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      Idiopathic Aquagenic Wrinkling of the Palms in Korean Patients

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          Abstract

          Dear Editor: A 23-year-old Korean man presented with wrinkled, whitish plaques on both palms with a 2-month history. On physical examination, whitish discoloration and intense edematous wrinkling appeared on his palms within 3 min after water immersion. This condition was transient and almost completely resolved within 25 min after drying of his palms (Fig. 1). At the onset of this phenomenon, dullness and mild hyperhidrosis were present on his palms. Neither his feet nor any other part of his body was affected. He had no relevant family or medical history of systemic diseases. He was not taking any regular medication. The laboratory and radiologic examinations showed nonspecific findings. The histopathologic findings of the lesional skin revealed orthokeratotic hyperkeratosis and mild acanthosis; however, there were no significant differences compared with the unaffected skin. He was treated with topical 20% aluminum hydrochloride, and his condition improved. Aquagenic wrinkling of the palm (AWP), a rare dermatosis, is also known as aquagenic syringeal acrokeratoderma, aquagenic palmoplantar keratoderma, aquagenic acrokeratoderma, or transient reactive papulotranslucent keratoderma1. Excessive and early palmar wrinkling occurs within 3 min after water exposure in AWP, whereas water immersion wrinkling is a normal physiologic response to prolonged water immersion and occurs at 11.5 min after water exposure2. It is often associated with hyperhidrosis, pruritus, and a burning or tingling sensation. AWP mainly occurs in Caucasian adolescent women. The diagnosis of AWP is primarily based on clinical manifestations, which is the rapid development of transient whitish edematous plaques with excessive wrinkling on the palm after water exposure ("hand-in-the-bucket" sign)3 4. Because of the whitish discoloration and tingling sensation in the acral area, it may be confused with the Raynaud phenomenon; however, it can be easily differentiated by its excessive wrinkling and the irrelevance of cold temperature. Histopathologic examination in a previous study revealed hyperkeratosis and dilated eccrine ostia3 5. Various treatment modalities, including topical 20% aluminum chloride solution, iontophoresis, or botulinum toxin injection, have all shown good response through the reduction of hyperhidrosis4. Although the exact pathogenesis is unclear, AWP is postulated to be caused by a sweat electrolyte disturbance that results in sodium retention within epidermal keratinocytes, resulting in an increase in osmotically induced cell volume1. Several conditions including cystic fibrosis (CF) and drug (tobramycin, rofecoxib, or aspirin) use have also been reported with AWP2. The association between CF and AWP is well established; Gild et al.2 suggested that patients with AWP should be offered a screening test for both CF and the carrier state. The occurrence of AWP in Asians is very rare. All five Asian AWP cases occurred idiopathically in young men with no evidence of association with either CF or drugs (Table 1)1 3 4 5. Further studies should be performed to elucidate the complete pathogenesis of AWP in Asians. To date, only one case of AWP in Koreans, with an unusual presentation on the dorsum of the fingers, has been reported as "aquagenic syringeal acrokeratoderma"3. Here, we report the second Korean case of AWP showing typical presentations, including a literature review of Asian cases.

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          Aberrant aquaporin 5 expression in the sweat gland in aquagenic wrinkling of the palms.

          Aquagenic wrinkling of the palms (AWP) is an uncommon disease characterized by the rapid and transient formation of edematous whitish plaques on the palms on exposure to water. Although this disease is occasionally accompanied by hyperhidrosis, the pathophysiology of AWP remains unknown. Herein we describe a patient with AWP. The location of wrinkling was limited to the areas positive for iodine-starch test after water exposure, which suggests that AWP is etiologically related to hyperhidrosis. Histologic examination revealed hyperplastic and papillated eccrine glandular epithelium with the enlarged diameter of eccrine coils. Immunohistochemically, while aquaporin 5 (AQP5), one of the water channel AQP families, was present exclusively in the dark cells of sweat glands of healthy donors, an aberrant AQP5 staining, extending to the clear cells, was found in the patient with AWP. The hyperplastic glandular epithelium and aberrant AQP5 staining in the patient's sweat glands suggest that AWP stems from dysregulation of sweating.
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            Aquagenic wrinkling of the palms in cystic fibrosis and the cystic fibrosis carrier state: a case–control study.

            Aquagenic wrinkling of the palms (AWP) is hyperwrinkling occurring within 3 min of exposure to water. It is associated with cystic fibrosis (CF) and has been reported in a CF carrier.
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              Aquagenic syringeal acrokeratoderma: unusual prominence on the dorsal aspect of fingers?

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                Author and article information

                Journal
                Ann Dermatol
                Ann Dermatol
                AD
                Annals of Dermatology
                Korean Dermatological Association; The Korean Society for Investigative Dermatology
                1013-9087
                2005-3894
                December 2015
                07 December 2015
                : 27
                : 6
                : 776-777
                Affiliations
                [1 ]Department of Dermatology, Chonbuk National University Medical School, Jeonju, Korea.
                [2 ]Department of Internal Medicine, Chonbuk National University Medical School, Jeonju, Korea.
                [3 ]Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Jeonju, Korea.
                Author notes
                Corresponding author: Jin Park, Department of Dermatology, Chonbuk National University Medical School, 20 Geonji-ro, Deokjin-gu, Jeonju 54907, Korea. Tel: 82-63-250-2745, Fax: 82-63-250-1970, airmd@ 123456jbnu.ac.kr
                Article
                10.5021/ad.2015.27.6.776
                4695439
                26719656
                991b8c0c-981b-4084-bac1-79a3898ecef0
                Copyright © 2015 The Korean Dermatological Association and The Korean Society for Investigative Dermatology

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 13 October 2014
                : 08 February 2015
                : 18 February 2015
                Categories
                Letter to the Editor

                Dermatology
                Dermatology

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