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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
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