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JCN Open Access LETTER TO THE EDITOR

pISSN 1738-6586 / eISSN 2005-5013 / J Clin Neurol 2019;15(4):585-587 / https://doi.org/10.3988/jcn.2019.15.4.585

A Case of Frey’s Syndrome Diagnosed and Followed Up


Using the Quantitative Sudomotor Axon Reflex Test

Jaehwan Kim Dear Editor,


Minkyung Kim Frey’s syndrome (also called focal gustatory hyperhidrosis) is a rare neurologic disorder
YeEun Kim presenting with abnormal facial sweating when consuming meals. The syndrome had been
Hye Lim Lee reported in up to 53% of parotidectomy patients, and may occur due to injury or inflamma-
Department of Neurology, tion of the parotid or submandibular glands, or the cervical sympathetic trunk.1,2 The diag-
Korea University Guro Hospital, nosis is traditionally made based on clinical symptoms and a starch iodine test.3 This test, while
Korea University College of Medicine,
Seoul, Korea
sensitive to the diagnosis of focal hyperhidrosis, does not adequately quantify sweat secretion.
We report a case in which we performed the quantitative sudomotor axon reflex test (QSART)
to diagnose focal hyperhidrosis, and assessed the therapeutic effect at follow-up.
A 45-year-old man visited a department of neurology for sweating in the left auriculotem-
poral area while consuming meals or chewing. He reported that the symptom began at ap-
proximately 30 years of age and was more severe when eating hot or sour foods. A benign
branchial cleft cyst had been surgically removed from the left side of his neck in infancy. Ad-
ditionally, he underwent endoscopic submucosal dissection due to early gastric cancer 1 month
prior to admission, but was not taking any medication. During a neurologic examination he
reported experiencing hypesthesia at the operation site since childhood. Laboratory tests in-
cluding thyroid function and blood sugar, autoimmune antibody tests, electrophysiologic tests,
and brain CT revealed no abnormalities.
We performed the QSART using the Q-Sweat device (WR Medical, Maplewood, MN,
USA) in both auriculotemporal areas, as well as on the left arm and leg.4 We observed ex-
cessive sweating in the left but not the right auriculotemporal area when the patient was
chewing gum (Fig. 1A). Hyperhidrosis was also observed in the left auriculotemporal area
upon stimulation with acetylcholine (Fig. 1B).
The nightly application of aluminum chloride cream greatly reduced his symptoms. The to-
tal sweat volume in the left auriculotemporal area while chewing was significantly reduced
at a 2-month follow-up (Fig. 1C).
Our use of the QSART confirmed the presence of excessive sweat secretion in the symp-
tomatic auriculotemporal site induced both by either stimulation with acetylcholine or chew-
ing. Frey’s syndrome may be caused by aberrant innervation of sympathetically innervated
facial sweat glands by postganglionic parasympathetic nerve fibers in damaged tissue.5 There-
fore, mastication, which releases acetylcholine from parasympathetic nerve endings, results
Received April 26, 2019
Revised June 18, 2019 in sweating. The mechanism underlying Frey’s syndrome in our patient was unclear, since the
Accepted June 26, 2019 patient had undergone neck surgery during infancy and the symptoms occurred only when
Correspondence he reached his thirties. However, a case of delayed Frey’s syndrome occurring 40 years after
Hye Lim Lee, MD surgery has been reported.6 Our patient reported hypesthesia at the operation site, which might
Department of Neurology,
Korea University Guro Hospital,
indicate the presence of similar aberrant re-innervation of the postganglionic nerve fibers near
Korea University College of Medicine, the site of his neck operation.
148 Gurodong-ro, Guro-gu, The QSART is a quantitative type of sudometry that is commonly used to diagnose early dia-
Seoul 08308, Korea
Tel +82-2-2626-3046 cc This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Com-

Fax +82-2-2626-2249 mercial License (https://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial


E-mail ‌chiffin@hanmail.net use, distribution, and reproduction in any medium, provided the original work is properly cited.

Copyright © 2019 Korean Neurological Association 585


JCN Frey’s Syndrome Using QSART

betic polyneuropathy or small-fiber neuropathy.7 A few studies Focal hyperhidrosis could have a negative impact on social
have assessed the effectiveness of treatments in patients with interactions, resulting in a reduced quality of life.3 It is there-
hyperhidrosis,8,9 and found the QSART to be useful in measur- fore important to diagnose focal hyperhidrosis and assess the
ing the therapeutic effects. The present case has demonstrated possible treatment effects in order to treat it satisfactorily, for
that the QSART may also be equally useful in Frey’s syndrome. which we believe that the QSART may be helpful.

1,500

Test site: Lt. cheek

Sweat rate (nL/min)


Total volume (μL): 9.784 1,000
Totalized time: 10:00

Chewing start

Chewing stop
Response latency: 0:20
Baseline rate (nL/min): 98.8
500
Ending offset (nL/min): 416.1

0
00:00 05:00 10:00
500

400
Test site: Rt. cheek
Sweat rate (nL/min)

Total volume (μL): 0.051 300


Totalized time: 10:00
Chewing start

Chewing stop
Response latency: 0:15
200
Baseline rate (nL/min): 57.5
Ending offset (nL/min): 3.6
100

0
A   00:00 05:00 10:00

1,200

1,000
Sweat rate (nL/min)

Test site: Lt. cheek 800


Ach injection

Total volume (μL): 4.697


Totalized time: 10:00 600
Response latency: 0:12
Baseline rate (nL/min): 328.5 400
Ending offset (nL/min): 366.1
200

0
00:00 05:00 10:00
500

400
Sweat rate (nL/min)

Test site: Rt. cheek


Total volume (μL): 0.225 300
Totalized time: 10:00
Ach injection

Response latency: 1:12


200
Baseline rate (nL/min): 62.7
Ending offset (nL/min): 0.9
100

0
B 00:00 05:00 10:00

Fig. 1. The results of QSART. A: QSART findings in both auriculotemporal areas. Excessive sweating was observed on the left side after chewing gum.
B: The sweat volume was larger in the left than the right auriculotemporal area. C: Sweating was significantly reduced on the left side after treatment
with aluminum chloride cream at a 2-month follow-up. QSART: quantitative sudomotor axon reflex test.

586 J Clin Neurol 2019;15(4):585-587


Kim J et al. JCN
500

400
Test site: Lt. cheek

Sweat rate (nL/min)


Total volume (μL): 1.055 300

Chewing start
Totalized time: 10:00

Chewing stop
Response latency: 0:21
200
Baseline rate (nL/min): 81.2
Ending offset (nL/min): 21.2
100

0
00:00 05:00 10:00
500

400
Test site: Rt. cheek Sweat rate (nL/min)
Total volume (μL): 0.323 300
Totalized time: 10:00

Chewing start

Chewing stop
Response latency: 1:06 200
Baseline rate (nL/min): 59.6
Ending offset (nL/min): 11.5
100

0
C 00:00 05:00 10:00

Fig. 1. The results of QSART. A: QSART findings in both auriculotemporal areas. Excessive sweating was observed on the left side after chewing
gum. B: The sweat volume was larger in the left than the right auriculotemporal area. C: Sweating was significantly reduced on the left side after
treatment with aluminum chloride cream at a 2-month follow-up. QSART: quantitative sudomotor axon reflex test (continued).

Author Contributions 2. Motz KM, Kim YJ. Auriculotemporal syndrome (Frey syndrome).
Conceptualization: Hye Lim Lee. Investigation: Jaehwan Kim, YeEun Kim, Otolaryngol Clin North Am 2016;49:501-509.
Minkyung Kim. Methodology: Jaehwan Kim, YeEun Kim, Minkyung Kim. 3. Haider A, Solish N. Focal hyperhidrosis: diagnosis and management.
Supervision: Hye Lim Lee. Writing—original draft: Jaehwan Kim. Writ- CMAJ 2005;172:69-75.
ing—review & editing: Hye Lim Lee. 4. Sletten DM, Weigand SD, Low PA. Relationship of Q‐sweat to quan-
titative sudomotor axon reflex test (QSART) volumes. Muscle Nerve
ORCID iDs 2010;41:240-246.
5. Schlereth T, Dieterich M, Birklein F. Hyperhidrosis--causes and treat-
Jaehwan Kim https://orcid.org/0000-0002-3415-6877
ment of enhanced sweating. Dtsch Arztebl Int 2009;106:32-37.
Minkyung Kim https://orcid.org/0000-0003-0694-9906
6. Chamisa I. Frey’s syndrome--unusually long delayed clinical onset
YeEun Kim https://orcid.org/0000-0002-9790-123X
post-parotidectomy: a case report. Pan Afr Med J 2010;5:1.
Hye Lim Lee https://orcid.org/0000-0002-9800-3263
7. Thaisetthawatkul P, Fernandes Filho JA, Herrmann DN. Contribution
of QSART to the diagnosis of small fiber neuropathy. Muscle Nerve
Conflicts of Interest
2013;48:883-888.
The authors have no potential conflicts of interest to disclose. 8. Braune C, Erbguth F, Birklein F. Dose thresholds and duration of the
local anhidrotic effect of botulinum toxin injections: measured by
REFERENCES sudometry. Br J Dermatol 2001;144:111-117.
9. Rystedt A, Swartling C, Naver H. Anhidrotic effect of intradermal in-
1. Malatskey S, Rabinovich I, Fradis M, Peled M. Frey syndrome--de- jections of botulinum toxin: a comparison of different products and
layed clinical onset: a case report. Oral Surg Oral Med Oral Pathol concentrations. Acta Derm Venereol 2008;88:229-233.
Oral Radiol Endod 2002;94:338-340.

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