Letters to the Editor
A 12-year-old Japanese female was referred to our hospital because of pain and edematous whitish plaques on her palms after taking a bath (Fig. 1a). She was otherwise healthy with an unremarkable medical history except for mild palmar thickening. Her father and sister had reported a similar condition. AWP has been reported as the presenting sign in cystic fibrosis, but this patient had neither a family history of cystic fibrosis nor any abnormality of the CFTR gene.4 The histopathological examination of her palm before and after water exposure showed exfoliation of the surface of the stratum corneum (Fig. 1b,c). After exposure to water, the edematous change with increased thickness and dilated eccrine ducts were seen in the stratum corneum. Immunohistochemical findings showed that aquaporin-5 (AQP5) was expressed in the plasma membrane of the clear cells and the luminal plasma membrane of the dark cells in eccrine sweat glands in both the patient and three age- and sex-matched healthy control subjects (Fig. 1d,e). Aquaporins are membrane proteins generally responsible for rapid osmotic water movement across the plasma membrane. AQP5 probably plays some roles in sweat secretion. However, the localization and roles of AQP5 remain unelucidated in humans.3 Examination on the palm before water exposure revealed decreased skin surface hydration (Corneometer CM 825; Courage+Khazaka Electronic, Cologne, Germany) and increased transepidermal water loss (TEWL) (Tewameter TM300; Courage+Khazaka Electronic) (Fig. 1f,g). Relative protein amounts evaluated by tape-stripping of the palm showed a tendency of increasing filaggrin and a decrease in desmoglein-1 as compared with controls. These data were measured according to a previous reported method.5 Although limited to a single case, our study added physiological evaluation by equipment measurement and tape-stripping to histopathological examination, and we confirmed the impaired functions of the stratum corneum and the skin barrier in AWP. To our knowledge, no study reported physiological evaluation of AWP. Sweat of the palms may have an influence on the numerical analysis of skin surface hydration and TEWL by equipment measurement. Moreover, increasing filaggrin and
decreasing desmoglein-1 in the stratum corneum obtained by tape-stripping results in increased moisturizing factor and decreased skin barrier function in AWP. We believe that increasing of water absorption to the palms by decreasing skin barrier function and water retention by increasing moisturizing factor cause the symptoms of AWP, namely, edematous whitish plaques on the palms after water exposure. In this study, although we examined AQP5 in immunohistochemical findings, we cannot show any evidence of aberrant sweat gland functions. For a better understanding, further case studies are required.
CONFLICT OF INTEREST:
None.
Hisako OKUHIRA,1 Hiroshi MATSUNAKA,1 Yoshifumi IWAHASHI,2 Yasushi NAKAMURA,2 Fukumi FURUKAWA,1 Yuki YAMAMOTO1,3 Departments of 1Dermatology, 2Clinical Laboratory Medicine, and 3 Cosmetic Dermatology and Photomedicine, Wakayama Medical University, Wakayama, Japan doi: 10.1111/1346-8138.12953
REFERENCES 1 Vale R, Adam DN. Idiopathic aquagenic wrinkling of the palms in sisters. J Dermatol 2012; 39: 471–472. 2 Itin PH, Lautenschlager S. Aquagenic syringeal acrokeratoderma (transient reactive papulotranslucent acrokeratoderma). Dermatology 2002; 204: 8–11. 3 Kabasima K, Takatoshi S, Miwa K et al. Aberrant aquaporin 5 expression in the sweat gland inaquagenic wrinkling of the palms. J Am Acad Dermatol 2008; 59: 28–32. ve E. Aquagenic pal4 Nadal M, Laudier B, Malinge MC, Binois R, Este mar keratoderma in a patient heterozygous for the mutation c.3197G>C in the CFTR gene. Ann Dermatol Venereol 2015; 142: 201–205. 5 Morita E, Takahashi H, Niihara H et al. Stratum Corneum TARC Level is a New Indicator of Lesional Skin Inflammation in Atopic Dermatitis. Allergy 2010; 65: 1166–1172.
Vesicles and pustules on the fingers as an initial manifestation of eosinophilic pustular folliculitis Dear Editor, Eosinophilic pustular folliculitis (EPF) usually affects the face and trunk, and occasionally involves the palms or soles.1 We present a case of EPF with initial manifestation of vesicles and pustules limited to the fingers.
A 38-year-old woman presented with a 2-year history of pruritic vesicles and pustules on the fingers, recalcitrant to topical potent corticosteroid. The patient had seasonal allergic rhinitis. Physical examination revealed multiple vesicles and a few pustules on the fingers, particularly on the finger pulp and nail
Correspondence: Chika Ohata, M.D., Ph.D., Department of Dermatology, Kurume University School of Medicine, 67 Asahimachi, Kurume, Fukuoka 830-0011, Japan. Email: [email protected]
914
© 2015 Japanese Dermatological Association
Letters to the Editor
(a)
(b)
(c)
(d)
(e)
(f)
Figure 1. (a) Multiple vesicles and desquamation on the fingers. (b) Pustules on the right thumb (arrowhead) with nail deformity. (c) Intraepidermal multilocular pustule (hematoxylin– eosin [HE], original magnification 940). (d) Collections of neutrophils with occasional eosinophils in the pustule (HE, 9400). (e) Indurated erythema with pustules on the left cheek and nose. (f) Extensive eosinophilic infiltrate in the sebaceous lobule (HE, 9200).
folds with nail deformity (Fig. 1a,b). Vesicles and pustules were approximately 1–2 mm in diameter. Although pustules were uniform in size and shape, some vesicles were confluent, large and irregularly shaped. Palms and soles were not involved. Laboratory examination revealed slight leukocytosis (9900/lL) and eosinophilia (6.4%; range, 2.0–4.0%). Anti-HIV antigen and antibody were negative. Because the palms were not affected, acrodermatitis continua of Hallopeau was suspected. A biopsy from the pustule on the right thumb (Fig. 1b) revealed intraepidermal multilocular pustule with occasional eosinophils and perivascular lymphocytic and eosinophilic infiltrate (Fig. 1c,d). A tentative diagnosis of acrodermatitis continua of Hallopeau was made. Treatment with topical difluprednate and maxacalcitol with anti-histamine was not effective on the lesion. Six months later, the patient developed indurated erythema with pustules on the cheek, nose and forehead (Fig. 1e). A biopsy from the left cheek revealed extensive eosinophilic infiltrate in the follicles and sebaceous lobules (Fig. 1f). A diagnosis of
© 2015 Japanese Dermatological Association
EPF was made. We retrospectively scrutinized the biopsy specimen from the right thumb, and concluded that it represented EPF. The lesions on the fingers and face dramatically improved with oral indomethacin 50 mg daily. Eosinophilic pustular folliculitis has three variants: (i) classic EPF; (ii) immunosuppression-associated EPF (mostly HIVrelated); and (iii) infancy-associated EPF.1 The present case was classic EPF, first reported by Ise and Ofuji in 1965.2 In a review of 207 Japanese cases of EPF, the palmoplantar region was affected almost 2 years earlier than other regions in 16 cases (8%), in which accurate EPF diagnosis was not made when the lesion was limited to the palmoplantar region.3 Most of those cases were diagnosed as palmoplantar pustulosis (PPP) or tinea pedis.3 In the present case, the fingers were affected, sparing the palms and soles, leading us to consider acrodermatitis continua of Hallopeau rather than PPP. Multiple vesicles led us to suspect concurrent contact dermatitis, which could be superimposed on pustular lesions to cause eosinophilic infiltrates and pruritus. Upon retrospective observation of the biopsy specimen from the fingers, we realized that multilocular pustules suggested EPF rather than PPP or acrodermatitis continua of Hallopeau, characterized by a unilocular pustule.3 Considering a previous report of the patient with EPF, who developed grouped vesicles without pustules on the fingers,4 vesicles may evolve into pustules during the course. Confluent vesicles in the present case may evolve into large and irregular pustules, which were reported as discriminating manifestations of EPF from PPP.5 EPF should be considered in a case of recalcitrant vesicles and/or pustules on the finger.
CONFLICT OF INTEREST:
None.
Chika OHATA, Saori NII, Minao FURUMURA, Takekuni NAKAMA Department of Dermatology, Kurume University School of Medicine, Kurume, Japan doi: 10.1111/1346-8138.12946
REFERENCES 1 Fujiyama T, Tokura Y. Clinical and histopathological differential diagnosis of eosinophilic pustular folliculitis. J Dermatol 2013; 40: 419–423. 2 Ise S, Ofuji S. Subcorneal pustular dermatosis. A follicular variant? Arch Dermatol 1965; 92: 169–171. 3 Aoyama H, Tagami H. Eosinophilic pustular folliculitis starting initially only with palmoplantar pustular lesions. Report of a case and review of the literature. Dermatology 1992; 185: 276–280. 4 Steffen C. Eosinophilic pustular folliculitis (Ofuji’s disease) with response to dapsone therapy. Arch Dermatol 1985; 121: 921–923. 5 Saruta T, Nakamizo Y. Eosinophilic pustular folliculitis–particularly with reference to lesions on the palm of the hand and sole of the foot. J Dermatol 1980; 7: 239–246.
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A 12-year-old Japanese female was referred to our hospital because of pain and edematous whitish plaques on her palms after taking a bath (Fig. 1a). She was otherwise healthy with an unremarkable medical history except for mild palmar thickening. Her father and sister had reported a similar condition. AWP has been reported as the presenting sign in cystic fibrosis, but this patient had neither a family history of cystic fibrosis nor any abnormality of the CFTR gene.4 The histopathological examination of her palm before and after water exposure showed exfoliation of the surface of the stratum corneum (Fig. 1b,c). After exposure to water, the edematous change with increased thickness and dilated eccrine ducts were seen in the stratum corneum. Immunohistochemical findings showed that aquaporin-5 (AQP5) was expressed in the plasma membrane of the clear cells and the luminal plasma membrane of the dark cells in eccrine sweat glands in both the patient and three age- and sex-matched healthy control subjects (Fig. 1d,e). Aquaporins are membrane proteins generally responsible for rapid osmotic water movement across the plasma membrane. AQP5 probably plays some roles in sweat secretion. However, the localization and roles of AQP5 remain unelucidated in humans.3 Examination on the palm before water exposure revealed decreased skin surface hydration (Corneometer CM 825; Courage+Khazaka Electronic, Cologne, Germany) and increased transepidermal water loss (TEWL) (Tewameter TM300; Courage+Khazaka Electronic) (Fig. 1f,g). Relative protein amounts evaluated by tape-stripping of the palm showed a tendency of increasing filaggrin and a decrease in desmoglein-1 as compared with controls. These data were measured according to a previous reported method.5 Although limited to a single case, our study added physiological evaluation by equipment measurement and tape-stripping to histopathological examination, and we confirmed the impaired functions of the stratum corneum and the skin barrier in AWP. To our knowledge, no study reported physiological evaluation of AWP. Sweat of the palms may have an influence on the numerical analysis of skin surface hydration and TEWL by equipment measurement. Moreover, increasing filaggrin and
decreasing desmoglein-1 in the stratum corneum obtained by tape-stripping results in increased moisturizing factor and decreased skin barrier function in AWP. We believe that increasing of water absorption to the palms by decreasing skin barrier function and water retention by increasing moisturizing factor cause the symptoms of AWP, namely, edematous whitish plaques on the palms after water exposure. In this study, although we examined AQP5 in immunohistochemical findings, we cannot show any evidence of aberrant sweat gland functions. For a better understanding, further case studies are required.
CONFLICT OF INTEREST:
None.
Hisako OKUHIRA,1 Hiroshi MATSUNAKA,1 Yoshifumi IWAHASHI,2 Yasushi NAKAMURA,2 Fukumi FURUKAWA,1 Yuki YAMAMOTO1,3 Departments of 1Dermatology, 2Clinical Laboratory Medicine, and 3 Cosmetic Dermatology and Photomedicine, Wakayama Medical University, Wakayama, Japan doi: 10.1111/1346-8138.12953
REFERENCES 1 Vale R, Adam DN. Idiopathic aquagenic wrinkling of the palms in sisters. J Dermatol 2012; 39: 471–472. 2 Itin PH, Lautenschlager S. Aquagenic syringeal acrokeratoderma (transient reactive papulotranslucent acrokeratoderma). Dermatology 2002; 204: 8–11. 3 Kabasima K, Takatoshi S, Miwa K et al. Aberrant aquaporin 5 expression in the sweat gland inaquagenic wrinkling of the palms. J Am Acad Dermatol 2008; 59: 28–32. ve E. Aquagenic pal4 Nadal M, Laudier B, Malinge MC, Binois R, Este mar keratoderma in a patient heterozygous for the mutation c.3197G>C in the CFTR gene. Ann Dermatol Venereol 2015; 142: 201–205. 5 Morita E, Takahashi H, Niihara H et al. Stratum Corneum TARC Level is a New Indicator of Lesional Skin Inflammation in Atopic Dermatitis. Allergy 2010; 65: 1166–1172.
Vesicles and pustules on the fingers as an initial manifestation of eosinophilic pustular folliculitis Dear Editor, Eosinophilic pustular folliculitis (EPF) usually affects the face and trunk, and occasionally involves the palms or soles.1 We present a case of EPF with initial manifestation of vesicles and pustules limited to the fingers.
A 38-year-old woman presented with a 2-year history of pruritic vesicles and pustules on the fingers, recalcitrant to topical potent corticosteroid. The patient had seasonal allergic rhinitis. Physical examination revealed multiple vesicles and a few pustules on the fingers, particularly on the finger pulp and nail
Correspondence: Chika Ohata, M.D., Ph.D., Department of Dermatology, Kurume University School of Medicine, 67 Asahimachi, Kurume, Fukuoka 830-0011, Japan. Email: [email protected]
914
© 2015 Japanese Dermatological Association
Letters to the Editor
(a)
(b)
(c)
(d)
(e)
(f)
Figure 1. (a) Multiple vesicles and desquamation on the fingers. (b) Pustules on the right thumb (arrowhead) with nail deformity. (c) Intraepidermal multilocular pustule (hematoxylin– eosin [HE], original magnification 940). (d) Collections of neutrophils with occasional eosinophils in the pustule (HE, 9400). (e) Indurated erythema with pustules on the left cheek and nose. (f) Extensive eosinophilic infiltrate in the sebaceous lobule (HE, 9200).
folds with nail deformity (Fig. 1a,b). Vesicles and pustules were approximately 1–2 mm in diameter. Although pustules were uniform in size and shape, some vesicles were confluent, large and irregularly shaped. Palms and soles were not involved. Laboratory examination revealed slight leukocytosis (9900/lL) and eosinophilia (6.4%; range, 2.0–4.0%). Anti-HIV antigen and antibody were negative. Because the palms were not affected, acrodermatitis continua of Hallopeau was suspected. A biopsy from the pustule on the right thumb (Fig. 1b) revealed intraepidermal multilocular pustule with occasional eosinophils and perivascular lymphocytic and eosinophilic infiltrate (Fig. 1c,d). A tentative diagnosis of acrodermatitis continua of Hallopeau was made. Treatment with topical difluprednate and maxacalcitol with anti-histamine was not effective on the lesion. Six months later, the patient developed indurated erythema with pustules on the cheek, nose and forehead (Fig. 1e). A biopsy from the left cheek revealed extensive eosinophilic infiltrate in the follicles and sebaceous lobules (Fig. 1f). A diagnosis of
© 2015 Japanese Dermatological Association
EPF was made. We retrospectively scrutinized the biopsy specimen from the right thumb, and concluded that it represented EPF. The lesions on the fingers and face dramatically improved with oral indomethacin 50 mg daily. Eosinophilic pustular folliculitis has three variants: (i) classic EPF; (ii) immunosuppression-associated EPF (mostly HIVrelated); and (iii) infancy-associated EPF.1 The present case was classic EPF, first reported by Ise and Ofuji in 1965.2 In a review of 207 Japanese cases of EPF, the palmoplantar region was affected almost 2 years earlier than other regions in 16 cases (8%), in which accurate EPF diagnosis was not made when the lesion was limited to the palmoplantar region.3 Most of those cases were diagnosed as palmoplantar pustulosis (PPP) or tinea pedis.3 In the present case, the fingers were affected, sparing the palms and soles, leading us to consider acrodermatitis continua of Hallopeau rather than PPP. Multiple vesicles led us to suspect concurrent contact dermatitis, which could be superimposed on pustular lesions to cause eosinophilic infiltrates and pruritus. Upon retrospective observation of the biopsy specimen from the fingers, we realized that multilocular pustules suggested EPF rather than PPP or acrodermatitis continua of Hallopeau, characterized by a unilocular pustule.3 Considering a previous report of the patient with EPF, who developed grouped vesicles without pustules on the fingers,4 vesicles may evolve into pustules during the course. Confluent vesicles in the present case may evolve into large and irregular pustules, which were reported as discriminating manifestations of EPF from PPP.5 EPF should be considered in a case of recalcitrant vesicles and/or pustules on the finger.
CONFLICT OF INTEREST:
None.
Chika OHATA, Saori NII, Minao FURUMURA, Takekuni NAKAMA Department of Dermatology, Kurume University School of Medicine, Kurume, Japan doi: 10.1111/1346-8138.12946
REFERENCES 1 Fujiyama T, Tokura Y. Clinical and histopathological differential diagnosis of eosinophilic pustular folliculitis. J Dermatol 2013; 40: 419–423. 2 Ise S, Ofuji S. Subcorneal pustular dermatosis. A follicular variant? Arch Dermatol 1965; 92: 169–171. 3 Aoyama H, Tagami H. Eosinophilic pustular folliculitis starting initially only with palmoplantar pustular lesions. Report of a case and review of the literature. Dermatology 1992; 185: 276–280. 4 Steffen C. Eosinophilic pustular folliculitis (Ofuji’s disease) with response to dapsone therapy. Arch Dermatol 1985; 121: 921–923. 5 Saruta T, Nakamizo Y. Eosinophilic pustular folliculitis–particularly with reference to lesions on the palm of the hand and sole of the foot. J Dermatol 1980; 7: 239–246.
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