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J Oral Maxillofac Surgxx:xxx, 2011

Frey Syndrome—An UnderreportedComplication to Closed Treatment ofMandibular Condyle Fracture? Case

Report and Literature Review

Tue W. Kragstrup, MD,* Jennifer Christensen, DDS,†

Karin Fejerskov, DDS,‡ and Ann Wenzel, PhD, DrOdont,§

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Frey syndrome (auriculotemporal syndrome, gusta-tory sweating) is characterized by episodes ofwarmth, flushing, and sweating of the face in thepreauricular region initiated by gustatory stimulus.1

Frey syndrome is a common complication afteroperations on the parotid gland and the temporo-mandibular joint.2,3 The most common hypothesiss that regenerating parasympathetic fibers to sali-ary glands connect in error with the sweat glandsnd subcutaneous blood vessels of the skin. Thenset has usually been 12 to 18 months after sur-ery. The most effective treatment has been subcu-aneous infiltration of botulinum toxin into the af-ected area.4

Fracture of the mandible is a common injury. Themandibular condyle will sustain a fracture in 10% to 40%of all mandibular fractures, and 19% of condylar frac-tures will be dislocated.5,6 The treatment of condylarractures is still under evaluation, and closed treatmentith maxillomandibular fixation has frequently been

elected.7

Few reports exist on the development of Frey syn-drome after acute mechanical trauma to the jaws, and

*Medical Intern, Department of Orthopedic Surgery, Bispebjerg

Hospital, University of Copenhagen, Copenhagen, Denmark.

†PhD Student, Departments of Oral Radiology and Oral and

Maxillofacial Surgery and Oral Pathology, Aarhus University

School of Dentistry, Aarhus, Denmark.

‡Clinical Teacher, Department of Clinical Oral Physiology, Aar-

hus University School of Dentistry, Aarhus, Denmark.

§Professor, Department of Oral Radiology, Aarhus University

School of Dentistry, Aarhus, Denmark.

Address correspondence and reprint requests to Dr Krag-

strup: Department of Orthopedic Surgery, Bispebjerg Hospital,

Bispebjerg Bakke 23, Copenhagen 2400, Denmark; e-mail:

t.kragstrup@dadlnet.dk

© 2011 American Association of Oral and Maxillofacial Surgeons

278-2391/11/xx0x-0$36.00/0

oi:10.1016/j.joms.2010.12.033

1

the frequency after closed treatment of mandibularcondyle fracture is unknown.

Case Report

A 23-year-old woman was referred to the Aarhus Univer-sity School of Dentistry for an insurance examination after abicycle accident 18 months earlier. She had had fractures ofseveral teeth, the mandibular body between the lower rightlateral and central incisor, the right coronoid process, andboth condyles. The right condylar head had been displacedmedially (Fig 1A,B). The patient had undergone open re-

uction and internal fixation of the symphysis and closedreatment with intermaxillary fixation of the coronoid andondylar fractures. She complained of hypoesthesia in thereauricular region and the cheek on the right side and

atigue and pain of both temporomandibular joints. Clinicalxamination by a temporomandibular joint specialist re-ealed tender masseter and temporal muscles in both sidesnd a reduced mouth opening. These problems were allell-known complications to mandibular fractures. The pa-

ient had also during the previous couple of months expe-ienced episodes of sweating, blushing, and warmth in thereauricular region and cheek on the right side while eat-

ng. At the first clinical examination, this problem was notlicited, because the patient did not connect it to therauma. She coincidentally confronted one of us (T.W.K.; aedical student at the time) with the problem, and he

uspected Frey syndrome. At the second clinical examina-ion some months later, clinical images were taken beforend after she had eaten a sandwich. These showed flushingnd sweat production at the right side (data not shown).ext, the minor starch-iodine test8 was performed, confirm-

ng sweating on the right side (Fig 2). Thermography waslso performed, with the thermograms visualizing heat pro-uction. After the patient chewed the sandwich, the tem-erature increased on average 2.2°C (from 32.5°C to4.7°C) on the right side and only 1.1°C (from 32.0°C to3.1°C) on the left side (Fig 3). The patient was diagnosedith Frey syndrome as a complication of the medially dis-

ocated mandibular condyle fracture managed by closedreatment. The patient was pleased by the explanation ofer symptoms and did not want any treatment.

Literature Review

One prospective study and 10 case reports de-

scribing the development of Frey syndrome after

2 FREY SYNDROME

FIGURE 1. Three-dimensional CT scan reconstruction of A, right side and B, left side showing fractures of several teeth, mandibular bodybetween the lower right lateral and central incisor, right coronoid process, left condyle without dislocation, and right condyle with medialdislocation.

Kragstrup et al. Frey Syndrome. J Oral Maxillofac Surg 2011.

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KRAGSTRUP ET AL 3

closed treatment of mandibular condyle fractureswere found in the period from 1969 to 2010 usingthe search criteria in PubMed: “Frey syndrome”combined with “mandibular fracture” or “jawtrauma.” These reports were thoroughly stu-died.

The single prospective study was published in 1977by Schmidseder and Scheunemann,9 who reported 2ases of Frey syndrome (0.8%) in 237 fractures of theandibular condyle managed by closed treatment

rom 1971 to 1975. All 10 case reports describedatients with Frey syndrome that developed afterlosed treatment of mandibular condyle fractures.10-18

Table 1 lists the patient characteristics from each casereport. In 9 of the 10 cases, the fractures had resultedfrom traffic accidents or falls from above ground level.The mandibular condyle was dislocated in 6 cases,and 9 patients had more than 1 mandibular fracturesite. In 5 of the 10 cases, the patient had had alteredsensibility in the affected area. The latency periodbefore the onset of symptoms ranged from 2 monthsto 2 years, and only 1 of the 10 patients had had

FIGURE 2. Clinical image showing minor starch-iodine test. Blackdots indicate sweating.

Kragstrup et al. Frey Syndrome. J Oral Maxillofac Surg 2011.

spontaneous remission.

Discussion

The incidence of Frey syndrome after closedtreatment of a condylar fracture in the 1 prospec-tive study was almost 1%.9 Considering the rela-tively high frequency of mandibular condyle frac-tures, it is noteworthy that only 10 cases have beenreported in the past 4 decades. This could suggestthat the complication might be underreported. Justas occurred in the present patient, the explanationscould be that the patient has not connected thesymptoms to the trauma or that the symptoms ordiagnosis were unknown to the clinicians examin-ing the patient. Frey syndrome might be known tootolaryngologists; however, many patients will pri-marily be examined by physicians or oral surgeonsand radiologists who might not be acquainted withthe characteristic symptoms. Also, the long latencyand the peculiar association between eating and thedevelopment of symptoms might make it difficultfor patients to explain the symptoms. It is thereforeessential that oral surgeons, temporomandibularjoint specialists, and dental-maxillofacial radiolo-gists consider Frey syndrome when examining pa-tients with previous mandibular condyle fracturemanaged by closed treatment.

The anatomy and pathology of Frey syndromeafter closed treatment of condylar fracture has beenpreviously reviewed.1,17,19 Frey syndrome developsbecause of nerve injury. The nerve in question isthought to be the auriculotemporal nerve, whichconsists of parasympathetic fibers to the parotidgland, sympathetic fibers to the sweat glands andsubcutaneous vessels of the skin, and sensory fibersto the skin in the preauricular area. The auriculo-temporal nerve is a branch of the mandibular divi-sion of the trigeminal nerve and passes just poste-rior to the mandibular condyle. Injury to theauriculotemporal nerve by a fractured and dislo-cated condyle is thought to be the mechanism lead-ing to the autonomic neuropathies of Frey syn-drome after closed treatment of condylar fractures.The gustatory sweating of Frey syndrome can beexplained by 2 hypotheses. The most common hy-pothesis is that the regenerating parasympatheticfibers to salivary glands connect in error with thesweat glands. The other hypothesis is that thesweat glands develop an increased sensitivity afterdegeneration of sympathetic fibers and are thenstimulated by acetylcholine released from adjacentparasympathetic fibers in the parotid gland. Thegustatory vasodilation of Frey syndrome causingblushing and warmth is more difficult to elucidate.However, it has mostly been hypothesized to becaused by the misdirected regeneration of parasym-

pathetic fibers to the subcutaneous blood vessels.

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4 FREY SYNDROME

Under these circumstances, gustatory stimuli willlead, not only to stimulation of the parotid gland,but also to stimulation of the sweat glands andsubcutaneous blood vessels. The symptoms ofthese autonomic neuropathies are episodes ofwarmth, flushing, and sweating of the face in thepreauricular region initiated by gustatory stimulus.The changed sensibility associated with Frey syn-drome can be explained by the injury to the auri-culotemporal nerve or smaller nerves in the area.

In the present case, and in most of the previous casereports, Frey syndrome after closed treatment of a man-dibular fracture developed after a traffic accident. An-other characteristic was the presence of more than 1fracture of the mandible. This could suggest thatinjury of the auriculotemporal nerve requirestrauma with a considerable impact. In more thanone half of the cases, the mandibular condyle wasdislocated. Dislocation of the condyle could also bea risk factor for developing Frey syndrome, consid-

FIGURE 3. Thermograms showing mean temperature at affectedunaffected region C, before and D, after patient chewed a sandw

Kragstrup et al. Frey Syndrome. J Oral Maxillofac Surg 2011.

ering the anatomy of the auriculotemporal nerve. t

Altered sensibility was also present in one half ofthe cases and in our patient and might also beassociated with the development of Frey syndrome,because this would indicate injury to the auriculo-temporal nerve. The difference in the latency pe-riod could be explained by the different hypothesesof the pathology of Frey syndrome.17 The theory ofregenerating nerves explains well the cases of thesyndrome developing after 6 months or longer. Thehypothesis of degenerating nerves could explainthe cases with a faster onset of symptoms. The onlypatient with spontaneous remission had had a fastonset, which might suggest that late onset predictsa worse prognosis.

The diagnosis of Frey syndrome is based on the char-acteristic symptoms. In addition, the minor starch-iodinetest is a sensitive diagnostic tool.8 In the present case,hermography displayed the heat production character-stic of Frey syndrome. Thermography has previouslyeen suggested as a valid method to document the

n A, before and B, after patient chewed a sandwich and at thehite indicates areas with highest temperature.

regioich. W

emperature differences between the 2 sides of the face

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KRAGSTRUP ET AL 5

(when only 1 side is affected).20 This can be an aid innsurance cases, when the degree of permanent injury

ust be reported.The treatment of Frey syndrome has been previously

eviewed.4 The condition can cause considerable socialembarrassment and social incapacity because of the pro-fuse flushing and sweating when eating, and the diag-nosis should have an effect on the insurance benefits forthe patient. Importantly, however, only a few patientswith Frey syndrome will need treatment other than anexplanation of the condition and reassurance. If treat-ment is required, the most effective treatment has beensubcutaneous infiltration of botulinum toxin to the af-fected area.

In conclusion, the present case report has de-scribed the development of Frey syndrome after adislocated mandibular condyle fracture managed byclosed treatment. The findings from the literaturereview suggest that the complication might be under-reported. The risk factors seem to be trauma with aconsiderable impact, more than 1 mandibular fracturesite, dislocation of the mandibular condyle, and al-tered sensibility in the preauricular region. The minorstarch-iodine test and thermographic imaging can

Table 1. PATIENT CHARACTERISTICS FROM PREVIOUS C

Investigator Trauma Other FracturesCond

Disloc

aws et al12 Fall from ascaffold

Ascending ramus Medialdisloc

Laws et al12 Bicycleaccident

None None

Martis et al13 Caraccident

Body, zygomaticarch

None

Storrs et al16 Trafficaccident

Symphysis,angle, othercondyle

Posteriodisloc

Olson et al15 Motorcycleaccident

Symphysis,coronoid,other condyle

None

Goodman et al11 Motorcycleaccident

Parasymphyseal,other condyle

Not stat

Zoller et al17 Caraccident

Symphysis,midface, skullbase

Medial

Dhaif et al1 Trafficaccident

Parasymphyseal Dislocat

Mellor et al14 Fall afterfainting

Parasymphyseal,other condyle

Open thauditomeatu

Gerbino et al10 Jump from3rd floor

Symphysis, othercondyle, LeFort I

Medialdisloc

Kragstrup et al. Frey Syndrome. J Oral Maxillofac Surg 2011.

supplement the clinical examination.

Acknowledgment

Professor Robert Gniadecki and Heidi Larsen from the Depart-ment of Dermatology, Bispebjerg Hospital, Denmark are thankedfor helping with the minor starch-iodine test.

References1. Frey: Le syndrome du nerf auriculo-temporal. Rev Neurol 2:97,

19232. Langdon JD: Complications of parotid gland surgery. J Maxillo-

fac Surg 12:225, 19843. Swanson KS, Laskin DM, Campbell RL: Auriculotemporal syn-

drome following the preauricular approach to temporomandib-ular joint surgery. J Oral Maxillofac Surg 49:680, 1991

4. de Bree R, van der Waal I, Leemans CR: Management of Freysyndrome. Head Neck 29:773, 2007

5. Sawazaki R, Lima-Junior SM, Asprino L, et al: Incidence andpatterns of mandibular condyle fractures. J Oral Maxillofac Surg68:1252, 2010

6. Zachariades N, Mezitis M, Mourouzis C, et al: Fractures of themandibular condyle: A review of 466 cases. Literature review,reflections on treatment and proposals. J Craniomaxillofac Surg34:421, 2006

7. Park JM, Jang YW, Kim SG, et al: Comparative study of theprognosis of an extracorporeal reduction and a closed treat-ment in mandibular condyle head and/or neck fractures. J OralMaxillofac Surg 68:2986, 2010

8. Hexsel D, Rodrigues TC, Soirefmann M, et al: Recommendations forperforming and evaluating the results of the minor test according to

EPORTS

ChangedSensibility Latency Course

Hypersensitivity inthe preauricularregion

6 mo Remission after resectionof thetemporomandibularnerve

None �6 yr Chronic

Hypersensitivity inpreauricularregion

1 mo Spontaneous remission

Paresthesia infront of ear

2 yr Chronic

None 3 mo Chronic

None Years Not stated

Hypoesthesia inpreauricularregion

2-3 mo Not stated

None 3 mo Not stated

Paresthesia inpreauricularregion

1 yr Chronic

None 6-7 mo Chronic

ASE R

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a sweating intensity visual scale. Dermatol Surg 36:120, 2010

6 FREY SYNDROME

9. Schmidseder R, Scheunemann H: Nerve injury in fractures ofthe condylar neck. J Maxillofac Surg 5:186, 1977

10. Dhaif G, Kuriakose S, Sleeman D, et al: Frey’s syndrome afterfracture of the mandibular condyle. Oral Surg Oral Med OralPathol Oral Radiol Endod 80:2, 1995

11. Gerbino G, Roccia F, Grosso M, et al: Pseudoaneurysm of theinternal maxillary artery and Frey’s syndrome after blunt facialtrauma. J Oral Maxillofac Surg 55:1485, 1997

12. Goodman RS: Frey’s syndrome: Secondary to condylar fracture.Laryngoscope 96:1397, 1986

13. Laws IM: Two unusual complications of fractured condyles.Br J Oral Surg 5:51, 1967

14. Martis C, Athanassiades S: Auriculotemporal syndrome (Frey’ssyndrome), secondary to fracture of the mandibular condyle.

Plast Reconstr Surg 44:603, 1969

15. Mellor TK, Shaw RJ: Frey’s syndrome following fracture of the man-dibular condyle: Case report and literature review. Injury 27:359,1996

16. Olson RE, Walters CL, Powell WJ: Gustatory sweating causedby blunt trauma. J Oral Surg 35:306, 1977

17. Storrs TJ: A variation of the auriculotemporal syndrome. Br JOral Surg 11:236, 1974

18. Zoller J, Herrmann A, Maier H: Frey’s syndrome secondary to asubcondylar fracture. Otolaryngol Head Neck Surg 108:751,1993

19. Harper KE, Spielvogel RL: Frey’s syndrome. Int J Dermatol25:524, 1986

20. Isogai N, Kamiishi H: Application of medical thermographyto the diagnosis of Frey’s syndrome. Head Neck 19:143,

1997