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Year : 2020  |  Volume : 24  |  Issue : 1  |  Page : 62-64

Isolated cold-induced sweating in a young Saudi male: Case report

Department of Dermatology, Aseer Central Hospital, Abha, Saudi Arabia

Date of Submission21-Aug-2019
Date of Acceptance11-Nov-2019
Date of Web Publication27-Mar-2020

Correspondence Address:
Dr. Ali Saeed Alghamdi
Department of Dermatology, Aseer Central Hospital, Abha
Saudi Arabia
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jdds.jdds_48_19

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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

How to cite this article:
Alghamdi AS, Hafez D, Alghanemy N, Alshehri H. Isolated cold-induced sweating in a young Saudi male: Case report. J Dermatol Dermatol Surg 2020;24:62-4

How to cite this URL:
Alghamdi AS, Hafez D, Alghanemy N, Alshehri H. Isolated cold-induced sweating in a young Saudi male: Case report. J Dermatol Dermatol Surg [serial online] 2020 [cited 2021 Jun 12];24:62-4. Available from: https://www.jddsjournal.org/text.asp?2020/24/1/62/281422

  Introduction Top

Cold-induced sweating is a paradoxical sweating occurring after exposure to cold weather. It is usually present as a manifestation of cold-induced sweating syndrome (CISS) or Crisponi syndrome or rarely in an acquired idiopathic form. We presenting herein a case of idiopathic acquired CISS in Saudi Arabia.

  Case Report Top

A 24-year-old Saudi Arabian male patient with no known medical problems presented to the Asser Central Hospital's Dermatology Clinic 6 years ago with excessive sweating in his upper half of the body (trunk, both arms, and forearms). The sweating occurred only in the cold weather and began in early childhood. Always wearing heavy clothes helped, and the sweating diminished dramatically when the climate was hot or warm.

The sweating during cold is severe enough to completely wet all his clothes over the upper part of his body [Figure 1]. His medical history was unremarkable. No one else in his family had the condition.
Figure 1: The clothes get wet over the whole trunk and upper limbs

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The general examination revealed no obvious physical abnormalities. His vital signs were all within normal limits, except for blood pressure which was 90/50. The neurological and skeletal examinations were normal. The only abnormal finding was profuse sweating of the trunk and both upper limbs. His clothes were wet [Figure 2]; there was no excessive sweating of the lower limbs. Hair and mucous membrane were normal.
Figure 2: Profuse sweating after exposure to cold air

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During 6 years of follow-up, he tried many medications without any benefit. Clonidine 0.1 mg was stopped due to postural hypotension. Amitriptyline (25 mg) was stopped due to drowsiness; fluoxetine was also tried, but was also discontinued due to drowsiness.

In March 2019, an iodine-starch test was done to evaluate the extent of the truncal sweating [Figure 3]. The entire trunk and upper limbs exhibited sweating. During the procedure, in which the room air was cold, he was profusely sweating. Based on the patient's report of more sweat from both axillae and chest, these areas were injected with 200 U of onabotulinum toxin A, which helps the patient in the injected area only.
Figure 3: Iodine-starch test

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  Discussion Top

Cold-induced sweating or hyperhidrosis represents a paradoxical sweating that occurs in response to cold environment. Most of the cases of cold-induced sweating in the literature were reported as a manifestation of Crisponi syndrome or CISS. Crisponi syndrome (MIM #601378) is a rare severe infantile autosomal recessive disorder characterized by paroxysmal contractions of the facial muscles, hyperthermia, feeding difficulties, and camptodactyly and dysmorphic facial features including prominent cheek bone, anteverted nostrils with broad nose, small mouth, and micrognathia.[1],[2],[3],[4],[5],[6] The syndrome is caused by mutations in the cytokine receptor-like factor 1 gene.[7],[8],[9],[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],[13],[14],[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 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.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Sohar E, Shoenfeld Y, Udassin R, Magazanik A, Revach M. Cold-induced profuse sweating on back and chest. A new genetic entity? Lancet 1978;2:1073-4.  Back to cited text no. 1
Alhashem AM, Majeed-Saidan MA, Ammari AN, Alrakaf MS, Nojoom M, Maddirevula S, et al. Crisponi/CISS1 syndrome: A case series. Am J Med Genet A 2016;170A:1236-41.  Back to cited text no. 2
Hahn AF, Waaler PE, Kvistad PH, Bamforth JS, Miles JH, McLeod JG, et al. Cold-induced sweating syndrome: CISS1 and CISS2: Manifestations from infancy to adulthood. Four new cases. J Neurol Sci 2010;293:68-75.  Back to cited text no. 3
Hahn AF, Jones DL, Knappskog PM, Boman H, McLeod JG. Cold-induced sweating syndrome: A report of two cases and demonstration of genetic heterogeneity. J Neurol Sci 2006;250:62-70.  Back to cited text no. 4
Bayraktar-Tanyeri B, Hepokur M, Bayraktar S, Persico I, Crisponi L. A new case series of crisponi syndrome in a Turkish family and review of the literature. Clin Dysmorphol 2017;26:66-72.  Back to cited text no. 5
Crisponi G. Autosomal recessive disorder with muscle contractions resembling neonatal tetanus, characteristic face, camptodactyly, hyperthermia, and sudden death: A new syndrome? Am J Med Genet 1996;62:365-71.  Back to cited text no. 6
Crisponi L, Crisponi G, Meloni A, Toliat MR, Nurnberg G, Usala G, et al. Crisponi syndrome is caused by mutations in the CRLF1 gene and is allelic to cold-induced sweating syndrome type 1. Am J Hum Genet 2007;80:971-81.  Back to cited text no. 7
Knappskog PM, Majewski J, Livneh A, Nilsen PT, Bringsli JS, Ott J, et al. Cold-induced sweating syndrome is caused by mutations in the CRLF1 gene. Am J Hum Genet 2003;72:375-83.  Back to cited text no. 8
Dagoneau N, Bellais S, Blanchet P, Sarda P, Al-Gazali LI, Di Rocco M, et al. Mutations in cytokine receptor-like factor 1 (CRLF1) account for both crisponi and cold-induced sweating syndromes. Am J Hum Genet 2007;80:966-70.  Back to cited text no. 9
González Fernández D, Lázaro Pérez M, Santillán Garzón S, Alvarez Martínez V, Encinas Madrazo A, Fernández Toral J, et al. Cold-induced sweating syndrome type 1, with a CRLF1 level mutation, previously associated with crisponi syndrome. Dermatology 2013;227:126-9.  Back to cited text no. 10
Alallah J, Shawli A, Hakami F. A case report of crisponi/cold-induced sweating syndrome 1 in a Saudi family. J Clin Neonatol 2018;7:273-8.  Back to cited text no. 11
  [Full text]  
Nakazato Y, Tamura N, Ninomiya M, Yoshimaru K, Araki N, Yamamoto T, et al. Acquired idiopathic cold-induced hyperhidrosis in a case of dementia with lewy bodies. Neurol Clin Pract 2015;5:357-9.  Back to cited text no. 12
Nakazato Y, Tamura N, Ohkuma A, Ninomiya M, Araki N. Acquired idiopathic cold-induced hyperhidrosis [in Japanese]. Auton Nerv Syst 2008;45:259-62.  Back to cited text no. 13
Yoshimaru K, Nakatzato Y, Tamura N, Araki N. Two cases of acquired cold-induced hyperhidrosis. Jpn J Persp Res 2010;17:63-5.  Back to cited text no. 14
Idiaquez J, Fadic R, Verdugo R, Idiaquez JF, Iodice V, Low DA, et al. Pure autonomic failure with cold induced sweating. Auton Neurosci 2013;176:98-100.  Back to cited text no. 15
Herholz J, Crisponi L, Mallick BN, Rutsch F. Successful treatment of cold-induced sweating in crisponi syndrome and its possible mechanism of action. Dev Med Child Neurol 2010;52:494-7.  Back to cited text no. 16


  [Figure 1], [Figure 2], [Figure 3]


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