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Journal of Government Dental College and Hospital, October 2014, Vol.-01, Issue- 01, P. 11-15 Case Report: A case report of Frey’s syndrome following TMJ surgery 1Dr.Hitarthi J.Kubavat , 2Dr.Kawar , 3Dr.Jaysankar Pillai , 4 Dr. Jigna Shah 1Assistant Professor, 2PG Student, Dept. of Oral Medicine and Dental Radiology, Govt. Dental College and Hospital, Ahmedabad-380016 3Tutor. 4 Dept. of oral Medicine and Dental Radiology, Govt. Dental College and Hospital, Ahmedabad-380016 Dept. of Oral Pathology, Govt. Dental College and Hospital, Ahmedabad-380016 Professor & Head,Dept of Oral Medicine & Radiology, Govt. Dental College & Hospital, Ahmedabad-380016 Corresponding author: Dr. Hitarthi J Kubavat ABSTRACT Frey’s syndrome is a well recognized complication of surgery in preauricular region. It is characterized mainly by recurrent episodes of hyperesthesia, flushing and warmth or sweating limited to cutaneous distribution of auriculotemporal nerve while eating foods that produce strong salivary stimulus. Although commonly encountered as a complication of total or partial parotidectomy, on rare occasions it follows surgery or fracture of temporomandibular joint. A case of Frey’s syndrome in a patient who developed symptoms 3 yrs after TMJ surgery is reported. Keywords : Frey’s syndrome Although relatively common in adults the INTRODUCTION Frey’s syndrome is a disorder characterized by condition in rare instances has been reported in unilateral sweating and flushing of facial skin in children and infants as a sequel of perinatal birth area of parotid gland occurring during meals. 1,2 The trauma from assisted forceps delivery. 9, 10 Through syndrome was initially termed ‘ auriculotemporal the years several theories have been proposed nerve syndrome’ and regarding the pathophysiology of the syndrome. 1,2,3 ‘gustatory sweating’. also been referred to as Duphenix first reported it in Lucia Frey believed that the damaged 1757 and it was Lucia Frey, a French neurologist auriculotemporal nerve is invaded and irritated by who in 1923 implicated auriculotemporal nerve in healing tissue.2,5 Freedberg suggested damage to pathogenesis of syndrome and used the term nerve may cause distribution of sympathetic fibers ‘auriculotemporal nerve syndrome’. 2, 3, 4 leading to parasympathetic hypersensitivity and Frey’s syndrome has been reported to occur stimulation. The theory of aberrant regeneration more frequently following parotid gland surgery. says that there is defective nervous regeneration Some authors believe that incidence vary from 10- following injury to auriculotemporal nerve. The others report it to misdirection of regenerating parasympathetic fibers . Over 90% of the patients test to denervated sweat glands results in simultaneous 30% of symptomatic patients vary from 30-50% 5 6, 7 positive for gustatory sweating many of whom are activation of parotid and sweat glands.4, 5, 6 asymptomatic.8 Less frequently it follows fracture of Various tests to asset the presence of gustatory mandible, surgery or fracture of TMJ, radical neck sweating have been described. The most widely dissection, submandibular gland excision, and used is minor’s starch iodine test. thyroidectomy or after thoracic sympathectomy. 5,8 1,2,3,11 include biosensoring method using Other test enzymatic 11 Journal of Government Dental College and Hospital, October 2014, Vol.-01, Issue- 01, P. 11-15 electrodes to detect L-lactate levels on skin of Ahmedabad affected area, use of thin facial tissue paper to preauricular incision. There were no substantial demonstrate areas of sweating, one step method complaints till 3 yrs after surgery but before 15 days using powder, patient complained that when beginning to eat he pyrogallol, ferric hydroxide or quinazarin and felt a sharp pain, intense warmth and sweating in dyes like bromophenol infrared medical thermography. blue 4,12 using gap arthroplasty with preauricular area on left side. The rest of his Differential diagnosis include crocodile tear syndrome, gustatory sweating associated with 8 9, 10, 13 diabetes and food allergy in case of children. medical history was non contributory. Examination revealed scar in left preauricular region (fig-1), restricted mouth opening of 30mm, Frey’s syndrome in children usually resolves and deviation of face to left side (fig-2). Intraoral spontaneously and no treatment is required. Several examination showed posterior cross bite on left side treatment options have been described in adults who and deviation of anterior midline to left side. An aim at reducing incidence of Frey’s syndrome but orthopantomogram showed condylectomy of left none of them have given promising results. There side (fig 3). The diagnosis of Frey’s syndrome was are mainly three options- surgical measures, made and confirmed by performing minor’s starch medicinal treatment and radiation therapy. Surgical iodine test. The affected areas were coated with 1% measures include interposition of sternocleido iodine solution and were allowed to dry. Starch 5,14 superficial musculoaponeurotic powder was applied on the skin (fig-4) and patient system, alloderm, fascia lata or sialasthic sheeting, was given a tablet of ascorbic acid as a salivary transmeatal intracranial stimulus. After a few minutes patient complained of neurolysis of glossopharyngeal nerve or transaction sharp pain and heat sensation and areas of blue mastoid flap, 15 tympanic neurectomy, 14,15,16 Medicinal black colorations could be observed (fig-5). These treatment includes systemic or local anticholinergics areas represented the combination of secretion of of Jacobson’s anastomosis. 16 aluminium sweat glands diluted with iodine which reacted with chloride hexahydrate, diphemanil methyl sulphate starch producing this coloration. Although the areas and intracutaneous injections of botulinum toxin were small, the diagnosis of Frey’s syndrome was such as scopolamine, glycopyrollate, type A. 6,17 confirmed .Available treatment options were CASE REPORT explained to the patient. Patient opted for topical A 26 years old male patient reported to oral application and was prescribed topical use of medicine G.D.C.H. Glycopyrrolate twice daily. After fifteen days of Ahmedabad with chief complaint of pain and follow up the patient stated improvement in pain sweating in left preauricular region while eating and gustatory sweating in the preauricular area since fifteen days. which and radiology dept. of he was experiencing pretherapeuticly. There was history of trauma when patient was However he experienced slight dryness of mouth for four years of age. His mouth opening gradually less than an hour after the topical application of decreased after that so he was operated for left TMJ glycopyrrolate cream. ankylosis at oral surgery dept. of G.D.C.H. 12 Journal of Government Dental College and Hospital, October 2014, Vol.-01, Issue- 01, P. 11-15 treatment.2,4,6 The mainstay of the treatment lies in DISCUSSION reassurance and an explanation of the condition. The specific mechanism involved in Frey’s syndrome Various forms of treatment have been advocated is yet unknown. However the theory of aberrant with varying reported degrees of success.4,5 Surgical regeneration theory. treatment procedures have possible complications Auriculotemporal nerve is the terminal of mandibular and no guarantees of permanent success.5,11 branch of trigeminal nerve. It is mixed nerve and has Medicinal treatment provides temporary relief and sympathetic The can cause local irritation to skin and sweat glands. secretomotor activity of parotid gland is controlled Injections of botulinum toxin are costly, have via parasympathetic fibers of this nerve. Injury to the adverse side effects and there are reports of branches of auriculotemporal nerve during preau- recurrence after intracutaneous injections.6,8,17 The ricular surgery damages it. In process of nerve use regeneration parasympathetic secretomotor fibers may glycopyrrolate become misdirected and grow along distal cut ends of documented. Glycopyrrolate, an anticholinergic sympathetic fibers to the skin vessels and sweat drug, is a quaternary ammonium compound that glands. As both parasympathetic and sympathetic does not cross the blood brain barrier. It also fibers are cholinergic, a new stimulus is made penetrates biological membrane more slowly than possible and a gustatory stimulus produces sweating other anticholinergics and appears to lead to fewer is and the most acceptable parasympathetic fibers. of 0.5% aqueous for solution of hyperhidrosis topical is well side effects.4,16 1, 2,3,4,5 and flushing. The symptoms of Frey’s syndrome usually CONCLUSION present 6 weeks to several months after surgery in Frey’s syndrome is an interesting illustration of how parotid gland but can present as late as 5 years after misdirected 2,3,4 nerve regeneration can manifest In present case patient presented with clinically. Frey’s syndrome has the potential to symptoms 3 years postoperatively. Although rarely cause great social distress for the patient and surgery. 2,7,12 but Frey’s syndrome do follow surgery of TMJ potential exist for negative psychological as is seen in present case. consequences. For this reason it is important not include only for dentist but also for general practitioner to flushing and warmness with overheating of affected be aware of this disorder and to be able to counsel areas of the skin which in some cases is associated patient on available treatment options. with pain2,6 as is seen in this case.Minor’s starch Fig.1 Photograph of patient’s face showing scar in iodine test performed in the present case is most left preauricular region. widely used test. The test is accurate, easy to Fig.2 Deviation of patient’s face towards left side perform, provide usual confirmation of gustatory Fig.3 OPG showing condylectomy of left side. sweating and can identify asymptomatic patients of Fig.4 Iodine and starch powder applied on skin Symptoms Frey’s syndrome. of Frey’s syndrome 4, 8,11 Fig.5 Black discoloration in area of scarring. Only 10-15% of patients with Frey’s syndrome have symptoms severe enough to seek 13 Journal of Government Dental College and Hospital, October 2014, Vol.-01, Issue- 01, P. 11-15 Figure 1 Figure 2 Figure 5 Figure 3 REFERENCES 1. Shelton Malatskey, Fradis et al Iaron Rabinovich, Milo Frey syndrome—delayed clinical onset: A case report Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002; 94:338-40 Figure 4 2. Cassio Edvard Sverzut, Alexandre Elias et al. Frey syndrome after condylar fracture: case report Braz.Dent journal 2004, 15; 2 3. Rishi kumar Bali, Naveen Chhabra,Zarina Frey’s syndrome as a sequele of superficial parotidectomy JIAMOR 2006, 18; 03, 181-183. 4. S.Sood , M.S. Qurashi, P.J. Bradley. Frey’s syndrome and parotid surgery Clin. Otolaryngol 1998,23; 291-301 5. S Sood, M S Quraishi,C R Jennings et al. Frey’s syndrome following parotidectomy: prevention using a rotation sternocleidomastoid muscle flap Clin Otolarynol 1999, 24, 365-368 6. 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