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Frey's syndrome, or gustatory sweating, is most commonly seen as a sequela of superficial parotidectomy. It can also be caused by other forms of trauma, including blunt trauma, but rarely does it occur without trauma.
Frey's syndrome is a phenomenon of hemifacial flushing and sweating after gustatory stimulus, usually secondary to surgical trauma over the parotid gland, although other injury mechanisms may be seen. It is accepted as a result of aberrant regeneration of facial autonomic nerve fibers. Treatment evolved from ineffective medical and surgical approaches to botulinum toxin. We evaluate the effectiveness and safety of botulinum toxin in the treatment of this complication in two patients.
Fourteen patients with severe Frey's syndrome (occurring after conservative parotidectomy) managed with intracutaneous injection of botulinum toxin type A were prospectively evaluated. Results were analyzed for effectiveness, complications, and adverse effects. Complications were not encountered. The only adverse effect noted was a temporary and slight partial paresis of the upper lip of 3 months' duration in 2 patients. Permanent paresis was not encountered. Frey's syndrome was always controlled within 2 days following the intracutaneous injection of botulinum toxin A. Frey's syndrome recurrence was not encountered with a follow-up duration that varied from 3 to 9 months (mean follow-up 7 months). This preliminary report confirmed that in patients who have Frey's syndrome after conservative parotidectomy, the intracutaneous injection of botulinum toxin is a valuable treatment option that should be further investigated.
Frey's syndrome or gustatory sweating is a common complication of parotid gland injury or surgery. Forty to 60 percent of the patients undergoing parotidectomy will develop manifestations of this syndrome but only 10-15 percent of these require treatment. To date tympanic neurectomy is the most efficacious method of treating the problem but relief is not obtained in all cases. Based on an experimental study in the dog, a new approach to the prevention of Frey's syndrome is presented. Since Frey's syndrome is due to the aberrent regeneration of parasympathetic nerve fibers of the parotid gland into the severed sympathetic nerve endings in skin overlying the gland, it is proposed that the insertion of a dermal graft barrier between the resected parotid gland and the skin will prevent Frey's syndrome. Histological sections of dermal grafts, buried for three to six months tend to confirm that this idea has some merit.
In 1990 the authors reported their preliminary study of the prevention of Frey's syndrome in 55 patients utilizing a superficial musculoaponeurotic system (SMAS) flap in parotid gland surgery. During the past 10 years, numerous studies have supported their original thesis that interposition of living tissue between the resected gland bed and the skin could prevent the development of this complication. The authors have expanded their own patient population and now consider this a definitive study on the prevention of Frey's syndrome. A total of 160 patients are presented with a follow-up period of 5 to 22 years. All patients underwent subtotal or total parotidectomy performed by one of the authors. A history was acquired and testing for Frey's syndrome (Minor's starch iodine test) was performed. As a result of this approach, and in spite of the intensive search for it, no cases of Frey's syndrome were encountered. The hoped-for secondary benefit of preventing the postparotidectomy retromandibular depression was somewhat less satisfactory, although most patients remain satisfied with their appearance. The debilitating symptoms in Frey's syndrome, which is reported to have an incidence of 5% to 50% in the typical parotidectomy patient, can be avoided with thoughtful preoperative planning. The authors favor an aesthetic incision followed by the development of an SMAS flap. The parotidectomy is then performed using the surgeon's preferred technique. The SMAS flap is then placed into the bed of the resected parotid gland. This institutes a protective tissue barrier guarding against the aberrant anastomotic communication between the postganglionic secretomotor fibers intended for the parotid gland, and the now adjacent sweat glands. Their patient population is large enough to provide significant evidence that Frey's syndrome can be prevented, compared with a meta-analysis of parotid patients in multiple other studies in the literature. Assuming the patient's history and pathology does not preclude its use, the SMAS flap should be considered the standard of care for preventing Frey's syndrome in the postparotidectomy patient. If the SMAS flap is not available, a temporoparietal fascial flap has proved to be a good alternative.
Frey's syndrome of the external auditory canal is extremely rare. A 55-year old woman presented with a 6 month history of unilateral gustatory otorrhea. She never complained of hearing impairment, tinnitus, vertigo or otalgia. No trauma or surgical signs were evident near the ear or parotid gland. Examination of the ear showed an intact tympanic membrane without disease. A diagnosis of gustatory sweating syndrome was suggested by the observation of sweat production after chewing and by Minor's starch-iodine test. Symptoms were relieved after tympanic neurectomy. The pathogenesis, differential diagnosis and treatment options are discussed.
Frey's syndrome is characterized by recurrent episodes of facial gustatory flushing and sweating (most frequent in adults) limited to the cutaneous distribution of the auriculotemporal nerve which, when injured, shows abnormal regeneration. The condition is relatively common in adults following nerve injury in parotid surgery but has rarely been reported in children as a sequel of perinatal birth trauma resulting from forceps assisted delivery. We report three patients with the same syndrome and two different causes. Two children had a history of forceps assisted delivery and one adolescent had undergone preauricular lymphadenectomy. This syndrome, which has more often been described by allergists and maxillofacial surgeons than by pediatricians, may pose problems of differential diagnosis with food allergy, leading to unnecessary exclusion diets. In the pediatric age group, Frey's syndrome is self limiting and does not require therapy if the diagnosis is correct.
Several therapeutic approaches exist to treat gustatory sweating (Frey's syndrome) following parotidectomy. Because of the lack of effective treatment, a new therapeutic modality using botulinum toxin injections was presented previously by our group. The duration of the demonstrated positive effect was essentially unknown so far. Based on our experiences using this technique since December 1993, the purpose of this clinical investigation was to make an up-to-date report and demonstrate the duration of effect of BOTOX injections in patients with severe gustatory sweating. Nineteen patients with severe gustatory sweating have been treated with BOTOX by intracutaneous injections into the affected skin areas. The maximal follow-up time was 33 months. The results were obtained by interviews and controls using Minor's starch iodine test. In all treated cases (n = 19 patients, 22 treated sides) gustatory sweating ceased completely within 2 days. Side effects were absent. In 12 patients gustatory sweating reappeared. The mean duration of effect was 17.3 months (subjective personal communication of 18 patients). Findings show that intracutaneous injection of BOTOX is a highly effective, safe, and minimally invasive treatment of Frey's syndrome with long-lasting therapeutic effect.
Botulinum toxin (BTX) is a safe and reliable drug for the treatment of Frey's syndrome and hypersialorrhea. Currently, the intradermal injection of BTX is the standard treatment of Frey's syndrome. A disappearance of Frey's syndrome for 1 to 1.5 year could almost be promised. Recently, BTX was introduced for the treatment of hypersialorrhea. Although some questions exist about the optimal dosage and the type of major salivary glands that should be treated, hypersialorrhea was reduced in almost all affected children and adults for up to 6 months by using ultrasound-guided intraglandular BTX injections.
CONCLUSIONS: Advanced age, long operation time and large specimen volume were significant risk factors for transient facial palsy after conservative parotidectomy. Revision operation was the only risk factor for development of a permanent palsy. Risk factors for Frey's syndrome were not found. The incidence of Frey's syndrome was not altered by the use of a sternocleidomastoid muscle flap or other implantation material. OBJECTIVES: The results of studies identifying risk factors for facial palsy and Frey's syndrome after parotidectomy are contradictory. This study attempted to identify these risk factors by performing a retrospective review of a large series of patients. PATIENTS AND METHODS: A total of 610 standardized conservative parotidectomies for benign diseases performed between 1989 and 2004 were studied retrospectively. The risk factors for facial palsy and Frey's syndrome were determined by univariate and if possible by multivariate analysis of variables related to patient demographics and operation characteristics. RESULTS: The rates of transient facial palsies, permanent facial palsies and Frey's syndrome were 18%, 4%, and 4%, respectively. Significant univariate and multivariate risk factors for development of a transient facial palsy were age>70 years, operation time>260 min, and a specimen volume>70 cm3. The only significant risk factor for the development of a permanent palsy was prior surgery. A significant risk factor for the development of Frey's syndrome could not be estimated.
We report on an unusual case of Frey's syndrome. A 45 year old male patient presented with gustatory sweating 14 years after parotidectomy. The sialography showed a post-parotidectomy status without any pathologic findings. The MRI also showed a normal rest-parenchyma of the parotid gland without any pathologic findings. Minor's starch iodine test [14], confirmed the gustatory sweating in the left preauricular area. Frey's syndrome can appear with a latency of 14 years postoperative or post-trauma. The pathogenic mechanism and the curative therapy for this auriculotemporal-syndrome are still unsolved.
PURPOSE: The purpose of this retrospective study was to evaluate the incidence of Frey's syndrome after parotid gland surgery with elevation of a skin flap versus the interposition of musculoaponeurotic tissue between the skin and the facial nerve. MATERIALS AND METHODS: The standard skin flap was used in 122 patients (1981 to 1989), whereas interposition of temporoparietal galea or superficial musculoaponeurotic system was performed in 146 consecutive patients (1989 to 1998). RESULTS: The incidence of Frey's syndrome was reduced from 33% to 4% with the use of a musculoaponeurotic layer, and the mean duration of postoperative facial nerve paresis decreased from 3 months to 6 weeks. CONCLUSIONS: Consideration should be given to the use of an interpositional layer of tissue between the facial nerve and the skin flap to reduce the incidence of Frey's syndrome after parotid gland surgery.
AIM: To identify the severity of Frey s syndrome and its response to botulinum toxin type A. METHODS: Minor test was performed in all cases to assess the extent of the affected area, using the contralateral side as control. Severity was assessed according to the proposal of Luna-Ortiz et al. Response was evaluated after 3 and 6 months, and was compared with the basal data. RESULTS: Frey s syndrome was documented in 38 patients, but only 23 cases accepted the botulinum toxin type A treatment. Severity was moderate in 8 (35%) and severe in 15 (65%) cases. Mean applied dose was 1.41 MU/cm2 in 21 patients (91%), whereas one patient was treated with 10 MU for a 0.8 cm2 affected area (12.5 MU/cm2) and another patient with 10 MU for a 0.5 cm2 affected area (20 MU/cm2) due to severity of their symptomatology. Average affected area at the beginning was 14.2 cm2, while after 3 and 6 months of treatment it was 4.1 cm2 and 4.4 cm2 respectively (p<0.001). The two patients that received higher doses of botulinum toxin A had complete response. Complete response was observed in 13 patients (56.5%) at 3 months, but in only nine (39%) this lack of symptomatology persisted at 6 months. In three cases (13%) no response was obtained at 3 months, and the application of an additional dose of botulinum toxin type A produced no response in two of them after 6 months. Comparison of the severity score of the average basal value vs. that obtained at 3 and 6 months revealed a significant difference (p<0.05); however, no statistically significant difference was found when comparing outcome at 3 vs. 6 months. There were no statistically significant differences using the independent samples test when comparing outcome after treatment in relation to gender, type of surgery, or use of postoperative radiation therapy (p>0.05). In conclusion, botulinum toxin A remains as the treatment of choice for Frey s syndrome.
OBJECTIVE: Objective quantification of Frey's syndrome (gustatory sweating), following total parotidectomy. A biosensoring method of enzymatic electrodes enabling the detection of L-lactate on intact skin with the use of a skin extraction device and enzymatic electrodes is presented and analyzed. DESIGN: A criterion standard study. SETTING: This prospective trial was undertaken at our research laboratory (University of Paris [France]). Parotidectomy was performed in our department, which is a tertiary care center for parotid gland pathology. PATIENTS: Twenty-eight patients with gustatory sweating following total parotidectomy and nine control patients not operated on were asked to take part in this prospective study. MAIN OUTCOME AND MEASURES: Gustatory sweating was assessed in all patients using a clinical scale, the Minor starch iodine test, and the L-lactate biosensoring method. RESULTS: Instrumentation and assay procedure for the L-lactate biosensoring method are detailed. Statistical analysis of data was performed using the Kruskal-Wallis H Test and the Mann-Whitney U Test. Results demonstrate that this method enables objective measurement of the L-lactate on skin without the need for chemical reagents, continuous nondestructive analysis in real time, and physiological dynamic monitoring of the L-lactate rate of production after stimulus. Data achieved strongly suggested that the aberrant regeneration theory is the main clue to Frey's syndrome pathogenesis. CONCLUSION: This safe, reliable, noninvasive, objective, and highly sensitive method provides an investigative tool for clinicians as well as physiologists involved with patients presenting gustatory sweating following parotid gland surgery.
BACKGROUND: Gustatory sweating, the main symptom of Frey's syndrome, usually occurs after parotid gland surgery. Numerous medical and surgical treatments have been proposed to treat this condition, but there are various drawbacks. Botulinum toxin type A intracutaneous injection is a relatively new treatment modality; its use has never been reported in Taiwan. Here, we present our preliminary experience with this technique and review the literature. METHODS: Between March 2002 and June 2005, 10 consecutive patients with severe gustatory sweating were managed with intracutaneous injection of botulinum toxin type A for a total of 16 times. The affected skin was visualized by Minor's starch-iodine test and then recorded. The interinjection distance was 1 cm and a mean dose of 46.4 U (at a concentration of 2.5 U/0.1 mL) was used. RESULTS: In all 10 cases, gustatory sweating improved within 2 days after injection, with no side effects. Patients in whom the first 13 injections were performed experienced recurrence of gustatory sweating. Mean duration of effectiveness was 9.3 months; the shortest duration of effectiveness was 2 months and the longest was 28 months. One patient also had gustatory flushing, but this symptom did not improve even after 3 treatments. CONCLUSION: Intracutaneous injection of botulinum toxin type A is a highly reliable, effective, safe, and minimally invasive treatment for gustatory sweating. Some patients had long-lasting therapeutic results. We recommend it as a valuable treatment option for severe cases of gustatory sweating. However, in our experience, it had no effect on facial skin flushing. Therefore, in addition to acetylcholine, there might be other neurotransmitters that are responsible for skin vasodilatation.
Gustatory sweating, also known as Frey's syndrome, is a recognized phenomenon after parotidectomy. The incidence of clinically significant gustatory sweating may be dependent on the thickness of the skin flap that is elevated at the time of surgery. Dissection in the subcutaneous level or the deeper sub-superficial musculoaponeurotic system level are 2 commonly used techniques in the operation. Twenty-eight patients were prospectively evaluated for Frey's syndrome after superficial parotidectomy. Thirteen patients underwent subcutaneous and 15 patients sub-superficial musculoaponeurotic system flap elevation. The overall incidence of gustatory sweating was determined, and a comparison of the two surgical techniques was assessed subjectively by patient interview-questionnaire and objectively evaluated with starch-iodine testing.
The recent trend in management of Frey's syndrome has been the use of prophylactic procedures performed at the time of parotidectomy to prevent its symptoms postoperatively. An additional benefit of this approach is the prevention of the typical cheek contour deformity after parotidectomy. We reviewed our experience with interposition of a vascularized temporoparietal fascial flap between the parotid bed and overlying skin immediately after complete superficial parotidectomies to prevent Frey's syndrome and contour defects. The results of seven consecutive attempts revealed it to be an effective technique, achieving both goals in all patients with minimal morbidity.
Parotidectomy may be associated with a significant depression in the retromandibular region and a significant incidence of gustatory sweating (Frey's syndrome). Superiorly and inferiorly based sternocleidomastoid flaps and posterior plication of the superficial musculoaponeurotic system were evaluated for their ability to ameliorate both consequences. Sixteen patients with sternocleidomastoid flaps and 16 patients with superficial musculoaponeurotic system plication were compared to a control group of 104 patients. The incidence of Frey's syndrome was 47.1% in the control group, 12.5% (P = 0.025) in the sternocleidomastoid flap group, and 0% (P = 0.005) in the superficial musculoaponeurotic system plication group. The surgical techniques are described. The prevalence of Frey's syndrome is discussed with respect to age, sex, radiation therapy, and the type of parotidectomy performed. The indications and contraindications of the three surgical techniques are described.