Presentación infrecuente de síndrome de Frey bilateral, tratado con toxina botulínica / An unusual presentation of bilateral Frey syndrome treated with botulinum toxin

El síndrome de Frey o síndrome auriculotemporal, incluye sudoración facial al estímulo gustatorio, más frecuentemente después de cirugía de la glándula parótida. Describimos el caso de un síndrome de Frey bilateral, después de una cirugía unilateral de la glándula parótida, y su tratamiento exitoso con toxina botulínica tipo A. Se trata de una mujer de 54 años de edad, remitida para tratamiento a nuestro servicio de rehabilitación por referir sudoración en la mejilla durante la masticación, 2 años después de cirugía de la glándula parótida. En la valoración con el test de iodo/almidón, se observó un área de sudoración en ambas mejillas, mayor en la homolateral a la cirugía. Ambas áreas fueron tratadas con toxina botulínica, 50 unidades en cada una con un total de 100 unidades reconstituidas en 2 ml de solución salina estéril. A los 6 meses y al año de seguimiento, la paciente sigue asintomática. Nuestra paciente presentaba una forma inusual de síndrome de Frey y fue tratada con éxito con toxina botulínica. No hemos encontrado casos similares en la literatura de aparición en la edad adulta. La patofisiología del síndrome queda todavía por esclarecer (AU)

Frey syndrome, also known as auriculotemporal syndrome, includes facial gustatory sweating, mainly after surgery of the parotid gland. We describe a case of bilateral Frey syndrome after unilateral surgery and its subsequent successful treatment with type A botulinum toxin. A 54-year-old woman was referred for treatment to our physical medicine and rehabilitation department 2 years after parotid gland surgery complaining of sweating while eating in her ipsilateral cheek. When the affected area was assessed with the iodine and starch test, a hyperhydrotic area was revealed in both cheeks, which was greater in the ipsilateral cheek to surgery. Both areas were treated with botulinum toxin, 50 units in each, with a total of 100 units reconstituted with 2 ml sterile saline solution. Injections were performed following recommended protocols every 1 cm2. At 6 months and 1 year of follow up, the patient was asymptomatic. Our patient had an unusual form of Frey syndrome and was successfully treated with botulinum toxin. To the best of our knowledge, this is the first reported case of bilateral Frey syndrome beginning in adulthood. Further research is warranted to clarify the pathophysiology of this syndrome (AU)

Intraparotid Location of the Great Auricular Nerve: A New Anatomical Basis for Gustatory Sweating Syndrome.

BACKGROUND: Gustatory sweating syndrome (also known as Frey syndrome or auriculotemporal nerve syndrome) is thought to result from a lesion of the auriculotemporal nerve. A lesion of this nerve can lead to aberrant regeneration of nerve fibers to the sweat glands and blood vessels. The occurrence of signs outside the region of the auriculotemporal nerve prompted the author to search for another anatomical basis for this syndrome. METHODS: The author dissected 46 great auricular nerves from their origin to the parotid gland and in the infratemporal fossa. The author investigated the different connections of the great auricular nerve with the facial nerve and the auriculotemporal nerve. RESULTS: The great auricular nerve was found to essentially be a parotid nerve. There was a set of intraparotid nerve connections on 14 of the 46 half-heads that were dissected. The author was able to discern three types of parotid great auricular nerve connections, which he designates as either type 1, connection with the trunk of the facial nerve and its branches; type 2, connection with the auriculotemporal nerve; or type 3, connection with the auriculotemporal nerve and the facial nerve with the formation of an intraparotid nerve circle. CONCLUSIONS: Having clearly established the nerve connections of the great auricular nerve, the author believes that it is primarily this nerve that is responsible for gustatory sweating syndrome. This allows for a better understanding of the sympathetic nervous system features and the manifestation of the syndrome outside the region of the auriculotemporal nerve.

Galvanic skin response test: a new quantitative diagnostic method for Frey syndrome.

OBJECTIVE: Frey syndrome is one of the most common complications following parotid surgery. The current most common test for objectively diagnosing Frey syndrome is Minor starch-iodine test. This test might be insufficient because its results are not quantitative and therefore tests with quantitative results are investigated. The objective of this study was to investigate the efficiency of galvanic skin response (GSR) test, which measures changes in skin resistance, as a method with quantitative results for diagnosis of Frey syndrome. METHODS: Thirty patients who underwent superficial parotidectomy were assessed postoperatively (mean, 24.7 ± 25.7 months; range, 6-109 months). Patients completed a symptomatic evaluation questionnaire and underwent Minor starch-iodine test and GSR. RESULTS: Diagnostic validity of GSR test was found to be >2.91 following analysis. Sensitivity and specificity of this value were 100% and 55%, respectively, based on symptomatic assessment. Sensitivity and specificity were 87.5% and 57.1%, respectively, based on Minor starch-iodine test. CONCLUSIONS: When compared to symptomatic evaluation of patients who underwent superficial parotidectomy, GSR test was shown to be 100% sensitive in diagnosing Frey syndrome and quantitative results of GSR test could determine severity of Frey syndrome.

Outcome, general, and symptom-specific quality of life after various types of parotid resection.

OBJECTIVES/HYPOTHESIS: To document the outcome and impact on general and symptom-specific quality of life (QOL) after various types of parotid resection. STUDY DESIGN: General and symptom-specific QOL assessment at least 1 year after performed surgery. Retrospective data and outcome analysis of patients. METHODS: Between 2004 and 2010, 353 parotid resections in 337 patients were conducted at the Department of Otorhinolaryngology, University Teaching Hospital, St. Mary's Hospital Gelsenkirchen, Gelsenkirchen, Germany. A total of 196 patients fit the inclusion criteria and were available for postoperative evaluation. The general QOL assessment was based on both the global health status and global QOL scales of the European Organisation for Research and Treatment of Cancer (EORTC) Quality-of-Life Questionnaire in 34 patients. Symptom-specific QOL was assessed with the Parotidectomy Outcome Inventory-8 (POI-8). In addition, aesthetic outcome was evaluated with an ordinal scale. RESULTS: Outcome of parotidectomies in benign disease has little impact on general QOL and global health status. However, hypoesthesia or dysesthesia, Frey's syndrome, and cosmetic discontent are quite common and may affect symptom-specific and general QOL. Correlation with extent of surgery and statistically significant differences of patient evaluation for aesthetic outcome, sensory impairment, and Frey's syndrome between various types of limited parotid surgery (enucleation, extracapsular dissection, partial superficial parotidectomy) and superficial parotidectomy could be shown. CONCLUSIONS: An adequate parotid resection technique must be chosen to achieve the least disturbing outcome. In addition, in our patient collective, there was no increased recurrence rate found after limited parotid resection for pleomorphic adenoma or cystadenolymphoma.

Lucja Frey (1889­1942): life destroyed by the Holocaust--on the 70th anniversary of her death.

Lucja Frey was the first to explain the pathogenesis of the auriculotemporal syndrome, and the syndrome is recognized today as the Frey syndrome. Patients with this disease are subjected to paroxysmal paraesthesia of half of the face combined with sweating and redness. This syndrome can be found in the differential diagnosis of contemporary dermatology. Among others, it is differentiated from food allergies. The life and scientific career of Lucja Frey was brutally interrupted by the tragic times of the Holocaust.

Management of Frey syndrome using botulinum neurotoxin: a case report.

Frey syndrome is manifested clinically by hemifacial flushing and sweating after a gustatory stimulus. Frey syndrome is usually secondary to traumatic injury in the parotid region and is thought to be the result of misdirected re-sprouting of damaged autonomic nerve fibres. In this case report, we highlight the clinical and psychosocial aspects of Frey syndrome from a patient"s perspective, outline the pathophysiology of the condition and current management strategies, and describe the use of botulinum neurotoxin in the treatment of Frey syndrome.

Marathon of eponyms: 6 Frey syndrome (Gustatory sweating).

The use of eponyms has long been contentious, but many remain in common use, as discussed elsewhere (Editorial: Oral Diseases. 2009: 15; 185). The use of eponyms in diseases of the head and neck is found mainly in specialties dealing with medically compromised individuals (paediatric dentistry, special care dentistry, oral and maxillofacial medicine, oral and maxillofacial pathology, oral and maxillofacial radiology and oral and maxillofacial surgery) and particularly by hospital-centred practitioners. This series has selected some of the more recognized relevant eponymous conditions and presents them alphabetically. The information is based largely on data available from MEDLINE and a number of internet websites as noted below: the authors would welcome any corrections. This document summarizes data about Frey' syndrome.

Thoracoscopic sympathectomy for palmar and axillary hyperhidrosis: four-year outcome and quality of life after bilateral 5-mm dual port approach.

BACKGROUND: During recent years, thoracoscopic sympathectomy has been the standard treatment for hyperhidrosis. Different surgical techniques have been described without proving their advantages compared with other procedures. This study was designed to evaluate our modification of thoracoscopic sympathectomy and to compare the effectiveness between axillary and palmar hyperhidrosis. METHODS: Ninety patients with axillary or palmar hyperhidrosis who underwent bilateral thoracoscopic sympathectomy with single-lumen ventilation with a dual 5-mm port approach were followed up for a median of 3.9 (range, 1-6) years. The clinical course and data during the hospitalization and consultation in our outpatient clinic were reviewed. The following parameters were evaluated: clinical improvement, satisfaction, changes in quality of life, and compensatory sweating and gustatory sweating. RESULTS: The perioperative mortality was 0, and the morbidity was 6.5%. In 81% clinical improvement of sweating was noticed; 55% did not sweat at all. A total of 88% of patients were satisfied with the result of the operation. The rates of compensatory sweating and gustatory sweating were 93.5% and 49.4%, respectively. The result of sympathectomy in patients with palmar hyperhidrosis were significantly better concerning rate of satisfaction (p = 0.006) and improvement of symptoms (p = 0.027) compared with patients with axillary symptoms. Additionally it was found that the compensatory sweating had significantly impacted the satisfaction rating of the operation. CONCLUSION: Currently different effective surgical approaches for the treatment of hyperhidrosis with improvement rates of more than 80% are available. The quality of the intervention has to be evaluated by changes in quality of life and intensity of compensatory sweating. Thoracoscopic sympathectomy as performed in our institution offers results and complications comparable to previously published trials; however, because of single-lumen ventilation the management is much easier. Therefore, this technique offers an interesting option for the treatment of patients with palmar and axillary hyperhidrosis.

Síndrome auriculotemporal (síndrome de Frey) en dos lactantes con presentación bilateral / Auriculotemporal nerve syndrome (Frey syndrome) in two infants with bilateral presentation

El síndrome auriculotemporal (síndrome de Frey) se caracteriza por episodios de enrojecimiento facial, sudoración o ambos, localizados en el territorio de distribución del nervio auriculotemporal, y que ocurren como respuesta a estímulos gustativos. Cuando se manifiesta en la infancia y coincide con la introducción de nuevos alimentos en lactantes, se puede llegar al diagnóstico erróneo de alergia alimentaria. Presentamos dos lactantes (de 5 y 6 meses) con enrojecimiento facial bilateral en el área temporal, extendiéndose hasta el borde oral; ambos manifestaron los síntomas inmediatamente a la toma de fruta (y posteriormente tras un tipo de golosina en uno de los niños) con desaparición de cuadro instantes después del cese de la toma. Los dos niños eran fruto de un parto eutócico. En uno de ellos, los síntomas desaparecieron antes del año, mientras que el segundo aún tiene 7 meses y presenta episodios de intensidad variable. Es conveniente reconocer este síndrome para evitar exploraciones innecesarias y restricciones dietéticas improcedentes (AU)

Patients with auriculotemporal nerve syndrome (Frey syndrome) have facial flushing, sweating, or both located along the distribution of the auriculotemporal nerve, and that occurs in response to gustatory stimuli. As the condition usually manifests during infancy with the introduction of solid food, it can lead to the misdiagnosis of food allergy by physicians unfamiliar with this syndrome. Two boys (5 and 6 months old) presented with a history of bilateral facial flushing extending from the corner of the mouth to the temporal area. Symptoms began at the moment of fruit introduction (orange, pear, apple and banana) and also with some type of candy in the first. Symptoms appeared immediate to fruit mastication and cutaneous erythema spontaneously disappeared in a few minutes after finishing fruit. Both cases had been born without instrumental delivery. In one case the symptoms disappeared before the end of the first year of age, and the second (he?s now 7 months old) has symptoms discontinuously. The recognition of this entity might avoid unnecessary referral and laboratory evaluation (AU)

Síndrome de Frey tratamiento con toxina botulínica tipo A botox®: reporte de dos casos / Frey syndrome treatment with botulinum toxin type A botox: report of two cases

Evaluar la efectividad del uso de la toxina botulínica tipo A Botox® en el síndrome aurículo-temporal (síndrome de Frey). Se realizó un estudio experimental en dos pacientes femeninas con síndrome de Frey a quienes se les realizó: parotidectomía superficial bilateral por sialodenitis crónica en una, y parotidectomía superficial derecha en la otra, por carcinoma de células acinares, para objetivar el síndrome se utilizó el test de Minor y se uso toxina botulínica tipo A Botox®, aplicándola por vía subcutánea, usando una dosis de 3.0 MU/0.1ml/cm2. Se realizó el test de Minor en dos pacientes con síndrome de Frey. El tiempo de aparición de los síntomas fue: de 6 meses del lado izquierdo y 12 meses en el lado derecho para la que presentaba síndrome bilateral y 12 meses para la segunda paciente, el test de Minor fue positivo en 100 por ciento. Se aplicó toxina botulínica, y con tres meses de seguimiento no se evidenció recidiva objetivizado por el test de Minor. No se presentaron efectos adversos tras la administración de la toxina botulínica tipo A Botox®. La toxina botulínica representa un tratamiento efectivo, seguro y fácil de reproducir en el tratamiento del síndrome de Frey, sin evidencia de efectos adversos, y mejora en la calidad de vida de los pacientes.

Gustatory sweating recurring on peritoneal dialysis but resolving during periods of hemodialysis.

Gustatory sweating is a rare disorder characterized by profuse sweating on the forehead, face, scalp, and neck occurring soon after ingesting food, which has been reported in diabetic patients. The mechanism is thought to be triggered by taste buds and not gastric stimulation. We report a case where gustatory sweating repeatedly developed on peritoneal dialysis that resolved on periods of hemodialysis. A 32-year-old woman with diabetic end-stage renal disease developed gustatory sweating shortly after beginning continuous ambulatory peritoneal dialysis despite excellent clearances. After 5 months, she changed to hemodialysis for 2 months and noticed complete resolution of her gustatory sweating; however, after her return to peritoneal dialysis 2 months later, her gustatory sweating recurred. While on peritoneal dialysis, she was treated with clonidine, which resulted in improvement but not resolution of her symptoms as had occurred on hemodialysis. Another period on hemodialysis resulted in the resolution of her symptoms that returned again after restarting peritoneal dialysis. Clonidine provided incomplete relief while topical glycopyrrolate was effective and without complications. We report recurrent gustatory sweating on peritoneal dialysis that resolved with hemodialysis. We have no data to suggest that intra-abdominal stimulation played a role, but rather that despite excellent clearances neuropathy may have played a role. Treatment with topical glycopyrrolate may be safe and effective given every third day if clonidine is ineffective.

Síndrome del nervio auriculotemporal en niños secundario a un parto instrumentado con fórceps / Auriculotemporal nerve syndrome in children secondary to a forceps delivery

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Síndrome de Frey en la infancia / Frey´s sydrome in childhood

El síndrome de Frey o auriculotemporal se caracteriza por la aparición de episodios recurrentes de eritema facial y/o hiperhidrosis, localizados en el territorio del nervio auriculotemporal, que ocurre en respuesta a los estímulos gustatorios. Este cuadro es poco frecuente en la infancia. Presentamos el caso clínico de una niña de 4 años de edad que presentaba eritema sin hiperhidrosis en la mejilla izquierda tras la masticación

Frey's syndrome, also known as auriculotemporal syndrome is characterized by recurrent episodes of facial flushing and/or sweating along the distribution of the auriculotemporal nerve, that occurs in response to gustatory stimuli. The disorder is rare in children. We report a 4-year-old girl with flushing without hyperhidrosis in the left cheek on eating