Vol. 101 No.

January 2006

ORAL MEDICINE

Editor: Martin S. Greenberg

Drooling of saliva: A review of the etiology and management options

Jean-Paul Meningaud, MD, PhD, FEBOMS,a Poramate Pitak-Arnnop, DDS, OMS,b Luc Chikhani, MD,c and Jacques-Charles Bertrand, MD,d Paris, France
UNIVERSITY OF PIERRE & MARIE CURIE (PARIS 6)

Drooling of saliva appears to be the consequence of a dysfunction in the coordination of the swallowing mechanism, resulting in excess pooling of saliva in the anterior portion of the oral cavity and the unintentional loss of saliva from the mouth. Drooling can produce signicant negative effects on physical health and quality of life, especially in patients with chronic neurological disabilities. Various approaches to manage this condition have been described in the literature, including oral motor therapy, behavior modication via biofeedback, orofacial regulation therapy, drug therapy, radiotherapy, and surgical treatments. Minimally invasive modalities, such as injection of botulinum toxin, photocoagulation, and acupuncture, have also been reported. This article provides a comprehensive and thorough overview of drooling, with an emphasis on understanding its etiologies and modalities of treatment. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:48-57)

Drooling of saliva is dened as the inability to control oral secretions. This condition is not due to excessive production of saliva, but is rather a problem in the coordinated control mechanism of orofacial and palatolingual musculatures. Even patients who produce less saliva can suffer from drooling (eg, patients with Parkinsons disease).1,2 This impaired neurological control results in poor swallowing function and leads to excessive pooling of saliva in the anterior portion of the oral cavity and the unintentional loss of saliva from the mouth. Hypersalivation does not necessarily lead to drooling. Drooling can be highly distressful for neurologically impaired patients and their parents or caregivers; the risk of social rejection, constant wetness of clothing,
This work was supported by a grant-in-aid 2005/2006 n04 for scien tic research from the Fondation des Gueules Cassees. a Consultant Maxillofacial Surgeon, Department of Maxillofacial ` Surgery, Teaching Pitie-Salpetriere Hospital, Paris, France. b Surgical fellow, Department of Maxillofacial Surgery, Teaching ` Pitie-Salpetriere Hospital, Paris, France. c Consultant Maxillofacial Surgeon, Department of Maxillofacial ` Surgery, Teaching Pitie-Salpetriere Hospital, Paris, France. d Professor, head of the department, Head Professor, Department of ` Maxillofacial Surgery, Teaching Pitie-Salpetriere Hospital, Paris, France. Received for publication Apr 7, 2005; returned for revision Jul 20, 2005; accepted for publication Aug 17, 2005. 1079-2104/$ - see front matter 2006 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2005.08.018

and physical discomfort adds further burden to the special attention required by these patients. Besides its cosmetic effects, drooling can impair masticatory function, interfere with speech, favor perioral infections, particularly by Candida albicans, and result in loss of uid, electrolytes, and proteins, deteriorating the quality of life of the patient. The inability to swallow adequately also increases the risk of aspiration pneumonia. The aim of this article is to provide a comprehensive and thorough overview of drooling with an emphasis on understanding the causal mechanisms and modalities of treatment. MATERIALS AND METHODS Articles, from 1966 onward, were identied with Medline using key words (drooling and sialorrhea) and the limits function. Only original clinical studies written in English and French were retained for review. Articles were selected keeping in mind the specic experience of each author. For the table summarizing the literature on salivary duct and gland procedures, data were gathered only from series including more than 10 patients and using an assessment tool. Many systems have been advocated for assessment of drooling. The following are just a few examples. To obtain an objective measurement, saliva produced spontaneously during a 5-minute period can be collected from the mouth. The drooling quotient (DQ)3,4 is a validated, semiquantitative, direct observational method. The

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presence or absence of drooling is assessed every 15 seconds during two 10-minute periods separated by a 60-minute break. The success of surgical treatment is generally assessed with the criteria described by Wilkie and Brody:5 outcome is considered excellent if salivary control is apparently normal, good if there is a slight loss of saliva, fair if drooling is improved but with signicant residual loss of saliva, and poor if no signicant control is observed. Successful surgical removal includes procedures with excellent or good outcomes. Physiology of swallowing Swallowing is a physiological process which can be initiated voluntarily but is thereafter under reex control. It begins with tactile stimulation of pharyngeal receptors that send impulses to the integrative areas for swallowingcalled swallowing centers in the medulla and pons.6 Motor output from this center, transmitted via the trigeminal, facial, glossopharyngeal, vagus, accessory, and hypoglossal nerves, controls the sequential peristaltic coordination of pharyngeal and upper esophageal muscles that contract during swallowing. Descending inputs from cortical and subcortical centers can initiate or regulate swallowing.7 Etiologies of drooling Causes of drooling are summarized in Table I. Drooling is a physiological phenomenon in infants, which usually resolves after 15-18 months of age as a result of the maturation process of the orofacial motor function and the coordination of swallowing; it has been dened as an abnormality in a child more than 4 years of age in the awake status.8 Drooling is a relatively common clinical sign; for instance, the Oxford Feeding Study estimated that 28% of children with neurological impairment suffer from continuous drooling.9 According to Tahmassebis10 survey, 58% of children with cerebral palsy have a drooling condition, which is severe in 33% of them. As noted in Hysons survey,11 46.5% of parkinsonian patients complained about drooling, 18.8% of whom felt that their drooling was socially disabling. Even in the early phases of Parkinsons disease, 15% of patients suffer from nocturnal drooling.12 Management of drooling A multidisciplinary team is indispensable for appropriate assessment and management of drooling. Any aggravating problem, for example, signicant dental disease, abnormal head position leading to abnormal salivary ow with gravity, severe malocclusion, airway obstruction, or certain drug effects, must be recognized and treated or relieved.

Table I. The etiology of drooling

Causes of drooling Neurological decits Cerebral palsy Motor neuron disease, notably amyotrophic lateral sclerosis (ALS) Facial paralysis Cerebrovascular accidents Seizures Parkinsons disease Congenital suprabulbar palsy Severe mental retardation; Down syndrome and patients with physical and/or learning disabilities Worster Drought syndrome Landau Kleffner syndrome Encephalitis Anglemans syndrome Hydrocephalus Hypoxic encephalopathy Freeman-Sheldon syndrome Moebius syndrome Idiopathic Major resection of oropharynx Adverse drug reaction: clozapine Early sign of Sjogrens syndrome Painful swallowing caused by infectious diseases: primary herpes, Coxsackie virus, etc. Oral/dental problems Malocclusion Resorbed dental ridge Tongue thrust Constant open mouth and poor lip control (lip incompetency) Anesthesia or hypoesthesia of lips Congenital or acquired deformities of tongue

Nasal obstruction Hypoactive gag reex Gastro-esophageal reex Head posture and sitting position Concentration on a task/ degree of concentration Emotional state

Depending on the modality used, treatment of drooling can potentially complicate oral health. Such effects, notably the impact of decreased salivation, must always be taken into account. Saliva contributes signicantly to oral health. It functions as a buffer and a source of ions used for remineralization of teeth. Complications such as gingivitis, burning sensation of the mucous membrane, rampant caries, rapid tooth destruction, cheilitis, commissure ssuring, tongue and palate crusting, and occasionally paresthesia of the tongue or mucous membrane should be addressed. Regular dental examination for caries is recommended for all patients, and uoride supplementation might be helpful.

Nonsurgical methods for treating drooling problems Oral motor therapy. Oral motor programs aim to develop oral skills such as sucking, lip closure, and tongue and jaw movement. The speech therapist plays a crucial role in evaluating the existing oral motor

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Table II. Success and major complications of duct ligation/relocation and/or gland removal procedures for drooling
Reference (yr) Ekedahl (1974) Wilkie and Brody (1977)5
41

Duct relocation SM 3 2 P32

Duct ligation SL 3 2

Gland removal SM 3 2

Success (%) 9/11 (81%) 106/123 (86%)

Major complications (cases, %) Hematoma (1/11), total: 9% Duct stenosis or cyst (25/123), oral/ dental problems (9/123), transient parotid swelling (5/123), wound dehiscence (3/123), septic parotitis (1/123), total: 35% Cyst (2/6), transient swelling (1/6), total: 50% in the relocation group, 0% in the ligation group TMJ ankylosis (1/14), but relationship not sure Ranula (2/18), total: 11% Ranula (2/25), oor of mouth infection (1/25), swelling requiring operation (1/25), total: 16% Hematoma (1/24), wound infection (1/24), total: 8% Ranula (15/194), lateral cervical cyst (4/194), total: 10% Wound infection (2/58), hematomas (2/58), parotid duct stula (1/58), parotitis (1/58), total: 10% 1 aspiration pneumonia, total: 6% Ranula (1/18), xerostomia (1/18), transient parotid swelling (1/18), total: 16% Retention cyst requiring operation (2/20), resolved retention cyst (2/20), 6 month swelling (1/20), total: 25% Transient swelling (6/14), ranula (1/14), total: 50% Parotid cyst (2/13), septic parotitis (1/13), xerostomia (1/13), total 38% (Wilkie group) mucoid cyst (2/5), total: 40% (SM ducts relocation group) Minor infection (6/39), ranula (5/39), increase in dental carries (5/39), dry mouth (9/39), aspiration (2/39), total: 69% Ranula (4/54), aspiration pneumonia (3/54), tooth extraction (3/54), total: 18% Transient swelling (6/20), wound infection (1/20), substantial bleeding (1/20), total: 40% Ranula (7/79), SM gland infection (1/79), transient swelling (1/79), total: 10% Ranula (7/71), transient airway obstruction (1/71), total: 11% Dry, chapped lips (4/35), periodontal disease (2/35), ranula (2/35), persistent swelling (2/35), lower lip droop (1/35), total: 31%

Glass et al. (1978)42

Compare P 3 2 ligation versus P 3 2 relocation SM 3 2 SM 3 2 P32

SM 3 2

Better results in the ligation group 12/14 (85%) 18/18 (100%) 24/25 (96%)

Dundas and Peterson (1979)43 Guerin (1979)44 Cotton and Richardson (1981)45 Shott et al. (1989)47 Crysdale and White (1989)48 Brundage and Moore (1989)49 O Dwyer et al. (1989)50 Rosen et al. (1990)51

SM 3 2

SM 3 2

P32 P32

SM 3 2 SM 3 2

21/24 (87%) 95/107 (89%) 50/58 (86%)

SM 3 2 P32

SM 3 2

13/16 16/18 (89%)

Burton et al. (1991)52

SM 3 2

17/20 (85%)

Varma et al. (1991)53 Jacquinet et al. (1993)54

SM 3 2

P32

13/14 (92%) Results slightly better in the Wilkie group

Compare Wilkie procedure and SM ducts relocation

Webb et al. (1995)55

SM 3 2

P31

Drooling quotient improved in 100% (N = 28) 94% of parents satised (N = 53)

O Dwyer and Conlon56 (1997) Ethunandan and Macpherson (1998)57 Mankarious et al. (1999)58 Wilson and Henderson (1999)59 Panarese et al. (2001)60

SM 3 2

SM 3 2

SL 3 2

16/19 (84%)

SM 3 2

47/59 (80%) mean time to follow-up = 5.46 years 12/16 (75%) 47/49 (96%) Short term: 82% long-term: 76% (N = 35)

SM 3 2 SM 3 2 SM 3 2

P31

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Table II. Continued

Reference (yr) Crysdale et al. (2001)
61

Duct relocation SM 3 2

Duct ligation

Gland removal

Success (%) Improvement of scores statistically similar (106 and 115 patients)

Major complications (cases, %) Additional surgery for persistent drooling (23/226), ranula (20/226), lateral neck cyst (7/226), total: 22% Additional surgery for persistent drooling (6/249), total: 2.5% Hematoma (1/93), transient swelling (1/93), parotitis (1/93), dry mouth (7/72), increase in dental caries (2/72), total: 15% Ranula (5/56), oral/dental problems (4/56), secondary hemorrhage (1/56), chest infection (1/56), total: 19% Sialadenitis (3/23), ranula (1/23), persistent swelling (1/23), total: 23% Ranula (1/21), sialadenitis (1/21), total: 10%

SM 3 2 Stern et al. (2002)62

P32

SL 3 2 SM 3 2 62/72 (87%)

De et al. (2003)63

SM 3 2

SL 3 2

49/56 (87%)

Uppal et al. (2003)64

SM 3 2

SL 3 2

20/23 (87%)

Shirley et al. (2003)65

P 3 2 and SM 3 2

17/21 (81%)

SM, submandibular gland/duct; SL, sublingual gland/duct; P, parotid gland/duct.

skills of the patient. Harris and Dignam13 had positive results in cerebral-palsied children participating in a training program using mirrors, games, relative competitiveness, and praise reinforcements by the physiotherapist, combined with a chin cup appliance to apply pressure on the chin to achieve an appropriate anterior oral seal. Behavioral modication via biofeedback. Biofeedback techniques condition the patient to swallow at the sound of an auditory stimulus. Koheil et al.14 reported the success of a training program using an auditory electromyography (EMG) feedback with electrodes placed on the orbicularis oris muscle. It was considered that patients must be old enough, have relatively good intellectual functions, be auditory-prompted, be reasonably well motivated, present only a moderate drooling problem, and not become oblivious to the auditory device or its signal. Subsequently, these techniques were not widely diffused in clinical practice. Orofacial regulation therapy. A functional appliance, employed according to the principles of CastilloMorales, has been used successfully in the management of drooling. It consists of an acrylic palatal plate with vestibular and lingual stimulators. The vestibular stimulator, formed by varying the depth of the ridge, stimulates the lip seal. The lingual stimulator is a median button with a central hole that induces sucking, and subsequently, tongue retrusion.15,16 This appliance is very helpful for patients with hypotonic perioral musculature and protruding tongue as commonly seen in Downs and Moebius syndromes.17 Limbrock et al. found 68%

improvement in 53 patients with Downs syndrome,18 and 72% improvement in cerebral-palsied patients with severe drooling.19 When this method does not stop the drooling, it can at least reduce it. Combination with other methods remains possible. The appliance is not well accepted by children less than 4 years old. Drug therapy. Salivation is mainly mediated through parasympathetic stimulation. Acetylcholine is the active neurotransmitter, binding at muscarinic receptors in the salivary glands. Thus, cholinergic muscarinic receptor antagonists such as atropine, scopolamine (hyoscine), or glycopyrronium bromide can be used to treat drooling. These drugs are contraindicated when there is a history of cardiac problems, closure glaucoma, prostate hypertrophy, paralytic ileus, or pyloric obstruction. Besides, the result is often incomplete with considerable individual variation sometimes leading to high doses with signicant side effects such as excessively dry mouth, constipation, urinary retention, blurred vision, irritability, confusion, and even toxic psychosis,20 which pose greater risks to the health of the patient than sialorrhea itself. Sublingually administered atropine reduces drooling.21,22 Sublingual delivery has many advantages. In contrast with botulinum toxin, the drug administered in the form of ophthalmic drops is readily available. Atropine is inexpensive, does not require any specialized skill for use, and unlike surgical removal, has a reversible effect. However, it is contraindicated in patients with cognitive impairment, dementia, and hallucination. Hyson et al.21 delivered 1 drop of atropine

52 Meningaud et al. (1% wt/vol solution containing 0.5 mg) sublingually twice a daily. Scopoderm transdermal therapeutic system (TTS) is a self-adhesive dermal patch delivering scopolamine, usually applied to prevent nausea associated with motion sickness. Dryness of the mouth is its most common side effect. It has been evaluated in patients who suffer from drooling induced by clozapine,23 brain injury,24 cerebral palsy,25,26 and major resection of the oropharynx.27 The lesser peak-concentration side effects are a specic advantage. Potential side effects include a localized dermal reaction.28 Zeppetella29 successfully used scopolamine via nebulization in 3 patients, 2 of whom had not improved with the transdermal form. With nebulized delivery, scopolamine is absorbed faster and can be used on an as required basis. In addition, nebulized scopolamine might be helpful for patients with problematic bronchial secretion. Recently, Jongerius et al.30 performed a systematic review to investigate the efcacy of anticholinergic drugs in the treatment of drooling in children with multiple handicaps. Although they reviewed the literature from 1966, only 7 articles t their chosen methodological criteria. Based on their study, there was some evidence that at least some anticholinergic drugs (benztropine, glycopyrrolate, and benzhexol hydrochloride) are effective, but the authors were unable to conclude that one drug is preferable. Radiotherapy. Borg and Hirst31 advised that radiotherapy should be avoided in children because of the risk of inducing malignancy, growth retardation, xerostomia, mucositis, radiation caries, and osteoradionecrosis. Conversely, in elderly patients with a limited life expectancy, these long-term side effects would not be expected to develop. After reviewing their results, the authors concluded that the desired response, with minimal discomfort of patients and without causing signicant morbidity, can be expected with ve 4-Gy fractions to a total dose of 20 Gy using 9-18 MeV electrons prescribed to the 100% isodose, encompassing both the parotid and submandibular glands with ipsilateral elds. The development of thick secretion may be avoided by sparing the upper part of parotid glands in the radiation eld; reducing total dose and dose per fraction might prevent temporomandibular joint brosis. Andersen et al.32 revealed their success in treating the severe drooling of 18 amyotrophic lateral sclerosis patients with a life expectancy of less than 2 years. Irradiation of the larger part of the parotid glands and the posterior part of the submandibular glands with 7.0-7.5 Gy in a single fraction reduced drooling without producing permanent xerostomia, except in 1 patient. Findings reported by Stalpers and Moser supported their results.33

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Surgical methods for treating drooling problems Neurectomy. Sectioning the parasympathetic neural pathway reduces the ow of saliva. The tympanic plexus and the chorda tympani nerves can be sectioned,34,35 either unilaterally or bilaterally, and either alone or in combination with other procedures such as submandibular gland removal.36 Chorda tympani neurectomy reduces the salivary ow rate of the submandibular/ sublingual complex, but it seems to be a poor method when used alone.37 Hearing loss is a possible complication. This method would thus be contraindicated in patients who already have hearing problems, except for those with total hearing loss. Since chorda tympani neurectomy always produces a loss of taste in the anterior two thirds of the tongue,38 it seems unethical to take away the enjoyment of taste from neurologically defenseless patients who have so few pleasures in life, just to control drooling.39 Despite an initially high success rate, the long-term results of neurectomies used alone are relatively disappointing.40 Besides, it must be stressed that drooling is not hypersalivation; neurectomies do not directly correct the cause. Salivary duct and gland procedures. Table II summarizes the success and major complications of salivary duct and gland procedures. 41,5,42-65 One of the earliest reports on surgical management of drooling concerned bilateral parotid duct relocation from the buccal vestibule in the area of the maxillary second molar to the tonsillar fossa, or the posterior part of the tonsillar pillar, to initiate the swallowing reex, accompanied by bilateral submandibular sialoadenectomy.66 Bilateral duct ligation of the parotid glands combined with submandibular gland removal has been proposed as a simpler procedure with good outcome (85%-86% success) in 3 studies totaling 96 patients.43,47,49 The purpose of duct ligation is to obtain gland atrophy. Submandibular duct relocation performed alone or in combination is a common procedure. The success rate has been good in all reported studies, ranging from 75% to 89%. This technique offers the following advantages: scarless procedure, relatively low complication rate, potential reversibility, and the physiological features. Saliva, in contact with the base of tongue, can initiate a swallowing reex. Many modications have been advocated. Ekedahl41 reported the combination of submandibular duct relocation with sublingual duct ligation. Adam et al.67 associated relocation of both parotid ducts for the treatment of 3 drooling children. After an experimental study,68 Ozgenel and Ozcan69 associated bilateral parotid-duct diversion using autologous venous grafts in order to eliminate the risk of ductal stenosis. Wilson and Henderson coupled the surgical procedure with unilateral parotid duct ligation.59

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Excision of sublingual glands at the same time reduces the occurrence of ranula.61 Tonsillectomy can be indicated to reduce the risk of obstruction of the relocated ducts. Attention must be paid to a potential increase of caries in the mandibular incisal and canine area.70,71 Klem and Mair72 advocated a technique consisting of ligation of both the parotid and submandibular ducts. Their success rate was 100% in 5 patients, without any major complications. With a larger case series, Shirley et al.65 reported a success rate of 81% with this very simple procedure. Submandibular duct relocation seems to be the physiological procedure to initiate the swallowing reex. In patients with a history of aspiration pneumonia, procedures that decrease salivary output, are more advisable. Minimally invasive methods for treating drooling problems Injection of botulinum toxin. The more signicant studies on botulinum toxin serotype A in the treatment of drooling are gathered in Table III.73-86,4,87-89 Xerostomia is one of the rst manifestations of botulism. This led the medical community to investigate an application to sialorrhea and drooling. When injected in the salivary gland, botulinum toxin serotype A (BTX/A) inhibits acetylcholine release in the cholinergic nerve endings, mainly at neurosecretory junctions. The BTX/A binds to a protein called SNAP25, forming a complex that impairs neuronal exocytosis by inhibiting fusion of the presynaptic vesicles containing the neurotransmitter.90 An initial animal model showed a signicant reduction in saliva production without direct toxicity to the acinar cells.91 Although only pilot studies with relatively small groups of patients are available (Table III), it is interesting to consider that the outcome is uniformly favorable, with very rare reports of serious complications. Up to two thirds of patients experienced a marked or moderate improvement of drooling after the treatment of both parotid glands or parotid and submandibular glands combined. The injection is made percutaneously, and ultrasound guidance improves efcacy and safety.85,87 For young children, general anesthesia may be necessary. Transductal injection is not recommended.92 The parotid gland is usually injected at 2 or 3 sites, and the submandibular gland at 1 to 3 sites. Generally, the duration of action varies from 6 weeks to 6 months. The reported side effects include weakness of adjacent muscles producing difculty of chewing and swallowing due to BTX diffusion into masseter or pharyngeal muscles, risk of aspiration, facial weakness, and local infection. Other potential side effects are hematoma, salivary duct calculi, local injuries of the carotid arteries or branches of the facial nerve, and recurrent jaw

dislocation.93 A randomized controlled trial84 testing 3 different doses of BTX/A stressed the fact that higher doses reduce drooling more, but the highest safe dose is still unknown. Besides, we do not know the effect of repeated injections of BTX/A over time, or the risk of developing antibodies. Botulinum toxin serotype B (BTX/B) displays different pharmacological properties than type A; success has also been documented in drooling patients with Parkinsons disease.94,95 Photocoagulation of salivary ducts. With the aim to minimize surgical complications, Chang and Wong96 used Nd:YAG laser (1064 nm) for intraductal laser photocoagulation of bilateral parotid ducts, at 7 or 10 watts during 10 seconds, in 48 cerebral-palsied patients. Under general anesthesia, operating time ranged from 25 to 65 minutes. The concepts of laser-tissue interaction of intraductal laser photocoagulation are based on partial destruction of the parotid gland and occlusion of the parotid ducts. Signicant improvement in drooling severity and frequency was measured in the majority of cases. Postoperatively, transient facial swelling was seen in all patients. The complications were 1 hematoma, 2 infections, and 2 cystic formations. Moreover, laser photocoagulation of submandibular ducts may be indicated in individuals with poor results or recurrent drooling. This technique is a simple and effective modality for reducing drooling in cerebral-palsied patients, but its objective is to decrease saliva ow leading to the mentioned complications. Tongue acupuncture. Based on a cohort study, Wong et al.97 assessed daily tongue acupuncture applied to 5 specic acupoints for the treatment of drooling in 10 children with severe physical and/or cognitive disability and who were unable to comply with a formal behavioral modication program or oral-motor training program. Thirty sessions were administered. The authors hypothesized that acupuncture would stimulate the rich neural network in the tongue, which is connected to salivary glands and tongue muscles via the cranial nerve nuclei, and improve salivary secretion and swallowing mechanisms. Children easily tolerated the treatment with signicant improvement of drooling and no complication. This technique may be an alternative or adjunctive option for children with intractable drooling. However, the technical skill and experience of the practitioners is a marked obstacle for this technique. Besides, larger case series with longer follow-up and quantitative assessment are expected. Optimal treatment The physician may feel disoriented with, on the one hand, so many causes of drooling with very different pathogenic mechanisms, and on the other hand, so

54 Meningaud et al. Table III. Studies on botulinum toxin serotype A (BTX/A) in the treatment of drooling
Reference (yr) Bhatia et al. (1999)
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No. patients 4 5 9 5

Diagnosis PD1, PSP1, Dy1, MND1 PD5 PD9 MND5

BTX/A dose (mouse unit) 10 or 20 (Dysport) 10 (BOTOX) 7.5-15.5 (BOTOX) 6-20 (BOTOX) 5 (BOTOX) 15-40 (BOTOX)

Sites of injection Each parotid Each parotid Each parotid Each parotid Each submandibular Each parotid

Technique Blind Blind Blind Blind

Jost (1999)74 Pal et al. (2000)75 Giess et al. (2000)76

Porta et al. (2001)77

10

MND4, PD2, CP1, PT1, PS1, SSPE1

US

10-15 (BOTOX) Jongerius et (2001)78 Friedman and Potulska (2001)79 Suskind and Tilton (2002)80 Bothwell et al. (2002)81 Ellies et al. (2002)82 Ellies et al. (2003)83 3 11 22 9 4 13 CP3 PD11 CP22 (children) CP, neuro decits (children) CA2, stroke1, PS1 CA8, neurodegenerative 4, stroke1 15, 20 and 25 U (BOTOX) 5 (BOTOX) 20-40 (BOTOX) 10-30 (BOTOX) 5 (not specied) 55-65 (BOTOX) in total 22.5 (BOTOX)

Each submandibular Each submandibular Each parotid Parotid Submandibular Each parotid

US Blind US Blind

Parotid, US submandibular Each parotid US

10 (BOTOX) Lipp et al. (2003)84 32 ALS12, PD12, MSA4, corticobasal degeneration 4 PD14 (idiopathic) MSA6

Mancini et al. (2003)85

20 double-blind, placebo-controlled

18.75, 37.5, 75 (BOTOX) doseeffectiveness analysis 150 (Dysport) Each parotid 75 (Dysport)

Each submandibular Each parotid Blind

US

Kahl et al. (2004)86 Jongerius et al. (2004)4 Dogu et al. (2004)87 Hassin-Baer (2004)88

Clozapine1

45 controlled clinical CP45 (children) trial 15 PD15 9 CP6, other neurologic disorders 3 CP21

150 IU (not specied) 15, 20 and 25 (BOTOX) 15 (BOTOX) 10-25 (BOTOX)

Each submandibular Each parotid Not specied Each US submandibular Each parotid Blind/US technique analysis Each parotid US

Savarese (2004)89

21

15 (BOTOX)

Each parotid

Blind (but with electromyographic needle)

One mouse unit of BOTOX is equivalent to ;2.5 to 4 units of Dysport. CA, carcinoma; CP, cerebral palsy; CZP, clozapine-induced hypersalivation; Dy, dystonia; MND, motor neuron disease; MSA, multiple system atrophy; PD, Parkinsons disease; PS, primary sialorrhea; PSP, progressive supranuclear palsy; PT, post traumatic encephalopathy; SSPE, subacute sclerosing panencephalitis; US, ultrasound.

many treatments available. Nevertheless, bearing in mind just a few therapeutic recommendations is sufcient to help most patients. The rst step is to correct the many situational factors that may worsen the drooling of the patient, such as abnormal head position, airway problems, unnecessary

medications, severe malocclusion, and signicant dental disease, and better yet, if possible, to correct the cause.98 Thereafter, it is important, whenever feasible, to propose the more physiological treatments like oral motor therapy, behavior modication, or orofacial regulation therapy. When this is impossible, unsuccessful, or not

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17. de Serpa Pinto MVX, de Magalhaes MHCG, Nunes FD. Moebius syndrome with oral involvement. Int J Paediatr Dent 2002;12: 446-9. 18. Limbrock GJ, Fischer-Brandies H, Avalle C. Castillo-Morales orofacial therapy: treatment of 67 children with Down syndrome. Dev Med Child Neurol 1991;33:296-303. 19. Limbrock GJ, Hoyer H, Scheying H. Drooling, chewing and swallowing dysfunctions in children with cerebral palsy: treatment according to Castillo-Morales. ASDC J Dent Child 1990;57:445-51. 20. Sennhauser FH, Schwarz HP. Toxic psychoses from transdermal scopolamine in a child. Lancet 1986;2(8514):1033. 21. Hyson CH, Johnson AM, Jog MS. Sublingual atropine for sialorrhea secondary to Parkinsonism: a pilot study. Mov Disord 2002; 17:1318-20. 22. Comley C, Galletley C, Ash D. Use of atropine eye drops for clozepine-induced hypersalivation. Aust N Z J Psychiatry 2000; 34:1033-4. 23. Gaftanyuk O, Trestman RL. Scopolamine patch for clozepineinduced sialorrhea. Psychiatr Serv 2004;55:318. 24. Dreyfuss P, Vogel D, Walsh N. The use of transdermal scopolamine to control drooling. A case report. Am J Phys Med Rehabil 1991;70:220-2. 25. Siegel LK, Klingbeil MA. Control of drooling with transdermal scopolamine in a child with cerebral palsy. Dev Med Child Neurol 1991;33:1013-4. 26. Brodtkorb E, Wyzocka-Bakowska MM, Lillevold PE, Sandvik L, Saunte C, Hestnes A. Transdermal scopolamine in drooling. J Mental Dec Res 1988;32:233-7. 27. Talmi YP, Finklestein Y, Zohar Y, Lurian N. Reduction of salivary ow with Scopoderm TTS. Ann Otol Rhinol Laryngol 1988;97:128-30. 28. Lewis DW, Fontana C, Mehallick LK, Everett Y. Transdermal scopolamine for reduction of drooling in developmentally delayed children. Dev Med Child Neurol 1994;36:484-6. 29. Zeppetella G. Nebulized scopolamine in the management of oral dribbing: three case reports. J Pain Symp Manage 1999;17: 293-5. 30. Jongerius PH, van Tiel P, van Limbeek J, Gabreels FJM, Rotteveel JJ. A systematic review for evidence of efcacy of anticholinergic drugs to treat drooling. Arch Dis Child 2003; 88:911-4. 31. Borg M, Hirst F. The role of radiation therapy in the management of sialorrhea. Int J Radiation Oncology Biol Phs 1998; 41:1113-9. 32. Andersen PM, Gronberg H, Franzen L, Funeg U. External ard radiation of the parotid glands signicantly reduces drooling in patients with motor neurone disease with bulbar paresis. J Neurol Sci 2001;191:111-4. 33. Stalpers LJA, Moser E. Results of radiotherapy for drooling in amyotropic lateral sclerosis. Neurology 2002;58:1308-10. 34. Arnold HG, Gross CW. Transtympanic neurectomy: a solution to drooling problems. Dev Med Child Neurol 1977;19:509-13. 35. Mullins WM, Gross CW, Moore JM. Long-term follow-up of tympanic neurectomy for sialorrhoea. Laryngoscope 1979;89: 1219-23. 36. Sellars SL. Surgery of sialorrhoea. J Laryngol Otol 1985;99: 1107-9. 37. Grewal DS, Hirandani NL, Rangwalla ZA, Sheoda JH. Transtympanic neurectomies for control of drooling. Auris Nasus Larynx 1984;11:109-14. 38. Grant R, Miller S, Simpson D, Lamey PJ, Bone I. The effect of chorda tympani section on ipsilateral and contralateral salivary secretion and taste in man. J Neurol Neurosurg Psychiatry 1980;52:1058-62. 39. Wilkie TF. On the bad effects of nerve sections for the control of drooling. Plast Reconstr Surg 1977;60:94-7. 40. Parisier SC, Blitzer A, Binder WJ, Freidman WF, Marovitz WF. Tympanic neurectomy and chorda tympanectomy for the control of drooling. Arch Otolaryngol 1978;104:273-7. 41. Ekedahl C. Surgical treatment of drooling. Acta Otolaryngol 1974;77:215-20.

sufcient, it is suitable to propose drug therapy. When these means have proven ineffective, more aggressive treatments are indicated, giving preference to the more reversible treatments like botulinum toxin. When surgical management is required, preference should be placed on the more conservative and physiological approaches such as rerouting the submandibular ducts. If the patient has a short life expectancy, more aggressive treatment such as radiotherapy or four-duct ligation may be indicated, depending upon the specic clinical status of the affected individual and the severity of the problem. Whatever the chosen treatment, it is mandatory to maintain regular oral examinations to prevent or to treat complications related to decreased salivation.
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65. Shirley WP, Hill JS, Woolley AL, Wiatrak BJ. Success and complications for four-duct ligation for sialorrhea. Int J Paediatr Otorhinolaryngol 2003;67:1-6. 66. Wilkie TF. The problem of drooling in cerebral palsy: a surgical approach. Can J Surg 1967;10:60-7. 67. Adam P, Billet J, Bennani F, Schmidt J. Traitement de lincontinence salivaire. Proposition dune nouvelle technique chirurgi cale et resultats preliminaires. Rev Stomatol Chir Maxillofac 1994;95:213-8. 68. Ozgenel GY, Ozcan M, Kahveci Z. An experience study of bilateral repositioning of the Stensens duct orices with the autologous vein and artery grafts in dog. Br J Plast Surg 2000;53: 106-8. 69. Ozgenel GY, Ozcan M. Bilateral parotid-duct diversion using autologous vein grafts for the management of chronic drooling in cererbral palsy. Br J Plast Surg 2002;55:490-3. 70. Arnrup K, Crossner CG. Caries prevalence after submandibular duct retroposition in drooling children with neurological disorders. Paediatr Dent 1990;12:98-101. 71. Andlin-Sobocki P, Arnrup K, Bensch J, Gertzen H, Wranne L. Submandibular duct retropostion reduces drooling but may cause caries in lower front teeth. Dev Med Child Neurol 1992;34:556. 72. Klem C, Mair EA. Four-duct ligation. A simple and effective treatment for chronic aspiration from sialorrhea. Arch Otolaryngol Head Neck Surg 1999;125:796-800. 73. Bhatia KP, Munchau A, Brown P. Botulinum toxin is a useful treatment in extensive drooling of saliva. J Neurol Neurosurg Psychiatry 1999;67:697. 74. Jost WH. Treatment of drooling in Parkinsons disease with botulinum toxin. Mov Disord 1999;14:1057-9. 75. Pal PK, Calnes DB, Calnes S, Tsui JKC. Botulinum toxin A as treatment for drooling saliva in PD. Neurology 2000;54:244-7. 76. Giess R, Naumann M, Werner E, Rieman R, Beck M, Puls I, et al. Injections of botulinum toxin A into the salivary glands improve sialorrhea in amyotrophic lateral sclerosis. J Neurol Neurosurg Psychiatry 2000;69:121-3. 77. Porta M, Gamba M, Bertacchi G, Vaj P. Treatment of sialorrhoea with ultrasound guided botulinum toxin type A injection in patients with neurological disorders. J Neurol Neurosurg Psychiatry 2001;70:538-40. 78. Jongerius PH, Rotteveel JJ, van den Hoogen F, Joosten F, van Hulst K, Gabreels FJ. Botulinum toxin A: a new option for treatment of drooling in children with cerebral palsy. Presentation of a case series. Eur J Pediatr 2001;160:509-12. 79. Friedman A, Potulska A. Quantitative assessment of parkinsonian sialorrhea and results of treatment with botulinum toxin. Parkinsonism Relat Disord 2001;7:329-32. 80. Suskind DL, Tilton A. Clinical study of botulinum A toxin in the treatment of sialorrhea in children with cerebral palsy. Laryngoscope 2002;112:73-81. 81. Bothwell JE, Clarke K, Dooley JM, Gordon KE, Anderson R, Wood EP, et al. Botulinum toxin A as a treatment for excessive drooling in children. Pediatr Neurol 2002;27:18-22. 82. Ellies M, Laskawi R, Rohrbach-Volland S, Arglebe C, Beuche W. Botulinum toxin to reduce saliva ow: selected indications for ultrasound-guided toxin application into salivary glands. Laryngoscope 2002;112:82-6. 83. Ellies M, Laskawi R, Rohrbach-Volland S, Arglebe C. Up-to-date report of botulinum toxin therapy in patients with drooling caused by different etiologies. J Oral Maxillofac Surg 2003;61:454-7. 84. Lipp A, Trottenberg T, Schink T, Kupsch A, Arnold G. A randomized trial of botulinum toxin A for treatment of drooling. Neurology 2003;61:1279-81. 85. Mancini F, Zangaglia R, Cristina S, Sommaruga MG, Martignoni E, Nappi G, Pacchetti C. Double-blind, placebo-controlled study to evaluate the efcacy and safety of botulinum toxin type A in the treatment of drooling in parkinsonism. Mov Disord 2003;18: 685-8. 86. Kahl KG, Hagenah J, Zapf S, Trillenberg P, Klein C, Lencer R. Botulinum toxin as an effective treatment of clozepine-induced salivation. Psychopharmacol 2004;173:229-30.

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87. Dogu O, Apaydin D, Sevim S, Talas DU, Aral M. Ultrasoundguided versus blind intraparotid injections of botulinum toxinA for the treatment of sialorrhoea in patients with Parkinsons disease. Clin Neurol Neurosurg 2004;106:93-6. 88. Hassin-Baer S, Scheuer E, Buchman AS, Jacobson I, Ben-Zeev B. Botulinum toxin injections for children with excessive drooling. J Child Neurol 2005;20:120-3. 89. Savarese R, Diamond M, Elovic E, Millis SR. Intraparotid injection of botulinum toxin A as a treatment to control sialorrhea in children with cerebral palsy. Am J Phys Med Rehabil 2004;83: 304-11. 90. Breidenbach MA, Brunger AT. Substrate recognition strategy for botulinum neurotoxin serotype A. Nature 2004;432:925-9. 91. Shaari CM, Bei-Lian WU, Biller HF, Chuang S-K, Sanders I. Botulinum toxin decreases the salivation from canine submandibular glands. Otolaryngol Head Neck Surg 1998;118:452-7. 92. Winterholler MG, Erbguth FJ, Wolf S, Kat S. Botulinum toxin for the treatment of sialorrhea in ALS: serious side effects of transductal approach. J Neurol Neurosurg Psychiatry 2001;70: 417-8. 93. Tan EK, Lo YL, Seah A, Auchus AP. Recurrent jaw dislocation after botulinum toxin treatment for sialorrhoea in amyotrophic lateral sclerosis. J Neurol Sci 2001;190:95-7. 94. Racette BA, Good L, Sagitto S, Perlmutter JS. Botulinum toxin B reduces sialorrhea in Parkinsonism. Mov Disrod 2003;18: 1059-61.

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Reprint requests: Jean-Paul Meningaud, MD, PhD Service de Chirurgie Maxillo-Faciale ` CHU Pitie-Salpetriere 47, Bd de lHopital 75651 Paris cedex 13, France meningaud@noos.fr