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Case Report

Isolated Cold‑Induced Sweating in a Young Saudi Male: Case Report


Ali Saeed Alghamdi, Dhafer Hafez, Noah Alghanemy, Hamza Alshehri
Department of Dermatology, Aseer Central Hospital, Abha, Saudi Arabia

Abstract
Cold‑induced sweating is a rare condition, which is reported in the literature as part of Crisponi syndrome in which patients have many
neurological and skeletal abnormalities. We report a case of isolated nonsyndromic cold‑induced sweating in a 24‑year‑old Saudi male. The
patient was treated with many treatments, including clonidine 0.1 mg, amitriptyline, and fluoxetine, but these were discontinued due to side
effects. The patient was treated with onabotulinum toxin A with partial satisfactory result. Treatment options for cold‑induced sweating are
limited, and in this case, inadequate.

Keywords: Crisponi, hyperhidrosis, sweating, syndrome

Introduction sweating of the lower limbs. Hair and mucous membrane


were normal.
Cold‑induced sweating is a paradoxical sweating occurring
after exposure to cold weather. It is usually present as a During 6 years of follow‑up, he tried many medications without any
manifestation of cold‑induced sweating syndrome (CISS) or benefit. Clonidine 0.1 mg was stopped due to postural hypotension.
Crisponi syndrome or rarely in an acquired idiopathic form. Amitriptyline (25 mg) was stopped due to drowsiness; fluoxetine
We presenting herein a case of idiopathic acquired CISS in was also tried, but was also discontinued due to drowsiness.
Saudi Arabia. In March 2019, an iodine‑starch test was done to evaluate the
extent of the truncal sweating [Figure 3]. The entire trunk and
Case Report upper limbs exhibited sweating. During the procedure, in which
A 24‑year‑old Saudi Arabian male patient with no known medical the room air was cold, he was profusely sweating. Based on
problems presented to the Asser Central Hospital’s Dermatology the patient’s report of more sweat from both axillae and chest,
Clinic 6 years ago with excessive sweating in his upper half of these areas were injected with 200 U of onabotulinum toxin A,
the body (trunk, both arms, and forearms). The sweating occurred which helps the patient in the injected area only.
only in the cold weather and began in early childhood. Always
wearing heavy clothes helped, and the sweating diminished Discussion
dramatically when the climate was hot or warm. Cold‑induced sweating or hyperhidrosis represents a
The sweating during cold is severe enough to completely wet paradoxical sweating that occurs in response to cold
all his clothes over the upper part of his body [Figure 1]. His environment. Most of the cases of cold‑induced sweating in
medical history was unremarkable. No one else in his family the literature were reported as a manifestation of Crisponi
had the condition. syndrome or CISS. Crisponi syndrome (MIM #601378)
The general examination revealed no obvious physical
abnormalities. His vital signs were all within normal limits, Address for correspondence: Dr. Ali Saeed Alghamdi,
except for blood pressure which was 90/50. The neurological Department of Dermatology, Aseer Central Hospital, Abha, Saudi Arabia.
E‑mail: asgh60@yahoo.com
and skeletal examinations were normal. The only abnormal
Received: 21 August 2019, Revised: 26 August 2019,
finding was profuse sweating of the trunk and both upper Accepted: 11 November 2019, Published: 27 March 2020
limbs. His clothes were wet [Figure 2]; there was no excessive
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DOI: How to cite this article: Alghamdi AS, Hafez D, Alghanemy N,


10.4103/jdds.jdds_48_19 Alshehri H. Isolated cold‑induced sweating in a young Saudi male: Case
report. J Dermatol Dermatol Surg 2020;24:62-4.

62 © 2020 Journal of Dermatology and Dermatologic Surgery | Published by Wolters Kluwer - Medknow
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Alghamdi, et al.: Isolated cold- induced sweating

micrognathia.[1‑6] The syndrome is caused by mutations in the


cytokine receptor‑like factor 1 gene.[7‑10] Mutations of this gene
have also been associated with CISS (MIM #272430). Most
of the reported cases were from Turkey and Italy; a few cases
were from Norway, Australia, and Canada, with one report
of two siblings from Saudi Arabia.[11] Few cases of idiopathic
acquired cold‑induced sweating have been reported, and most
of them are from Japan.[12‑15]
Many treatment options have been mentioned including
α2‑adrenoreceptor agonist clonidine with a maximum dose
of 3 µg/kg bodyweight per day and moxonidine with a
maximum dose of 6 µg/(kg/d) with reasonable response.[4,16]
An α2‑adrenoreceptor agonist such as clonidine may reduce
sweating by acting on the presynaptic adrenoceptor to reduce
the release of noradrenaline. In case of our patient, no treatment
Figure 1: The clothes get wet over the whole trunk and upper limbs was effective, except for onabotulinum toxin A which was
partially effective due to the limited area that we could inject.
Declaration of patient consent
The authors certify that they have obtained all appropriate
patient consent forms. In the form the patient(s) has/have
given his/her/their consent for his/her/their images and other
clinical information to be reported in the journal. The patients
understand that their names and initials will not be published
and due efforts will be made to conceal their identity, but
anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.

Figure 2: Profuse sweating after exposure to cold air References


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64 Journal of Dermatology and Dermatologic Surgery  ¦  Volume 24  ¦  Issue 1  ¦  January-June 2020

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