Case Report Pure Cold-Induced Cholinergic Urticaria in a...
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Case Report Pure Cold-Induced Cholinergic Urticaria in a Pediatric Patient Tina Abraham, 1 David P. McGarry, 2 John Frith, 1 Jason Casselman, 1 Haig Tcheurekdjian, 3 and Robert Hostoffer 3 1 Department of Allergy and Immunology, University Hospitals, Regional Hospitals, Richmond Heights, OH, USA 2 Department of Pediatrics, Akron Children’s Hospital, Akron, OH, USA 3 Allergy/Immunology Associates, Inc., Mayfield Heights, OH, USA Correspondence should be addressed to David P. McGarry; [email protected] Received 14 July 2016; Accepted 10 November 2016 Academic Editor: Necla Tulek Copyright © 2016 Tina Abraham et al. is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Cold urticaria and cholinergic urticaria are two distinct entities. e presentation of exclusive cold-induced cholinergic urticaria is very rare. e patient described herein had experienced urticaria in the exclusive setting of exercising in a cold environment. Urticarial testing including laboratory and in-office testing was all negative. e patient has prevented urticaria symptoms with oral antihistamine therapy. Pure cold-induced cholinergic urticaria is rarely described in literature. is form of urticaria has yet to be described in a pediatric patient. 1. Case Report Cold urticaria and cholinergic urticaria have been clearly defined as two separate entities. Review of literature shows that the cooccurrence of both as one distinct clinical feature is rare. While there have been a few reported cases of isolated cold-induced urticaria with cholinergic features in adults, we present the first case of pure cold-induced cholinergic urticaria in a pediatric patient. A 16-year-old female presented with complaints of urticaria for the past 18 months. She developed the urticaria and pruritus exclusively in the setting of exercising in a cold environment. Urticaria was never triggered with only cold exposure or exercise in a warm environment. ere was no significant contributing family history of disease. She also denied any other associated symptoms such as swelling, difficulty in breathing, dizziness, or any systemic symptoms. e patient’s skin lesions were described as erythematous, pinpoint papules which covered the entire body but spared the face. An ice cube test was performed and was negative. All lab- oratory findings of a complete blood count, basic metabolic panel, tryptase, C-reactive protein, aspartate aminotrans- ferase (AST), alanine aminotransferase (ALT), bilirubin, alkaline phosphatase, chronic urticarial index, and thyroid panel were within normal limits. e patient was initiated on oral hydroxyzine therapy for cold-induced cholinergic urticaria and saw success with symptomatic improvement. e exact mechanisms of both cold and cholinergic urticaria are unclear. e first report of a patient with cold urticaria was described in 1866 [1]. Cold urticaria is believed to be associated skin antigens manifested during cold expo- sure which are detected by IgE antibodies leading to mast cell activation and histamine release [2]. Cholinergic urticaria was first described in 1924 [3]. A suggested mechanism of cholinergic urticaria includes an allergic response to sweat antigens [2]. However, it is also seen in a patient with anhidrosis [4]. In cold-induced cholinergic urticaria, Ormerod et al. [5] proposes a common circulating factor, like IgE, lead- ing to urticaria. Kaplan and Garofalo [6] first described cold-induced cholinergic urticaria in 1981 in four patients who clinically demonstrated cholinergic appearing urticaria caused by cold exposure. ese patients showed morpho- logical cholinergic urticaria in the setting of negative ice cube testing. Only one of these patients, an adult, had a similar history to our patient in that urticaria presented with exercising only in a cold environment. Geller [7] described a 9-year-old patient with cholinergic appearing urticaria with cold exposure but was not induced with physical activity. In Hindawi Publishing Corporation Case Reports in Immunology Volume 2016, Article ID 7425601, 2 pages http://dx.doi.org/10.1155/2016/7425601
Transcript of Case Report Pure Cold-Induced Cholinergic Urticaria in a...
Case ReportPure Cold-Induced Cholinergic Urticaria in a Pediatric Patient
Tina Abraham,1 David P. McGarry,2 John Frith,1 Jason Casselman,1
Haig Tcheurekdjian,3 and Robert Hostoffer3
1Department of Allergy and Immunology, University Hospitals, Regional Hospitals, Richmond Heights, OH, USA2Department of Pediatrics, Akron Children’s Hospital, Akron, OH, USA3Allergy/Immunology Associates, Inc., Mayfield Heights, OH, USA
Correspondence should be addressed to David P. McGarry; [email protected]
Received 14 July 2016; Accepted 10 November 2016
Academic Editor: Necla Tulek
Copyright © 2016 Tina Abraham et al. This is an open access article distributed under the Creative Commons Attribution License,which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Cold urticaria and cholinergic urticaria are two distinct entities. The presentation of exclusive cold-induced cholinergic urticariais very rare. The patient described herein had experienced urticaria in the exclusive setting of exercising in a cold environment.Urticarial testing including laboratory and in-office testing was all negative. The patient has prevented urticaria symptoms withoral antihistamine therapy. Pure cold-induced cholinergic urticaria is rarely described in literature. This form of urticaria has yetto be described in a pediatric patient.
1. Case Report
Cold urticaria and cholinergic urticaria have been clearlydefined as two separate entities. Review of literature showsthat the cooccurrence of both as one distinct clinical featureis rare. While there have been a few reported cases of isolatedcold-induced urticaria with cholinergic features in adults,we present the first case of pure cold-induced cholinergicurticaria in a pediatric patient.
A 16-year-old female presented with complaints ofurticaria for the past 18 months. She developed the urticariaand pruritus exclusively in the setting of exercising in acold environment. Urticaria was never triggered with onlycold exposure or exercise in a warm environment. Therewas no significant contributing family history of disease. Shealso denied any other associated symptoms such as swelling,difficulty in breathing, dizziness, or any systemic symptoms.The patient’s skin lesions were described as erythematous,pinpoint papules which covered the entire body but sparedthe face.
An ice cube test was performed and was negative. All lab-oratory findings of a complete blood count, basic metabolicpanel, tryptase, C-reactive protein, aspartate aminotrans-ferase (AST), alanine aminotransferase (ALT), bilirubin,alkaline phosphatase, chronic urticarial index, and thyroid
panel were within normal limits. The patient was initiatedon oral hydroxyzine therapy for cold-induced cholinergicurticaria and saw success with symptomatic improvement.
The exact mechanisms of both cold and cholinergicurticaria are unclear. The first report of a patient with coldurticaria was described in 1866 [1]. Cold urticaria is believedto be associated skin antigens manifested during cold expo-sure which are detected by IgE antibodies leading to mastcell activation and histamine release [2]. Cholinergic urticariawas first described in 1924 [3]. A suggested mechanism ofcholinergic urticaria includes an allergic response to sweatantigens [2]. However, it is also seen in a patient withanhidrosis [4].
In cold-induced cholinergic urticaria, Ormerod et al.[5] proposes a common circulating factor, like IgE, lead-ing to urticaria. Kaplan and Garofalo [6] first describedcold-induced cholinergic urticaria in 1981 in four patientswho clinically demonstrated cholinergic appearing urticariacaused by cold exposure. These patients showed morpho-logical cholinergic urticaria in the setting of negative icecube testing. Only one of these patients, an adult, had asimilar history to our patient in that urticaria presented withexercising only in a cold environment. Geller [7] described a9-year-old patient with cholinergic appearing urticaria withcold exposure but was not induced with physical activity. In
Hindawi Publishing CorporationCase Reports in ImmunologyVolume 2016, Article ID 7425601, 2 pageshttp://dx.doi.org/10.1155/2016/7425601
2 Case Reports in Immunology
a study of 220 patients with a history of cold urticaria, fifteenconcomitantly had cholinergic urticaria as determined bypositive ice cube and methacholine testing [8]. Two had neg-ative ice cube tests but positive methacholine tests and werethus called cold-induced cholinergic urticaria. Ormerod et al.[5] described thirteen patients with cold-induced cholinergicurticaria defined by testing. Each had cholinergic urticariafrom sweat provoking stimuli and then morphologicallycholinergic urticaria in the setting of cold contact or coldexposure. Other patients have been described who developcholinergic and cold urticaria but are not exclusively seentogether [2]. Our patient is unique in that the urticaria is onlyseen with exercise in the cold.
Outside of preventing precipitating factors, treatmentof cold-induced cholinergic urticaria should initiate withantihistamine therapy. In our case and other descriptionsof the disease, antihistamines were used and have generallybeen successful. In the setting of antihistamine failure, omal-izumab has been suggested.
The clinical discernment of urticarial disease requires astrict history collection strategy in order to remove triggersaccurately. To our knowledge, this is the first pediatric patientwith pure cold-induced cholinergic urticaria.
Competing Interests
The authors of this manuscript have no financial competinginterests to disclose.
Authors’ Contributions
Tina Abraham, DO, David P. McGarry, DO, John Frith, DO,and Robert Hostoffer, DO, were involved in the conceptionand design of the study, data generation, analysis, andinterpretation of the data, as well as preparation and clinicalrevision of the manuscript. Jason Casselman, DO, and HaigTcheurekdjian, MD, were involved in the conception anddesign of the study and data generation, as well as analysisand interpretation of the data.
References
[1] E. F. Hochstadter and M. Ben-Shoshan, “Cold-induced urticar-ia: challenges in diagnosis andmanagement,” BMJCase Reports,2013.
[2] B. Torabi and M. Ben-Shoshan, “The association of cholinergicand cold-induced urticaria: diagnosis and management,” BMJCase Reports, vol. 2015, 2015.
[3] W. W. Duke, “Urticaria caused specifically by the actions ofphysical agents,” Journal of the American Medical Association,vol. 83, no. 1, p. 3, 1924.
[4] Y.Oda, A. Fukunaga,M. Tsujimoto,M.Hatakeyama, K.Washio,and C. Nishigori, “Combined cholinergic urticaria and cold-induced cholinergic urticaria with acquired idiopathic general-ized anhidrosis,” Allergology International, vol. 64, no. 2, article40, pp. 214–215, 2015.
[5] A. D. Ormerod, A. Kobza-Black, A. Milford-Ward, and M. W.Greaves, “Combined cold urticaria and cholinergic urticaria
clinical characterization and laboratory findings,” British Jour-nal of Dermatology, vol. 118, no. 5, pp. 621–627, 1988.
[6] A. P. Kaplan and J. Garofalo, “Identification of a new physicallyinduced urticaria: cold-induced cholinergic urticaria,” Journalof Allergy and Clinical Immunology, vol. 68, no. 6, pp. 438–441,1981.
[7] M. Geller, “Cold-induced cholinergic urticaria—case report,”Annals of Allergy, vol. 63, no. 1, pp. 29–30, 1989.
[8] H. Neittaanmaki, “Cold urticaria,” Journal of the AmericanAcademy of Dermatology, vol. 13, no. 4, pp. 636–644, 1985.
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