular and
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American Journal of Clinical Neurology
etiologies, CP is the most common cause in children. The
factors that contribute to drooling in children with CP include
an inefficient coordination of the oral phase of swallowing
and poor lip closure [7]. This has been confirmed by
comparing drinking tasks in normal children with children
with CP [8]. Drooling children with CP had more trouble
initiating swallowing than normal children or children with
CP who did not drool. Other factors that might contribute to
drooling include muscle hypotonia, macroglossia, dental
malocclusion, abnormal posture, and impaired nasal airway
patency [7].
observational method; it is defined as the number of times
when drool was present or absent measured at 15-s intervals
during two 10-min periods separated by a 60-min break.
Despite the absence of studies comparing subjective and
objective measures of saliva production, subjective measures
frequently show an earlier and more significant response to
the therapeutic intervention being studied than objective
measures, and appear to parallel changes in quality of life
assessments [19]. This may be because objective measures
assess drooling at a given fixed period of time, whereas most
subjective measures assess drooling from observations over a
more extended period of time. However, as drooling is
variable throughout the day, subjective tools may actually be
more reflective of the true nature and burden of the problem
for children with drooling, and necessarily complement
information obtained from objective tools.
Drooling is reported to be a significant problem in 10–37%
of patients with CP [9, 10], especially children with
quadriplegic CP attending special schools, where drooling
was found in up to 54% of the children [11].
ASSESSMENT OF DROOLING
A questionnaire was developed to document the social
impact and self-esteem of treating drooling [20]. It consists
of sets of multiple choice questions and a visual analog scale
in which parents evaluate their experiences as well as the
personal reactions of the child.
A detailed history helps to assess the severity of drooling, its
effects on quality of life for the patient and family, and to
decide the therapeutic intervention. Both objective and
subjective measures need to be utilized in assessing
therapeutic intervention. Objective measures are usually
aimed at measuring the amount of saliva. The commonly
reported and utilized objective measures include calculating
the weight of drool using dental bibs [12], weighing dental
rolls placed in different areas of the mouth, positioning
absorbent cotton rolls at the salivary gland duct orifice [13],
and the drool quantification method [14]. The drool
quantification method is an effective non-invasive evaluation
tool that includes a cup-like collection device, a vacuum
pump, plastic tubing, an airtight collection chamber, and a
calibrated test tube. The commonly reported subjective tools
include the drooling severity and frequency rating scale,
Teacher Drooling Scale (TDS), Drooling Quotient (DQ),
caregivers’ questionnaires, and a visual analog scale for
frequency and severity of drooling [15–18]. Table 1 shows the
drooling severity and frequency rating scale. The TDS is a
useful tool for outpatient visits, in which the classroom
teachers indicate the degree of drooling by means of a fivepoint scale. The DQ is a validated, semiquantitative, direct
BEHAVIORAL TREATMENT
Various behavioral techniques have been described for the
treatment of drooling. Despite their appeal because of their
non-invasive nature, there is a paucity of clinical research
documenting their efficacy, and most reports are based on
anecdotal case descriptions. Proposed techniques include
various oral appliances to modify and improve oral motor
function and aid lip closure [21], oral motor stimulation
techniques that emphasize the enhancement of sensorimotor
feedback mechanisms [22, 23], and biofeedback and automatic cueing techniques [24, 25]. However, with the complex
and demanding nature of these techniques, and their
dependency on the cognitive abilities of the patient, they
seem to have fallen out of favor. In the authors’ opinion,
behavioral techniques may have a role as adjunctive therapy
with other treatment modalities; however, this requires
further investigation before definitive recommendations can
be made.
Table 1. Drooling Frequency and Severity Rating Scale
SURGICAL TREATMENT
Frequency
Surgery is indicated when conservative treatment has been
tried for at least 6 months without reduction in drooling [19].
It is best deferred until the patient is 6 years old as, by this
age, there should be full maturation of oral motor function
and coordination.
15 Never drools
25 Occasionally drools (not every day)
35 Frequently drools (part of every day)
45 Constantly drools
Various surgical approaches have been reported, such as
parotid duct ligation with submandibular gland excision,
submandibular gland duct relocation with or without
sublingual gland excision, and parasympathetic neurectomy
[26–29]. Salivary gland resection is associated with significant morbidity including external scar, xerostomia, and, in
the worst cases, facial weakness [26]. Nowadays, the gold
standard surgical procedure is submandibular duct relocation. Crysdale and White [27] reported significant reduction
Severity
15 Dry (never drools)
25 Mild (only lips wet)
35 Moderate (wet on lips and chin)
45 Severe (drool extends to clothes)
55 Profuse (hands, tray, and objects wet)
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Management of sialorrhea in children with CP
decrease side-effects. However, this study is limited by small
sample size. In another double-blind, placebo-controlled,
dose-escalating trial, we reported that bilateral parotid gland
injection with BoNT-A significantly reduced the frequency
and severity of drooling compared with placebo in the smaller
dose, but the reduction was not statistically significant with a
higher dose due to a high dropout rate from the placebo
group [39]. Another dose-finding trial comparing injections
into the submandibular glands alone with injections into the
submandibular and parotid glands found more responders
with the second approach, but no conclusions were made
regarding the ideal dose [17]. In an open-label, randomized
controlled trial, Reid and his group [40] reported that
injection with BoNT-A into the parotid and submandibular
glands in children with CP significantly reduced drooling
compared with a control group. This reduction was maximal
at 1 month after injection and remained significant for
6 months; nine families were still happy with the results at
1 year. No child was reinjected during this time. This is the
first study with long-term follow-up.
in drooling in the majority of children who underwent this
procedure as the salivary flow is redirected to the back of the
mouth, a site more conducive to swallowing than drooling.
The absence of an external scar is appealing to both the
patient and the surgeon. The disadvantages of this procedure
include the need for hospitalization and ranula formation in
8% of patients. The addition of sublingual gland resection to
avoid this complication was found to be ineffective and
increases the chance of bleeding and pain [28]. Patients with
a history of recurrent tonsillitis should have a tonsillectomy 2
or 3 months before the duct relocation.
PHARMACOLOGICAL THERAPY
Salivation is mediated through the autonomic nervous
system, primarily by way of the cholinergic system muscarinic
receptor sites. Blockage of these receptors inhibits nervous
stimulation to the salivary glands. Anticholinergic drugs used
to decrease drooling have widespread effects on all endorgans that are governed by muscarinic stimulation.
Several clinical trials have used anticholinergic drugs to
decrease drooling, but the response was usually partial and at
the price of side-effects [30–32]. A double-blind, placebocontrolled, crossover trial reported that transdermal scopolamine demonstrated ‘‘a significant reduction in drooling’’
with low toxicity [30]. However, the patient population was
heterogeneous, and the method of measurement of improvement was not sufficiently described. Benztropine in one
double-blind trial produced a 65% decrease in drooling, but
the trial term was short and had a 30% dropout rate [31].
Glycopyrrolate, a synthetic antimuscarinic agent that does not
cross the blood–brain barrier, making it an interesting
compound to use, has been investigated in a double-blind,
dose-ranging clinical trial, and showed reduction in drooling.
However, 20% had adverse effects leading to drug withdrawal
[32].
Botulinum toxin type B (BTxB) has been proposed as a
treatment for drooling in patients with neurological disorders
such as PD and amyotrophic lateral sclerosis (ALS) [41, 42].
In a double-blind, placebo-controlled trial in 20 patients with
ALS, 2500 U of BTxB or placebo was injected into the bilateral
parotid and submandibular glands under electromyography
(EMG) guidance [42]. Patients treated with BTxB reported a
global impression of improvement of 82% at 2 weeks
compared with 38% of those treated with placebo. At
12 weeks, 50% of patients who received BTxB continued to
report improvement compared with 14% of those who
received placebo. Based on this study, in a recent practice
parameter update, the Quality Standards Subcommittee of the
American Academy of Neurology has recommended the use
of BTxB for patients with ALS who have medically refractory
drooling (level B evidence) [43].
Botulinum toxin (BoNT), a potent exotoxin produced by
Clostridium botulinum, the same organism responsible for
tetanus, is another medication that may be effective in the
treatment of drooling. It blocks the release of acetylcholine at
the cholinergic neurosecretory junction of the target organs
including the salivary glands. The first published results of
BoNT-A injection to treat drooling appeared in 2000 in
patients with Parkinson’s disease (PD) [33]. Since then,
several case series and unblinded, open-label cohort studies
have reported its effect on drooling in children with CP and
other neurological disorders [34–37]. The injected salivary
glands were the parotid gland alone in one study [34], the
submandibular gland alone in two studies [35, 36], and both
sets of glands in the third study [37]. Three controlled trials
have recently been published [38–40]. A double-blind,
placebo-controlled trial studying the effect of unilateral
injection of the parotid and bilateral submandibular glands
with BoNT-A reported that this approach reduced the
drooling frequency and severity scale and drooling quotient
at most follow-up periods [38]. The investigators adopted
this approach with the intention of keeping at least 50% of
resting and post-prandial saliva production in an attempt to
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In children with CP, Wilken et al [44] randomly assigned
children to receive BoNT-A or BTxB into the parotid and
submandibular glands on both sides, and found that
reduction of drooling was achieved 2 weeks after injection,
with a positive effect lasting about 3–4 months, but this
reduction was not significant between both types of
botulinum toxin.
Although most trials have used ultrasound guidance for
injection, our group reported that injection using anatomical
landmarks was also effective [39]. Trials comparing blind
injection based on anatomical landmarks and injection with
ultrasound guidance are limited.
Side-effects reported with botulinum toxin injection
included thicker, more viscous saliva. The parotid glands
produce thin, serous secretions unlike the submandibular
glands, which produce more viscous saliva. Therefore, this
side-effect is believed to be secondary to decreased parotid
saliva production. Another reported side-effect is difficulty
swallowing, and this is believed to be secondary to local
diffusion of botulinum toxin producing weakness of
surrounding muscles. Some children might require general
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anesthesia to administer botulinum toxin, which could
increase the cost and risk of complications and side-effects.
Other potential effects are hematoma, salivary duct calculi,
and local injuries to the carotid arteries or branches of the
facial nerve.
using BoNT in treating drooling in children with CP, showing
the differences in the dosages used, outcome measures, and
injection sites.
Despite the fact that many studies have reported that
botulinum toxin is effective in treating drooling in children,
there are still unanswered questions. Most studies have used
small heterogeneous patient samples and subjective measures
to evaluate response. Optimally, future studies should use
large homogeneous patient samples and both subjective and
objective outcome measures. The optimum dose, the dilution,
and the number of injection sites need to be standardized.
Table 2 lists some of the recent randomized clinical trials
Radiotherapy (RT) to the salivary glands in doses of 10–
20 Gy in a single fraction or two fractions was used to treat
drooling in patient with PD and ALS, with variable results
[45–47]. The most frequent adverse events reported with RT
were xerostomia and loss of taste. Xerostomia is believed to
be due to the delivery of RT to the parotid glands. Such
adverse events can hopefully be avoided by delivering the RT
to the submandibular glands instead of the parotid glands.
Another rare, but serious adverse event is the potential risk of
RADIATION THERAPY
Table 2. Effect of Botulinum Toxin on Drooling in Children with Cerebral Palsy
Citation
Study group
Study type
Outcome
Results
Comments
Suskind et al
(2002) [17]
22 children
(8–21 years)
CP
Prospective, open-label, doseescalating study: 12 children
with only submandibular
gland injection 10, 20, 30
units and 10 children with
30 units in submandibular
and 20, 30, 40 units in
parotid
Drooling rating scale,
dental roll weight,
DQ
Drooling rating scale:
33% responders in
group 1 and 80%
responders in group 2;
DQ: eight patients in
group 2 have
decreased DQ
Different outcome
measures between
groups, no ideal dose
Jongerius et al
(2004) [18]
45 children
(3–18 years)
CP
Controlled, open-label, clinical
trial. Treatment with
scopolamine patches, then
with BoNT-A into
submandibular glands.
Total dose: 30 U if ,15 kg;
40 U if 15–25 kg; 50 U if
.25 kg
Saliva secretion
(measured by DQ,
TDS, and VAS)
DQ: 53% responded to
scopolamine and 64%
responded to BTX-A
at 2 weeks and 49%
at 24 weeks. TDS:
61.5% good
responders 8 weeks
post BTX-A and 36%
at 24 weeks
71% patients experienced
moderate–severe
side-effects with
scopolamine. With
BTX-A, only nonsevere side-effects
Lin et al
(2008) [38]
13 children
(mean age
14.2¡
1.8 years)
Double-blind, placebocontrolled, randomized
clinical trial. Treatment
with BoNT-A 2 u/kg into
one parotid and the
contralateral submandibular gland; control
group, 1.5 mL of saline
Drooling severity and
frequency scale,
saliva weight, DQ
Significant improvement
in all three measures
within 14 weeks,
except drooling
severity and frequency
scale and DQ at week
4 of BoNT-A
Small sample size with
statistical power only
69.5%
Alrefai et al
(2009) [39]
24 children
(21 months–
7 years) CP
Double-blind, placebocontrolled, randomized
clinical trial. Treatment
with BoNT-A 50 U into
each parotid; control
group, 0.5 mL of saline
Drooling severity and
frequency scale
Significant reduction in
median severity and
median frequency
scale in treatment
group
High dropout rate in the
placebo group with
the higher dose (70 U
into each parotid)
Reid et al
(2008) [40]
48 children
(6–18 years)
31 with CP
Placebo-controlled,
randomized clinical
trial. Treatment with
BoNT-A 25 U into each
parotid and submandibular
glands; control group, no
treatment
DrI
Significant difference
between treatment
and control groups
in DrI scale at
1 month up to
6 months
Measurement bias as the
placebo group
received no treatment
Wilken et al
(2008) [42]
30 children
(1–18 years)
12 children
with CP
Randomized, repeated,
open-label clinical trial.
Treatment with BoNT-A
total dose 80 u up to 100
u or serotype B 100 u/kg
up to 120 u/kg (parotid
and submandibular)
TDS
83% of all children
responded. No
significant difference
between serotypes
A and B
Five children developed
viscous saliva; only
50% of children
continued treatment
DQ, drooling quotient; DrI, Drooling Impact Scale questionnaire; TDS, Teacher Drooling Scale; VAS, visual analog scale.
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Management of sialorrhea in children with CP
malignancy in the irradiated field [46]. However, this risk is
minimal, particularly in patients with ALS, as these patients
have limited life expectancy.
option. In these situations, regional regulations regarding the
age of legal capacity and consent procedures must be
consulted, and physicians are urged to seek legal advice.
RT has been criticized and abandoned from use for the
pediatric population because of its long-term hazards of
growth retardation and risk of malignancy [48–50].
CONCLUSIONS
Drooling can be a significant source of functional and social
disability for a child with CP. Treatment of drooling is part of
the multidisciplinary care that these patients require, which
should consist of a pediatric neurologist, an otolaryngologist,
a pediatric dentist, and a speech pathologist. Treatment
recommendations are developed through a group decisionmaking process and are based on clinical evaluation of the
type and severity of drooling and associated structural or
neurodevelopmental problems. The parents and patients are
included in developing a stepwise plan of no immediate
treatment, intervention with oral motor techniques or
equipment, biofeedback, pharmacotherapy, or surgery.
There are no clinical trials comparing different treatment
options (e.g., pharmacotherapy vs surgery or pharmacotherapy vs behavioral treatment), making treatment guidelines
more difficult.
ETHICAL CONSIDERATIONS
From the above discussion on the treatment options for
drooling in children with CP, it is clear that, with the
exception of non-invasive behavior modification techniques,
adequate explanation of the advantages and disadvantages of
each treatment option is required, and treatment can only be
offered after appropriate consent is obtained. Obtaining
consent before providing care is both a fundamental part of
good practice and a legal requirement. The process of
obtaining consent will vary from simple situations such as
providing behavior modifications or administering oral
medications to more complex situations where a considerable
amount of information would be needed to support the
decision-making process, as is the case with surgical options
or botulinum toxin injections. There are unique issues
relating to obtaining consent from children under 16 years
of age. It is automatically assumed in these cases that
obtaining parental consent is sufficient. The authors stress
the importance of confirming that the consenting parent is
indeed the parent with legal parental responsibility as defined
by regional laws and regulations. Once children reach the age
of 16 years, they are considered to have reached the age of
legal capacity in most countries, and are considered to be able
to provide consent to medical interventions. However,
physicians are urged to encourage children between the ages
of 16 and 18 years to involve their families in decisionmaking, especially when the young person is making the
decision to refuse a certain treatment. In addition, in many
countries, for treatment decisions that are unlikely to have
grave consequences, even a young person under 16 years can
legally consent to treatment provided he or she is competent
to understand the nature, purpose, and possible consequences of the treatment proposed, as stated in the landmark
Gillick case [51]. Consent considerations are more complex
for children with CP, as many are not deemed competent to
make such decisions. However, a disabled child should never
automatically be presumed to be incapable of making
decisions regarding care, and many will be able to contribute
to the decision-making process if information is presented to
them appropriately and they are adequately supported during
the decision-making process. Even where children are not
able to give consent for themselves, it is very important to
involve them as much as possible in decisions about their
own health and care. Even very young children will have
opinions about their health and care, and methods appropriate to their age and understanding should be used to
enable these views to be taken into account.
Future long-term studies with a large, homogeneous patient
population should provide guidelines for the best treatment
approach for this problem.
Disclosure: The authors have nothing to disclose.
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