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Sialorrhea: a review of a vexing, often unrecognized sign of oropharyngeal and esophageal disease.

Saliva is produced by the major salivary glands (parotid, submandibular, and sublingual), as well as several smaller glands. Salivary flow can be altered by multiple entities. There is much written regarding xerostomia ("dry mouth"), the condition related to inhibited or decreased salivary flow. This condition is widely recognized in certain systemic diseases, particularly Sjögren syndrome, diabetes mellitus, after anticholinergic, antihistamine, and decongestant medications, as well as states of enhanced sympathetic drive, such as anxiety or emotional disturbances and various other psychosocial conditions. On the other hand, sialorrhea or ptyalism, the condition of increased salivary flow, is rarely discussed in the clinical literature. Sialorrhea can occur with various neurologic disorders, infections, the secretory phase of the menstrual cycle, heavy metal poisoning, Wilson disease, Angelman syndrome, as well as a relatively unknown condition called idiopathic paroxysmal sialorrhea. Normal salivation may be altered by drugs (such as clozapine, risperidone, nitrazepam, lithium, and bethanecol) that have a cholinergic effect that induces sialorrhea. This report focuses on sialorrhea as it relates to disorders of the oropharynx and esophagus. The patient typically recognizes a problem with excessive "foamy mucus" but does not understand its origin. Infections and obstruction are the most common oropharyngeal causes. Increased salivary flow occurs as a typically subtle manifestation of gastroesophageal reflux disease. This occurrence is referred to as water brash. Idiopathic achalasia and megaesophagus due to the parasite Trypanosoma cruzi are regularly associated with sialorrhea. Esophageal obstruction (foreign body, cancer, or stricture formation), infection, and nasogastric intubation are the more common conditions associated with the symptomatic sequelae of sialorrhea. Sialorrhea-related respiratory and pulmonary complications are greatest in those with a diminished sensation of salivary flow and hypopharyngeal retention. Extremes of age, the chronically debilitated, or those in chronic care facilities, especially associated with cerebrovascular accidents and esophageal cancer, typically comprise this population. For the patient with an intact awareness of saliva, sialorrhea can present with significant social stigmas. Occult drooling or regular oral evacuation into a tissue or "spit cup" is socially incapacitating. This report provides a review of the physiology, pathogenesis, clinical manifestations, and therapeutic options for sialorrhea. Physicians and other healthcare professionals should recognize the importance of sialorrhea as a possible indicator or complication of a variety of disease states of the oropharynx and esophagus as well as its impact on the patient's physical and social quality of life.

Animals↗

Long-term follow-up of submandibular duct rerouting for the treatment of sialorrhea in the pediatric population.

OBJECTIVE: To determine the long-term control of sialorrhea in children who underwent submandibular duct rerouting (SMDR) and to identify potential preoperative predictors of outcome. DESIGN: Retrospective chart review of children who underwent SMDR; information updated by discussion with the permanent caregiver. SETTING: Tertiary care center. PATIENTS: Children who had significant sialorrhea resulting from a variety of neuromuscular disabilities between January 1980 and December 1995. OUTCOME: We report the outcome on 59 patients who underwent SMDR for the treatment of sialorrhea. Patients were ascribed a preoperative sialorrhea and global neurologic deficit score. Postoperative outcome was scored as marked, moderate, no improvement, or worse. Twenty-eight of 59 (47.4%), 28 of 59 (47.4%), and 3 of 59 (5.1%) of the patients had preoperative sialorrhea scores of 3 (profuse), 2 (moderate), and 1 (mild), respectively. Twenty of 59 (33.9%), 29 of 59 (49.2%), and 10 of 59 (16.9%) had preoperative scores of 3 (severe), 2 (moderate), and 1 (mild) neurologic impairment, respectively. Mean time to follow-up of the 59 patients was 5.46 years. Postoperative improvement scores were as follows: 50.8% had marked, 28.8% had moderate, and 20% had no to minimal improvement in their sialorrhea. Two patients were transiently worse. A complication rate of 11.3% (9 of 79) was demonstrated: 7 ranulae, 1 transient swelling of the floor of the mouth, and 1 submandibular gland infection. The preoperative global neurologic deficit score was found to be more predictive of surgical outcome than sialorrhea score.

Adolescent↗

[Sialorrhea in patients with Parkinson. A six year prospective study].

INTRODUCTION: The sialorrhea has been recognised as one of the manifestations of Parkinson's disease, with a frequency of up to 80%. The evolution of patients with sialorrhea is unknown. PATIENTS AND METHODS: We study 26 male and 17 female patients with Parkinson's disease and sialorrhea. RESULTS: Evaluation at three years showed that sialorrhea had persisting in 18 of them, at six years showed that persisted in 8 patients. We found significant impairment in the sialorrhea group. CONCLUSION: If we accept the principle, that sialorrhea are levodopa-resistant disorders as non nigrostriatal symptoms, secondary to a diffuser and more extended cerebral stem degeneration, we could assume that in these patients with sialorrhea cortical-sub cortical circuits are more widely involved leading to motor and cognitive impairment and depression.

Aged↗

Validation of a new scale for the evaluation of sialorrhea in patients with Parkinson's disease.

Sialorrhea is common in Parkinson disease (PD), affecting approximately 70% to 75% of patients. Several tools for measuring saliva volume or production exist, but none are designed specifically for assessing sialorrhea-related discomfort. The objective of this study was to develop and validate a clinical scale for subjective evaluation of sialorrhea in PD. In Phase I, internal consistency of the Sialorrhea Clinical Scale for PD (SCS-PD) was established in 39 PD patients. In Phase II, scale validity was proven through saliva volume measurements obtained in 49 PD patients and 27 healthy volunteers. Internal consistency estimated using Cronbach's alpha was 0.78, indicating none of the original seven items tested needed to be removed. Twenty-one patients complaining of sialorrhea (63%) studied during Phase II, showed higher SCS-PD scores but no differences in saliva volume. SCS-PD scores showed significant correlation with saliva volume (r = 0.41; P = 0.004) and with total Unified Parkinson's Disease Rating Scale Part III (UPDRS III) scores (r = 0.70). Furthermore, saliva volume showed inverse relation to age in PD patients as well as in controls (r = -0.3 and r = -0.4; P < 0.05), but SCS-PD scores did not. The SCS-PD scale showed good internal consistency and validity, providing support for its use in routine clinical sialorrhea-related discomfort evaluation.

Adult↗

Botulinum toxin improves sialorrhea and quality of living in bulbar amyotrophic lateral sclerosis.

Sialorrhea is frequently a socially disabling symptom in patients with bulbar amyotrophic lateral sclerosis (ALS). In this open-label prospective study, we report the effect of botulinum toxin A (Botox) injection into the parotid glands in 10 patients with bulbar ALS and socially disabling sialorrhea. We applied three different outcome measures to determine the effect of Botox therapy on sialorrhea. Botox significantly improved the degree of sialorrhea and a drooling impact score and, by inference, the quality of living, in over half of the patients with bulbar ALS and severe sialorrhea. The beneficial effect of Botox lasted for at least 2 months in those who responded. No major adverse effects were noted. Local injection of a small dose of Botox into the parotid glands can control sialorrhea and potentially improve living quality in some patients with bulbar ALS.

Aged↗

Success and complications of four-duct ligation for sialorrhea.

OBJECTIVE: Sialorrhea can have a significant negative effect on quality of life, impeding social interactions and severely limiting employment opportunities. Many surgical options to control sialorrhea have been reported. One of the newest procedures is combined ligation of the submandibular and parotid ducts, reported only once in the literature in one series of five patients to date. We have compared results in our first 21 patients undergoing this procedure with results reported in the literature for other procedures to treat sialorrhea. MATERIALS AND METHODS: We retrospectively reviewed medical records of all 21 children we treated with four-duct ligation, a relatively simple intraoral procedure to control sialorrhea, between August 1999 and September 2000 and contacted primary caregivers by telephone to answer a questionnaire regarding objective and subjective results of surgery. Surgery was considered successful when caregivers rated patients as 'much better' or 'better' after surgery. RESULTS: Follow-up was completed in all 21 of the patients 1-14 months after surgery. The success rate of four-duct ligation ('much better' or 'better' after surgery) was 81%, and no patient's sialorrhea problem was worse after surgery. Major complications occurred in two (10%) of the patients (one ranula and one case of sialoadenitis), which were both successfully treated surgically. Minor complications occurred in four (19%) of the patients, tongue swelling that prolonged hospitalization, a ranula that resolved, and prolonged submandibular gland swelling that resolved (two cases). More than half of patients were discharged the day of or the day after surgery. CONCLUSIONS: Four-duct ligation should be considered when surgery is indicated to treat sialorrhea.

Follow-Up Studies↗

[Pharmacological therapy of sialorrhea in patients with motor neuron disease].

A comparative trial of amithryptiline and dysport (botulinic toxin type A) in the treatment of sialorrhea in patients with motor neuron disease (MND) was conducted in 10 MND patients with sialorrhea, of whom 5 were treated with subcutaneous injections of Dysport and 5 with Amithriptiline, and 6 controls without salivary dysfunction. Gravimetry and scintigraphy of salivary glands were used before and after treatment. Compared to controls, saliva production was significantly decreased in MND patients. Both amithryptilin and dysport used in mean therapeutic doses decreased sialorrhea with similar effect. However, 3 patients, receiving amythryptiline in dosage 50 mg/day, experienced side effects (constipation, accommodation disturbances, dry mouth, sleepiness and poor concentration). Reducing of amithryptiline dose, along with prescribing dysport, removed the side-effects in these patients, while sialorrhea did not increase. The authors concluded that due to high efficacy and low cost of amithryptiline therapy of sialorrhea proved to be a golden standard of palliative care in MDN. However, in these terms dysport can not be an alternative to amithryptiline in sialorrhea therapy. Nevertheless, in cases when amithryptiline treatment is accomplished with side-effects, the drug dosage can be reduced and combined with dysport.

Adrenergic Uptake Inhibitors↗

Treatment of sialorrhea with glycopyrrolate: A double-blind, dose-ranging study.

OBJECTIVE: To determine the safety and efficacy of glycopyrrolate in the treatment of developmentally disabled children with sialorrhea. DESIGN: Placebo-controlled, double-blind, crossover dose-ranging study. SETTING: Outpatient facilities in 2 pediatric hospitals. PATIENTS: Thirty-nine children with both developmental disabilities and excessive and bothersome sialorrhea. MAIN OUTCOME MEASURES: Parent and investigator evaluation of change in sialorrhea and adverse effects. RESULTS: Glycopyrrolate in doses of 0.10 mg/kg per dose is effective at controlling sialorrhea. Even at low doses, 20% of children may exhibit adverse effects severe enough to require discontinuation. CONCLUSIONS: Glycopyrrolate is effective in the control of excessive sialorrhea in children with developmental disabilities. Approximately 20% of children given glycopyrrolate may experience substantial adverse effects, enough to require discontinuation of medication. Arch Pediatr Adolesc Med. 2000;154:1214-1218.

Adolescent↗

Experience with the Wilkie procedure for sialorrhea.

Sialorrhea (drooling) is most commonly seen in children with cerebral palsy or mental retardation. Surgical procedures for the control of sialorrhea include salivary gland excision, parasympathetic nerve section, and salivary duct ligation and/or rerouting. Eighteen children between the ages of 5 and 17 years underwent bilateral submandibular gland excision and rerouting of Stensen's duct (Wilkie procedure). All children had severe drooling associated with cerebral palsy or mental retardation. Follow-up at 7 years showed satisfactory control of sialorrhea in 16 of 18 patients (89%). There was one major complication: xerostomia. Our results indicate that submandibular gland excision together with parotid duct retropositioning provides effective control of sialorrhea in most cases. Unfavorable head and mandibular posturing seemed to cause persistent sialorrhea in one case.

Adolescent↗

A double-blind placebo-controlled trial of botulinum toxin B for sialorrhea in Parkinson's disease.

BACKGROUND: Injections of botulinum toxin A are an effective treatment for sialorrhea in Parkinson's disease (PD). Based on the relatively high rates of dry mouth seen with botulinum toxin B, there is reason to suspect that it may also improve sialorrhea. OBJECTIVE: To determine whether botulinum toxin B (Myobloc; Elan Pharmaceuticals, New York, NY) is a safe and effective treatment for sialorrhea in patients with PD. METHODS: Demographics, PD treatments, head posture, the Unified Parkinson's Disease Rating Scale (UPDRS), two questionnaires regarding drooling, Visual Analogue Scale, global impressions, salivary gland imaging, and a dysphagia questionnaire were assessed in 16 PD subjects with problematic sialorrhea. Patients were then randomized to receive either botulinum toxin B (1,000 units into each parotid gland and 250 units into each submandibular gland) or a pH-matched placebo, using only anatomic landmarks. Patients returned 1 month later to undergo an identical assessment. RESULTS: Compared with placebo, those randomized to drug reported improvement on the Visual Analogue Scale (p < 0.001), global impressions of change (p < 0.005), Drooling Rating Scale (p < 0.05), and Drooling Severity and Frequency Scale (p < 0.001). There was no change in UPDRS, head posture, or Dysphagia Scale. Adverse events were mild and included dry mouth (three patients), worsened gait (two), diarrhea (one), and neck pain (one) in the botulinum toxin B group. CONCLUSION: Anatomically guided injections of botulinum toxin B into the parotid and submandibular glands appear to effectively improve sialorrhea without compromising dysphagia in patients with PD.

Aged↗

Sialorrhea - therapeutic drug options.

OBJECTIVE: To review the efficacy and safety of various drug treatments for sialorrhea. Pharmacotherapy for drug-induced sialorrhea is not addressed. DATA SOURCES: Clinical studies were identified using PubMed (1966-October 2001). Key search terms included sialorrhea and drug therapy. DATA SYNTHESIS: Sialorrhea is a social and physical detriment to patients. Drug treatment, although not necessarily the treatment of choice for all patients, can offer some symptom relief. CONCLUSIONS: Literature has documented that benztropine, glycopyrrolate, and scopolamine can reduce the incidence of sialorrhea. Although the literature evaluating the therapeutic options has limitations (e.g., small sample size, inconsistent outcome measurements), glycopyrrolate may have an advantage over the other agents due to fewer adverse effects.

Adolescent↗

Botulinum toxin A in the treatment of sialorrhea.

OBJECTIVE: To review the efficacy and safety of botulinum toxin A (BTX) in the treatment of sialorrhea. DATA SOURCES: A PubMed search and reference list review was performed to find clinical trials on BTX in the treatment of sialorrhea. The search was limited to English-language trials involving human subjects. Articles from 1965 to October 2006 were searched, using key words botulinum, botulinum toxin, Botox, sialorrhea, saliva, salivary gland, and drooling. STUDY SELECTION AND DATA EXTRACTION: Reports of 15 studies were found on BTX in the treatment of sialorrhea in human subjects. Four controlled trials are presented in detail, and 11 uncontrolled studies are summarized. DATA SYNTHESIS: One controlled trial found that BTX caused significant reductions in patients' severity and frequency of drooling compared with placebo. Another found significant saliva reductions in the treatment group receiving the highest dose of BTX but found no subjective improvement compared with the placebo group. A third study comparing BTX with placebo found significant improvements in subjective and objective measures of drooling in the treatment group. A fourth controlled trial found significant reductions from baseline levels of drooling after patients received BTX injections. Similar efficacy was found between the BTX and scopolamine treatment groups. CONCLUSIONS: Evidence indicates that BTX is a safe and effective treatment for sialorrhea. More research is needed to determine the ideal dose, injection location, and technique of BTX injections.

Botulinum Toxins, Type A↗

[Sialorrhea: hypersalivation and drooling. An underestimated problem in medical practice].

On the contrary to hyposalivation/xerostomia there is a little attention paid in the literature for discussing the problem of the patient having too much saliva. In most of cases, pathogenesis of sialorrhea is not known. Due to the small number of patients involved in the studies it is difficult to get exact data. In addition to that, there is no consensus either on the terminology of sialorrhea or a preferable method which should be used to achieve the best diagnostic and therapeutic approaches. Sialorrhea may occur in all age groups of population either as a consequence of increased salivary gland secretion or oral incontinence and impaired swallowing. Based on a recently published paper hypersalivation should be considered as the primary form of sialorrhea, and drooling corresponds to its secondary form. Each condition may seriously affect the physical activity, the functioning of different organs, communication, the psychosocial status of the patients. Based on a wide horizon of published data this review is focused on the terminology, methods of assessment, etiology, frequency, symptoms, and therapy of sialorrhea with special emphasis on its dental aspects.

Deglutition↗

Bilateral submandibular gland excision with parotid duct ligation for treatment of sialorrhea in children: long-term results.

BACKGROUND: Multiple procedures have been advocated for the surgical control of chronic sialorrhea in children. However, some of them are associated with significant complications or only short-term success. OBJECTIVES: To evaluate the safety of bilateral submandibular gland excision (SGE) with parotid duct ligation (PDL) and to assess its long-term complications and efficacy in the treatment of chronic sialorrhea in children. DESIGN: Case series. Telephone interview of patients' families. SETTING: Tertiary care children's hospital. PATIENTS: Ninety-three patients with chronic sialorrhea who underwent bilateral SGE with PDL from 1988 to 1997. MAIN OUTCOME MEASURES: Operative and postoperative complications, length of postoperative hospitalization, postoperative drooling, care requirements, xerostomia, dental caries, and overall satisfaction. RESULTS: The mean postoperative stay was 2.4 days. There were 3 postoperative complications. Seventy-two families were interviewed (follow-up time, 1-10 years): 62 (87%) reported no further drooling or significant improvement; 7 reported the occurrence of dry mouth; and 2 reported an increase in dental caries. CONCLUSION: Bilateral SGE with PDL is a safe and consistently efficient procedure for the treatment of chronic sialorrhea in children.

Chronic Disease↗

Surgical management of sialorrhea.

Sialorrhea is a distressing problem in children with cerebral palsy, in children with other neurologic disorders, and in normal children. The constant need for clothing or bib changes in handicapped patients frequently results in further social isolation and causes normal children to be viewed as "impaired." The surgical treatment of sialorrhea at Children's Hospital Medical Center, Cincinnati, Ohio, has evolved over the past 5 years. Since 1981, 70 surgical procedures have been performed for drooling at this institution; follow-up information is available on 52 patients. The surgical procedures performed included submandibular rerouting alone, submandibular duct rerouting with bilateral tympanic neurectomy with bilateral submandibular gland excision, and bilateral submandibular gland excision with bilateral parotid duct ligation. Our review indicates that bilateral submandibular gland excision with parotid duct ligation is the only procedure in which consistent control of sialorrhea can be expected. Postoperative complications after this procedure were minimal and only one parent complained that the child had a dry mouth after the procedure. Postoperative hospitalization after bilateral submandibular gland excision and bilateral parotid duct ligation was not significantly different from that required after any of the other procedures. In conclusion, this surgical procedure appears to be the most effective surgical treatment for sialorrhea and precludes the need for multiple operative procedures for this problem.

Humans↗

Is clonidine useful for treatment of clozapine-induced sialorrhea?

Clozapine has shown superior efficacy in treatment of refractory schizophrenia, but its use is limited by emergent side-effects. Among other adverse effects, sialorrhea is a troublesome side-effect, its stigmatizing nature results in poor treatment compliance. Several hypotheses have been put forward in the etiology of clozapine-induced sialorrhea. 2 adrenergic antagonism is hypothesized to be involved in its pathophysiology, based on the response to clonidine and lofexidine. Oral clonidine (50 to 100 g/day) was tried on 12 stable outpatients of schizophrenia maintained on clozapine. Wet area over the pillow as reported by the patients was recorded at baseline and at 4 weeks of treatment along with the subjective response after the treatment. Most of the patients reported a decrease in sialorrhea without any adverse events. We describe encouraging results in an open case series of oral clonidine for clozapine-induced sialorrhea.

Adrenergic alpha-Agonists↗

Sialorrhea: a management challenge.

Sialorrhea (drooling or excessive salivation) is a common problem in neurologically impaired children (i.e., those with mental retardation or cerebral palsy) and in adults who have Parkinson's disease or have had a stroke. It is most commonly caused by poor oral and facial muscle control. Contributing factors may include hypersecretion of saliva, dental malocclusion, postural problems, and an inability to recognize salivary spill. Sialorrhea causes a range of physical and psychosocial complications, including perioral chapping, dehydration, odor, and social stigmatization, that can be devastating for patients and their families. Treatment of sialorrhea is best managed by a clinical team that includes primary health care providers, speech pathologists, occupational therapists, dentists, orthodontists, neurologists, and otolaryngologists. Treatment options range from conservative (i.e., observation, postural changes, biofeedback) to more aggressive measures such as medication, radiation, and surgical therapy. Anticholinergic medications, such as glycopyrrolate and scopolamine, are effective in reducing drooling, but their use may be limited by side effects. The injection of botulinum toxin type A into the parotid and submandibular glands is safe and effective in controlling drooling, but the effects fade in several months, and repeat injections are necessary. Surgical intervention, including salivary gland excision, salivary duct ligation, and duct rerouting, provides the most effective and permanent treatment of significant sialorrhea and can greatly improve the quality of life of patients and their families or caregivers.

Humans↗

Parasympathetic nerve section for control of sialorrhea.

Twenty mentally retarded patients with a considerable drooling problem underwent transtympanic sectioning of selected preganglionic parasympathetic nerve fibers to the major salivary glands. Following surgery, cessation of trouble-some sialorrhea occurred in 15 to 20 patients; but by six months postoperatively, only ten patients remained controlled. The best results obtained were in a subgroup of five patients who underwent bilateral Jacobson nerve and chorda tympani nerve section. In four of these patients, sialorrhea was controlled after a minimum follow-up of two years. Parasympathetic denervation of the major salivary glands via a transtympanic approach is offered as a primary modality of therapy in those mentally retarded patients judged to have considerable sialorrhea. Sacrifice of both chorda tympani nerves appears to enhance the surgical results.

Adolescent↗