Not only gustatory sweating and flushing: Signs and symptoms associated to the Frey syndrome and the role of botulinum toxin A therapy
Corresponding Author
Maria Raffaella Marchese
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Correspondence
Maria Raffaella Marchese, Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, L.go “A. Gemelli,” 8-00168 Rome, Italy.
Email: [email protected]
Search for more papers by this authorFrancesco Bussu
Division of Otolaryngology, Azienda Ospedaliero Universitaria di Sassari, Sassari, Italy
Division of Otolaryngology, Università di Sassari, Sassari, Italy
Search for more papers by this authorStefano Settimi
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Search for more papers by this authorEmanuele Scarano
Division of Otolaryngology, Azienda Ospedaliera Pia Fondazione di Culto e Religione Cardinale G. Panico, Tricase, Italy
Search for more papers by this authorGiovanni Almadori
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Division of Otorhinolaryngology, Università Cattolica del Sacro Cuore, Rome, Italy
Search for more papers by this authorJacopo Galli
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Division of Otorhinolaryngology, Università Cattolica del Sacro Cuore, Rome, Italy
Search for more papers by this authorCorresponding Author
Maria Raffaella Marchese
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Correspondence
Maria Raffaella Marchese, Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, L.go “A. Gemelli,” 8-00168 Rome, Italy.
Email: [email protected]
Search for more papers by this authorFrancesco Bussu
Division of Otolaryngology, Azienda Ospedaliero Universitaria di Sassari, Sassari, Italy
Division of Otolaryngology, Università di Sassari, Sassari, Italy
Search for more papers by this authorStefano Settimi
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Search for more papers by this authorEmanuele Scarano
Division of Otolaryngology, Azienda Ospedaliera Pia Fondazione di Culto e Religione Cardinale G. Panico, Tricase, Italy
Search for more papers by this authorGiovanni Almadori
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Division of Otorhinolaryngology, Università Cattolica del Sacro Cuore, Rome, Italy
Search for more papers by this authorJacopo Galli
Division of Otorhinolaryngology, Department of Aging, Neuroscience, Orthopedics and Head and Neck Sciences, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy
Division of Otorhinolaryngology, Università Cattolica del Sacro Cuore, Rome, Italy
Search for more papers by this authorAbstract
Background
The classic symptoms of Frey syndrome are gustatory sweating and flushing. Aims of the study were to describe prevalenceand severity of typical and atypical presentations of the disorder and to assess the effects of botulinum neurotoxin A (BoNT-A) therapy in patients with Frey syndrome after parotidectomy.
Methods
In this prospective, observational study on 18 patients, we assessed symptom severity before therapy, after 15 days, 1, 3 and 6 months' follow-up with the sweating-flushing-itch-paresthesia-pain (SFIPP) Frey scale specifically designed by the authors themselves for this study.
Results
Before BoNT-A injection, all patients (100%) complained gustatory sweating, 80% paresthesia, 77% gustatory flushing, 60% pain and 60% gustatory itch. The SFIPP-Frey overall score and the symptom-specific ones decreased significantly at each post-therapy control.
Conclusions
The prevalence of “unusual” manifestations is not negligible. BoNT-A improves symptoms severity. The SFIPP-Frey scale may be useful to assess symptoms and to monitor post-therapy outcomes.
Open Research
DATA AVAILABILITY STATEMENT
The data that support the findings of this study are available from the corresponding author upon reasonable request.
REFERENCES
- 1Bjerkhoel A, Trobbe O. Frey's syndrome: treatment with botulinum toxin. J Laryngol Otol. 1997; 111: 839-844.
- 2Baillarger JGF. Memoire sur l'obliteration du canal de Stenon. Gaz Med (Paris). 1853; 23: 194-197.
- 3Frey L. Le syndrome du neuf auriculo-temporal. Rev Neurol (Paris). 1923; 30: 97-104.
- 4Drummond PD. Mechanisms of gustatory flushing in Frey's syndrome. Clin Auton Res. 2002; 12: 144-146.
- 5Rustemeyer J, Eufinger H, Bremerich A. The incidence of Frey's syndrome. J Craniomaxillofac Surg. 2008; 36: 34-37.
- 6Neumann A, Rosenberger D, Vorsprach O, Dazert S. The incidence of Frey syndrome following parotidectomy: results of a survey and follow-up. HNO. 2011; 2: 173-178.
10.1007/s00106-010-2223-6 Google Scholar
- 7Gardner WJ, McCubbin JW. Auriculotemporal syndrome: gustatory sweating due to misdirection of regenerated nerve fibers. J Am Med Assoc. 1956; 160: 272-277.
- 8Rouyer J. Nte sur l'hyperhidrose parotidienne. J de la Physiol de l'homme et des Animaux. 1959; 2: 447-449.
- 9Botkin S. Uberdie Refexerscheinungen im Gebiete der Hautgefasse und uber den reflectorischen Schweiss. Berliner Klinischen Wochenschrift. 1875; 7: 81-83.
- 10Weber FP. Clinical case, V: a case of localized sweating and blushing on eating, possibly due to temporary compression of vasomotor fibers. Trans Clin Soc London. 1897; 31: 277-280.
- 11New GB, Bozer HE. Hyperhydrosis of the cheek associated with the parotid region. Minn Med. 1922; 5: 652-657.
- 12Linder TE, Huber A, Schmid S. Frey's syndrome after parotidectomy: a retrospective and prospective analysis. Laryngoscope. 1997; 107: 1496-1501.
- 13Bussu F, Parrilla C, Rizzo D, Almadori G, Paludetti G, Galli J. Clinical approach and treatment of benign and malignant parotid masses, personal experience. Acta Otorhinolaryngol Ital. 2011; 31: 135-143.
- 14May JS, McGuirt WF. Frey's syndrome: treatment with topical glycopyrrolate. Head Neck. 1989; 11: 85-89.
- 15Laskawi RT, Schott M, Mirzaie-Petri M, Schroeder M. Surgical management of pleomorphic adenomas of the parotid gland: a follow-up study of three methods. J Oral Maxillofac Surg. 1996; 54: 1176-1179.
- 16Laage-Hellman JE. Treatment of gustatory sweating and flushing. Acta Otolaryngol. 1958; 49: 132-143.
- 17Hays LL, Novack AJ, Worsham JC. The Frey syndrome: a simple, effective treatment. Otolaryngol Head Neck Surg. 1982; 90: 419-425.
- 18Coldwater KB. Surgical treatment of the auriculotemporal syndrome. AMA Arch Surg. 1954; 69: 54-57.
- 19Redleaf MI, McCabe BF. Gustatory otorrhea: Frey's syndrome of the external auditory canal. Ann Otol Rhinol Laryngol. 1993; 102: 438-440.
- 20Claes G, Drott C. Hyperhidrosis. Lancet. 1994; 343: 247-248.
- 21Bushara KO, Park DM. Botulinum toxin and sweating. J Neurol Neurosurg Psychiatry. 1994; 57: 1437-1438.
- 22Lovato A, Restivo DA, Ottaviano G, Marioni G, Marchese-Ragona R. Botulinum toxin therapy: functional silencing of salivary disorders. Acta Otorhinolaryngol Ital. 2017 Apr; 37(2): 168-171.
- 23Fox RH, Goldsmith R, Kidd DJ. Cutaneous vasomotor control in the human head, neck, and upper chest. J Physiol. 1962; 161: 298-312.
- 24Dale HH, Feldberg W. The chemical transmission of secretory impulses to the sweat glands of the cat. J Physiol. 1934; 82: 121-128.
- 25Dulguerov P, Quinodoz D, Cosendai G, Piletta P, Lehmann W. Frey syndrome treatment with botulinum toxin. Otolaryngol Head Neck Surg. 2000; 122(6): 821-827.
- 26Drobik C, Laskawi R. Frey-Syndrom: Behandlung mit Botulinum-Toxin. HNO Aktuell. 1994; 2: 142-144.
- 27Drobik C, Laskawi R, Schwab S. Treatment of Frey's syndrome with botulinum toxin A: experiences with a new method. HNO. 1995; 43: 644-648.
- 28Schulze-Bonhage A, Schroder M, Ferbert A. Botulinum toxin in the therapy of gustatory sweating. J Neurol. 1996; 243: 143-146.
- 29Naumann M, Zellner M, Toyka KV, Reiners K. Treatment of gustatory sweating with botulinum toxin. Ann Neurol. 1997; 42: 973-975.
- 30Laskawi R, Drobik C, Schonebeck C. Up-to-date report of botulinum toxin type A treatment in patients with gustatory sweating (Frey's syndrome). Laryngoscope. 1998; 108: 381-384.
- 31Laccourreye O, Akl E, Gutierrez-Fonseca R, Garcia D, Brasnu D, Bonan B. Recurrent gustatory sweating (Frey syndrome) after intracutaneous injection of botulinum toxin type A. Arch Otolaryngol Head Neck Surg. 1999; 125: 283-286.
- 32Laccourreye O, Muscatelo L, Naude C, Bonan B, Brasnu D. Botulinum toxin type A for Frey's syndrome: a preliminary prospective study. Ann Otol Rhinol Laryngol. 1998; 107: 52-55.
- 33Bussu F, Rigante M, Giglia V, et al. Clinical history, prognostic factors, and management of facial nerve in malignant tumors of the parotid gland. Clin Exp Otorhinolaryngol. 2014; 7: 126-132.
- 34Bussu F, Tagliaferri L, Mattiucci GP, et al. HDR interventional radiotherapy (brachytherapy) in the treatment of primary and recurrent head and neck malignancies. Head Neck. 2019; 41: 1667-1675.
- 35Bednarek J, Reid W, Matsumoto T. Frey's syndrome. Am J Surg. 1976; 131: 592-594.
- 36De Benedettis G. Auriculotemporal syndrome (Frey's syndrome) presenting as tic douloureux. J Nuerosurg. 1990; 72: 955-958.
- 37Ricks R, Banga R, Porter M. Gustatory itching: an unusual complication following superficial parotidectmy. J Laryngol Otol. 2010; 124(11): 1227-1228.
- 38Haddock A, Porter SR, Scully C, Smith I. Submandibular gustatory sweating. Oral Surg Oral Med Oral Pathol. 1994; 77: 317.
- 39Jansen S, Jerowski M, Ludwig L, Fischer-Krall E, Beutner D, Grosheva M. Botulinum toxin therapy in Frey's syndrome: a retrospective study of 440 treatments in 100 patients. Clin Otolaryngol. 2017; 42: 295-300.
- 40Beerens AJ, Snow GB. Botulinum toxin A in the treatment of patients with Frey syndrome. Br J Surg. 2002; 89(1): 116-119.
- 41de Bree R, Duyndam JE, Kuik DJ, Leemans CR. Repeated botulinum toxin type A injections to treat patients with Frey syndrome. Arch Otolaryngol Head Neck Surg. 2009; 135(3): 287-190.
- 42Van Weel C. Functional status in primary care: COOP/WONCA charts. Disabl Rehabil. 1993; 15: 96-101.
- 43Blitzer A, Brin MF, Fahn S, Lange D, Lovelace RE. Botulinum toxin (BOTOX) for the treatment of “spastic dysphonia” as part of a trial of toxin injections for the treatment of other cranial dystonias. Laryngoscope. 1986; 96: 1300-1301.
- 44Blitzer A, Benson BE, Guess J. Botulinum Neurotoxin for Head and Neck Disorders. New York, NY: Thieme; 2012.
10.1055/b-002-85483 Google Scholar
- 45Blitzer A, Sulica L. Botulinum toxin: basic science and clinical uses in otolaryngology. Laryngoscope. 2001; 111: 218-226.
- 46Marchese MR, D'Alatri L, Bentivoglio AR, Paludetti G. Onabotulinumtoxin A for adductor spasmodic dysphonia (ADSD): functional results and the role of dosage. Toxicon. 2018; 155: 38-42.
Citing Literature
