Cholinergic urticaria associated with acquired generalized hypohidrosis: report of a case and review of the literature

Itakura, Eijun; Urabe, Katsuhiko; Yasumoto, Sawa; Nakayama, Juichiro; Furue, Masutaka

doi:10.1046/j.1365-2133.2000.03845.x

Cited by 45 publications

(50 citation statements)

References 16 publications

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“…In addition, AIGA is often associated with cholinergic urticaria and is also known as "cholinergic urticaria associated acquired generalized hypohidrosis" (3). Steroid therapy has been reported to be effective for AIGA (2)(3)(4)(5)(6) and may be associated with other autoimmune diseases (7), thus implying that the autoimmunological background of AIGA is related to its pathogenesis. In most AIGA patients, skin biopsies show no remarkable abnormalities in the eccrine glands or postganglionic sudomotor nerve fibers, except for nonspecific lymphocyte infiltration around the eccrine glands (2-6) and reduced muscarinic M3 receptors (M3Rs) on the eccrine glands (6).…”

Section: Introductionmentioning

confidence: 99%

“…Meanwhile, acquired idiopathic generalized anhidrosis (AIGA) is characterized by anhidrosis or hypohidrosis of the whole body with no identified cause or other autonomic or neurological abnormalities (2). In addition, AIGA is often associated with cholinergic urticaria and is also known as "cholinergic urticaria associated acquired generalized hypohidrosis" (3). Steroid therapy has been reported to be effective for AIGA (2)(3)(4)(5)(6) and may be associated with other autoimmune diseases (7), thus implying that the autoimmunological background of AIGA is related to its pathogenesis.…”

Section: Introductionmentioning

confidence: 99%

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Investigation of Antimuscarinic Receptor Autoantibodies in Patients with Acquired Idiopathic Generalized Anhidrosis

et al. 2013

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Objective The autoimmune mechanism is considered to play an important role in the development of acquired idiopathic generalized anhidrosis (AIGA), and muscarinic M3 receptors (M3Rs) on eccrine glands are possible autoimmune targets. We investigated the existence of autoantibodies against M3Rs in AIGA patients. Methods We immunostained M3R-expressing cultured cells with the serum of 12 AIGA patients (mean age: 35.0±11.7 years, mean disease duration: 26.6±25.8 months) and 10 healthy subjects (mean age: 32.4± 10.4 years). Results The surface of the M3R-expressing cells was stained by the serum obtained from one of the 12 AIGA patients but not by the serum obtained from the remaining 11 patients or healthy subjects. Conclusion The presence of M3R autoantibodies may therefore be related to the underlying mechanism of disease in a subset of AIGA patients.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Investigation of Antimuscarinic Receptor Autoantibodies in Patients with Acquired Idiopathic Generalized Anhidrosis

et al. 2013

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“…In both of our cases, histologically, their sweat glands were not atrophic, which differs from typical cases of acquired generalized anhidrosis accompanied by degeneration of eccrine glands [11, 12, 13]. Itakura et al [3]reported that there were no histological abnormalities in their case of CU associated with acquired generalized hypohidrosis. In contrast, in our case 1, we definitely found spongiotic changes in the sweat duct wall in the epidermis and an inflammatory cell infiltrate in the upper dermis together with dilatation of the sweat duct suggestive of leakage of proinflammatory sweat due to ductal occlusion at the superficial acrosyringium.…”

Section: Discussionmentioning

confidence: 71%

“…The fact that there are scattered reports of cases of coexisting CU and hypohidrosis as reviewed by Itakura et al [3]suggests that the sweat leakage due to occlusion of the superficial acrosyringium in the pathomechanism of CU may not be a very rare event. When sweat materials, which contain numerous enzymes, a renin-like substance, secretory IgA, cytokines including interleukins 1α and β [16], interleukin 8 [17]and IgE [18], once leak out from the sweat ducts, they will definitely induce local inflammatory reactions to cause wheals.…”

Section: Discussionmentioning

confidence: 99%

“…However, the intradermal test with cholinergic drugs in these patients is unreliable to induce wheals and is thought to be not specific enough to be useful [1]. In contrast, there are scattered reports of CU associated with acquired generalized hypohidrosis [3]. We have recently examined 2 patients with CU in both of whom we noticed the presence of extensive hypohidrosis that occurred only in winter.…”

Section: Introductionmentioning

confidence: 99%

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Cholinergic Urticaria, a New Pathogenic Concept: Hypohidrosis due to Interference with the Delivery of Sweat to the Skin Surface

et al. 2002

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Background: Acetylcholine has been suspected to be a pathogenetic factor for cholinergic urticaria (CU), without definite evidence. In contrast, there are scattered reports of CU associated with acquired generalized hypohidrosis. We have recently examined 2 patients with CU in both of whom we noticed the presence of extensive hypohidrosis that occurred only in winter. Objective: In these 2 patients, acquired hypohidrosis due to superficial obstruction of the acrosyringium was suspected as the cause. Both case 1, a 22-year-old Japanese man, and case 2, a 21-year-old Japanese man, began to have anhidrosis and numerous red macules on their body whenever they felt hot in winter. These symptoms ceased to appear in summer. Methods: We studied histologically their lesional skin in addition to provocation tests for CU. Results: The diagnosis of CU was confirmed by the provocation of typical wheals after physical exercise in both cases. Histological study revealed findings suggestive of the presence of occlusion of the superficial acrosyringium. Conclusion: We think that such a hypohidrosis due to occlusion of superficial sweat ducts may also play a role in many other patients with CU of unknown etiology that becomes exacerbated in winter when sweating is not a frequent event.

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An inherited disorder with splenomegaly, cytopenias, and vision loss

Tantravahi¹,

Williams²,

Digre³

et al. 2012

American J of Med Genetics Pt A

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We describe a novel inherited disorder consisting of idiopathic massive splenomegaly, cytopenias, anhidrosis, chronic optic nerve edema, and vision loss. This disorder involves three affected patients in a single non-consanguineous Caucasian family, a mother and two daughters, who are half-sisters. All three patients have had splenectomies; histopathology revealed congestion of the red pulp, but otherwise no abnormalities. Electron microscopic studies of splenic tissue showed no evidence for a storage disorder or other ultrastructural abnormality. Two of the three patients had bone marrow examinations that were non-diagnostic. All three patients developed progressive vision loss such that the two oldest patients are now blind, possibly due to a cone-rod dystrophy. Characteristics of vision loss in this family include early chronic optic nerve edema, and progressive vision loss, particularly central and color vision. Despite numerous medical and ophthalmic evaluations, no diagnosis has been discovered.

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Cholinergic urticaria associated with acquired generalized hypohidrosis: report of a case and review of the literature

Cited by 45 publications

References 16 publications

Investigation of Antimuscarinic Receptor Autoantibodies in Patients with Acquired Idiopathic Generalized Anhidrosis

Investigation of Antimuscarinic Receptor Autoantibodies in Patients with Acquired Idiopathic Generalized Anhidrosis

Cholinergic Urticaria, a New Pathogenic Concept: Hypohidrosis due to Interference with the Delivery of Sweat to the Skin Surface

An inherited disorder with splenomegaly, cytopenias, and vision loss

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