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Aquagenic Urticaria: a Report of Two Cases Ann Dermatol Vol Aquagenic Urticaria: A Report of Two Cases Ann Dermatol Vol. 23, Suppl. 3, 2011 http://dx.doi.org/10.5021/ad.2011.23.S3.S371 CASE REPORT Aquagenic Urticaria: A Report of Two Cases Hoon Park, M.D., Hee Su Kim, M.D., Dong Soo Yoo, M.D., Jin Woo Kim, M.D., Chul Woo Kim, M.D.1, Sang Seok Kim, M.D.1, Jong Ik Hwang, M.D., Jun Young Lee, M.D., Yoon Jeong Choi, M.D. Department of Dermatology, College of Medicine, The Catholic University of Korea, 1College of Medicine, Hallym University, Seoul, Korea Aquagenic urticaria is a rare form of physical urticaria, in literature. To the best of our knowledge, only one case of which contact with water evokes wheals. A 19-year-old man AU has been reported in the Korean literature. AU is a and a 4-year-old boy complained of recurrent episodes of rare form of physical urticaria, in which contact with urticaria. Urticaria appeared while taking a bath or a shower, water, regardless of its temperature and source, evokes in the rain, or in a swimming pool. Well-defined pin head to wheals2. Skin lesions may be confused with eruptions of small pea-sized wheals surrounded by variable sized cholinergic urticaria; however, they cannot be evoked by erythema were provoked by contact with water on the face, exercise, sweating, heat, or emotional stress2. Lesions are neck, and trunk, regardless of its temperature or source. located mainly on the upper body (neck, trunk, shoulder, Results from a physical examination and a baseline labo- arms, and back)2. We present two cases of AU in young ratory evaluation were within normal limits. Treatment of the patients. Clinical manifestations, diagnostic tests, and ava- 19-year-old man with 180 mg fexofenadine daily was suc- ilable treatments are reviewed. cessful to prevent the wheals and erythema. Treatment with 5 ml ketotifen syrup bid per day resulted in improvement of CASE REPORT symptoms in the 4-year-old boy. (Ann Dermatol 23(S3) S371 ∼S374, 2011) The first case was that of a 19-year-old man who was referred to our department due to recurrent episodes of -Keywords- urticaria. He presented with a 3-year history of pinpoint Aquagenic urticaria, Water sized wheals affecting the shoulders, arms, trunk, abdomen, and back when he took a bath or shower. These symptoms appeared within 10 to 20 minutes of INTRODUCTION contact with water and provoked intense pruritus. Each episode lasted for 20∼40 minutes and spontaneously Aquagenic urticaria (AU) was first described by Shelley resolved. The patient did not complain of angioedema, and Rawnsley1, who reported three cases in 1964, and wheezing, or dyspnea with these episodes. He had no fewer than 100 cases have since been published in the personal history of allergies or drug allergy and no family history of urticaria. The diagnosis of AU was confirmed by applying a room temperature wet compress to the upper Received September 17, 2010, Revised June 14, 2011, Accepted for publication June 14, 2011 body for 30 minutes (Fig. 1). A cold-water and hot-water Corresponding author: Hoon Park, M.D., Department of Dermatology, compress were also applied for 30 minutes. In all cases, Uijeongbu St. Mary’s Hospital, College of Medicine, The Catholic the response to the tests was positive, with induction of University of Korea, 65-1 Geumo-dong, Uijeongbu 480-130, Korea. pinpoint wheals at the site of compress application. A Tel: 82-31-820-3509, Fax: 82-31-846-4799, E-mail: ph7163@han- water-challenge test with tap water, distilled water, and mail.net normal saline showed similar results. A pressure test, This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http:// exercise test, and ice-cube test were performed to rule out creativecommons.org/licenses/by-nc/3.0) which permits unrestricted other physical urticaria. A 6,000 gm weight was applied to non-commercial use, distribution, and reproduction in any medium, the skin for a period of 20 minutes. After 8 hours, no provided the original work is properly cited. lesions had appeared. Lesions were not reproduced after Vol. 23, Suppl. 3, 2011 S371 H Park, et al Fig. 1. Pin-head to match-head sized wheal surrounded by Fig. 2. Pin-head sized wheal surrounded by erythema on the erythema on the upper trunk after the water provocation test. face after the water provocation test. running. An ice-cube-filled plastic bag was applied to the contact with water. patient’s forearm for 20 minutes. No lesions were noted Both patients were asymptomatic upon water ingestion. on the forearm after removal of cold stimulation. Based on Symptoms appeared after contact with water regardless of these findings, a diagnosis of AU was made. He had no its temperature or source. In both cases, the physical treatment history before visiting our clinic. Fexofenadine examination revealed no other abnormalities. Results from was prescribed initially at a dose of 180 mg daily for laboratory tests, including a complete blood count with symptom relief. After 2 weeks, no lesions had developed differential, liver function tests, electrolytes, complement on contact with water. Once the symptoms were relieved, (C)3, C4, and urinalysis were normal. the dose was reduced to 180 mg every other day. The patient was still symptom free at the 1 year follow-up. DISCUSSION The second case was that of a 4-year-old boy who visited our department due to recurrent episodes of urticaria. He AU is more common in women than in men and appears presented with a 1-year history of pinhead sized wheals during puberty or several years later3,4. Most cases are affecting the face, extremities, chest, abdomen, and back sporadic; however, familial AU has been reported3,5,6. The when he took a bath or shower. These symptoms ap- clinical picture consists of pruritic follicular wheals on peared within 10 to 30 minutes of contact with water and skin areas that have come in contact with water. The small provoked pruritus. Each episode lasted for 30 to 60 mi- pruritic wheals (1∼3 mm in diameter) appear in a cho- nutes and showed spontaneous resolution. The patient did linergic urticaria-like erythematous areas several to 30 not complain of angioedema, wheezing, or dyspnea with minutes after exposure to water and are usually located on these episodes. He had no personal history of allergies or the neck, upper trunk, and arms. Wheals generally fade drug allergy and no family history of urticaria. The diag- within 30 to 60 minutes. Wheal formation is not influ- nosis of AU was confirmed by applying a room tem- enced by temperature or water source. Alcohol and other perature wet compress to the face for 30 minutes (Fig. 2). organic solvents applied to the skin do not cause wheal The response to the test was positive, with induction of formation2. Systemic symptoms are rare but have been pinhead sized wheals at the site of compress application. reported7,8. AU is sometimes associated with other forms His mother said that the lesions were not reproduced after of physical urticaria6,8. sweating when the boy played with his friends. An Evaluations for AU consist of a clinical history and water ice-cube-filled plastic bag was applied to the patient’s challenge test2,7. The standard test for AU is application of forearm for 20 minutes. No lesions were noted on the a 35oC water compress to the upper body for 30 mi- forearm after removal of cold stimulation. Based on these nutes2,9. Water of any temperature can provoke AU; how- findings, a diagnosis of AU was made. He had no treat- ever, keeping the compress at room temperature avoids ment history before visiting our clinic. Ketotifen syrup was confusion with cold-induced or local heat urticaria. In prescribed initially at a dose of 5 ml bid per day for symp- addition, a forearm or hand can be immersed in water of tom relief. After 4 weeks, no lesions had developed on varying temperatures9. A diagnosis of AU requires ex- S372 Ann Dermatol Aquagenic Urticaria: A Report of Two Cases 11 Table 1. Summary of the provocation test of physical urticaria mation when applied to the skin before water contact . However, another study failed to reproduce this finding Physical urticaria Provocation test when pretreatment with atropine did not result in sup- Pressure urticaria A 6,000 gm weight is applied to the skin pression of subsequent wheal formation6. Methacholine for 20 minutes. injection testing is negative in patients with AU; however, Cold urticaria An ice-cube-filled plastic bag is applied 2 to the patient’s forearm for 20 minutes. it is often positive in cholinergic urticaria . Serum his- 2 Heat urticaria A heated cylinder (50∼55oC) is applied tamine levels are variable from patient to patient . Anti- to the upper trunk for 30 minutes histamines have been used to treat AU; however, the the- 2 Cholinergic urticaria Exercise test until sweating rapeutic effect and prognosis vary . In some cases, com- Warm bath －43oC plete control of symptoms with antihistamine has been Aquagenic urticaria The application of a room temperature reported, whereas in other cases, there is a failure to wet compress to the upper body for adequately control symptoms8,15. Refractory cases have 30 minutes －35oC been treated with ultraviolet (UV) radiation (both psoralen plus UVA therapy and UVB), either alone or in com- bination with antihistamines. It is hypothesized that the clusion of other types of physical urticaria, so an exercise effect of ultraviolet therapy is mediated by thickening of test and ice cube test should be performed to rule out the epidermis, which may prevent water penetration, other types of physical urticaria9.
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