Letters to the editor
613
(a)
(b)
(c)
(d)
Figure 2 Immunohistochemistry demonstrates markedly increased and displaced expression to the higher parakeratotic epidermal layers of suprabasal Cytokeratine 17 in a punch biopsy of palmar hyperkeratotic lesion (a) and vulvar hyperkeratotic lesion (b). Negative control of Cytokeratine 17 immunochemistry on palmar skin (c) and vulvar skin (d).
tions, keratine 17 is not expressed in normal stratified human epidermis. Although keratines 6b/17 are mutated in Type 2 autosomal dominant Pachyonychia congenita, our patient lacked onychodystrophy as well as oral leukoplakia. Consequently, this diagnosis could not be retained. Other differential diagnoses of mucosal leukoplakia are the white sponge naevus typically associated with mutations of keratins 4/13, dyskeratosis congenita with the dyskerin1, Nop10 or Nhp2 mutations and finally acquired leukoplakia secondary to any inflammatory condition like atrophic lichen sclerosus, lichen planus or chronic lichen simplex. This case could potentially represent a new subtype of palmoplantar keratoderma with accompanying leukokeratosis anogenitalis associated with a cytokeratine 17 mutation. M. Nantel-Battista,1,* D. Friedmann,1 V. Kokta,2 D. Bouffard,3 D. Funaro1 de Montre al, Division of Dermatology, Centre Hospitalier de l’Universite CHUM, Montreal, QC, Canada, 2Division of Pathology, Centre Hospitalier Universitaire de Ste-Justine, CHU, Montreal, QC, Canada, 3Division of de Montreal, CHUM, Pathology, Centre Hospitalier de l’Universite Montreal, QC, Canada *Correspondence: M. Nantel-Battista. E-mail: melissa.nantel-battista @umontreal.ca 1
References 1 Lautenschlager S, Pittelkow MR. Plamoplantar keratoderma and leukokeratosis anogenitalis: the second case of a new disease. Dermatology 1998; 197: 300–302.
JEADV 2015, 29, 607–616
2 Itin PH, Rufli T. Collodion baby with evolution to palmoplantar keratoderma and leukokeratosis anogenitalis: a new disease. Eur J Dermatol 1994; 4: 589–592. DOI: 10.1111/jdv.12412
Erythroderma as drug eruption induced by intravesical mitomycin C therapy Editor Intravesical chemotherapy using anticancer agents, such as mitomycin C (MMC), represents an important therapeutic modality after transurethral resection of superficial bladder carcinoma.1 The development of hypersensitivity to chemotherapeutic agents is often observed, however, drug eruption due to intravesical instillation of these agents is relatively uncommon. A 78-year-old man consulted in our hospital for severe itchy generalized erythema in February 2004. He had a past history of an operation for superficial bladder cancer in April 2002. A month after the operation, he had started monthly therapy with intravesical instillation of MMC (10 mg), which continued until the day he consulted in 2004. Physical examination revealed an obvious generalized erythema with scaling (Fig. 1a). There was no sign of cystitis or eruption in the genital area. Skin biopsy
© 2014 European Academy of Dermatology and Venereology
Letters to the Editor
614
(a)
(c)
60% to 70%.2 Drug patch tests are positive in only 30–50% of patients with suspected drug eruption.3 Therefore, from the present work, we conclude that a careful evaluation of clinical course, and in some cases also a rechallenge test, is very important for diagnosis of drug eruption. K. Igawa,* M. Konishi, Y. Moriyama, K. Fukuyama, H. Yokozeki
(b)
Department of Dermatology, Tokyo Medical and Dental University, Graduate School of Medicine, Tokyo, Japan *Correspondence: K. Igawa. E-mail: [email protected]
References 1 Malkowicz SB. Intravesical therapy for superficial bladder cancer. Semin Urol Oncol 2000; 18: 280–288. 2 Ebo DG, Leysen J, Mayorga C, Rozieres A, Knol EF, Terreehorst I. The in vitro diagnosis of drug allergy: status and perspectives. Allergy 2011; 66: 1275–1286. 3 Barbaud A. Drug patch testing in systemic cutaneous drug allergy. Toxicology 2005; 209: 209–216. DOI: 10.1111/jdv.12415
Figure 1 (a) Physical examination of the patient upon admission. Generalized erythema with scaling was obvious. (b) Histopathology of the patient’s erythema. (c) Generalized erythema induced by rechallenge test with intravesical instillation of mitomycin C.
specimens taken from the right forearm revealed non-specific superficial perivascular infiltration of inflammatory cells without spongiosis (Fig. 1b). Laboratory findings including aspartate aminotransferase and alanine aminotransferase were within normal limits and no eosinophilia was observed. Thus, one of the most apparent differential diagnoses was drug eruption, and all medications, including MMC therapy, were discontinued immediately. After discontinuation of all drugs and starting topical steroid therapy (betamethasone dipropionate; Shionogi & Co., Ltd., Osaka, Japan, for 3 days), the generalized erythema was remarkably improved with no recurrences. Neither patch tests nor druginduced lymphocyte stimulation tests (DLST) for all drugs, including MMC, were positive. However, from his clinical course, intravesical instillation of MMC was the leading candidate for the factor affecting his generalized erythema. Therefore, we performed a rechallenge test with intravesical instillation of MMC (5 mg, or half the previous dosage). After 24 h, generalized erythema had developed (Fig. 1c), and we considered that the rechallenge test was positive and immediately stopped MMC instillation. Topical steroid therapy resulted in prompt recovery to normal skin. In the present case, although DLST and patch test were negative, the patient’s generalized erythema was assumed to be due to MMC treatment, as determined by rechallenge test. In general, the reliability of DLST in drug eruption is relatively low, and positive rates for the test have been reported to range from
JEADV 2015, 29, 607–616
Newly identified phenotypes in a FIP1L1/PDGFRA-associated paediatric HES patient: thrombocytosis, mHPA, young stroke and blindness Editor Hypereosinophilic syndromes (HES) were defined as persistent and marked blood eosinophilia (>1.5 9 109/L, ≥6 months) associated with eosinophil-induced organ damages, where other causes of hypereosinophilia have been excluded.1 Although virtually any tissue at any age can be variably concerned, complications arise most frequently in the skin, heart, lungs and nervous system. A crude incidence of 0.035 per 100,000 mostly at the age of 50, with only 33 paediatric HES cases was reported in literature.2 A 9-year-old girl was admitted to our department with the complaints of malnutrition, fever, cough, diarrhoea and polymorphous pruritic skin eruptions (Fig. 1). Her mother kept work in a chemical factory during pregnancy at the age of 43 years. The patient was born after 43-week pregnancy. She developed erythematous, pruritic papules and plaques on face 1 month after birth, which gradually increased and almost spread onto the whole body surface in 2 years. Bilateral keratomalacia, corneal erosions, ulcerations and perforations occurred
© 2014 European Academy of Dermatology and Venereology
613
(a)
(b)
(c)
(d)
Figure 2 Immunohistochemistry demonstrates markedly increased and displaced expression to the higher parakeratotic epidermal layers of suprabasal Cytokeratine 17 in a punch biopsy of palmar hyperkeratotic lesion (a) and vulvar hyperkeratotic lesion (b). Negative control of Cytokeratine 17 immunochemistry on palmar skin (c) and vulvar skin (d).
tions, keratine 17 is not expressed in normal stratified human epidermis. Although keratines 6b/17 are mutated in Type 2 autosomal dominant Pachyonychia congenita, our patient lacked onychodystrophy as well as oral leukoplakia. Consequently, this diagnosis could not be retained. Other differential diagnoses of mucosal leukoplakia are the white sponge naevus typically associated with mutations of keratins 4/13, dyskeratosis congenita with the dyskerin1, Nop10 or Nhp2 mutations and finally acquired leukoplakia secondary to any inflammatory condition like atrophic lichen sclerosus, lichen planus or chronic lichen simplex. This case could potentially represent a new subtype of palmoplantar keratoderma with accompanying leukokeratosis anogenitalis associated with a cytokeratine 17 mutation. M. Nantel-Battista,1,* D. Friedmann,1 V. Kokta,2 D. Bouffard,3 D. Funaro1 de Montre al, Division of Dermatology, Centre Hospitalier de l’Universite CHUM, Montreal, QC, Canada, 2Division of Pathology, Centre Hospitalier Universitaire de Ste-Justine, CHU, Montreal, QC, Canada, 3Division of de Montreal, CHUM, Pathology, Centre Hospitalier de l’Universite Montreal, QC, Canada *Correspondence: M. Nantel-Battista. E-mail: melissa.nantel-battista @umontreal.ca 1
References 1 Lautenschlager S, Pittelkow MR. Plamoplantar keratoderma and leukokeratosis anogenitalis: the second case of a new disease. Dermatology 1998; 197: 300–302.
JEADV 2015, 29, 607–616
2 Itin PH, Rufli T. Collodion baby with evolution to palmoplantar keratoderma and leukokeratosis anogenitalis: a new disease. Eur J Dermatol 1994; 4: 589–592. DOI: 10.1111/jdv.12412
Erythroderma as drug eruption induced by intravesical mitomycin C therapy Editor Intravesical chemotherapy using anticancer agents, such as mitomycin C (MMC), represents an important therapeutic modality after transurethral resection of superficial bladder carcinoma.1 The development of hypersensitivity to chemotherapeutic agents is often observed, however, drug eruption due to intravesical instillation of these agents is relatively uncommon. A 78-year-old man consulted in our hospital for severe itchy generalized erythema in February 2004. He had a past history of an operation for superficial bladder cancer in April 2002. A month after the operation, he had started monthly therapy with intravesical instillation of MMC (10 mg), which continued until the day he consulted in 2004. Physical examination revealed an obvious generalized erythema with scaling (Fig. 1a). There was no sign of cystitis or eruption in the genital area. Skin biopsy
© 2014 European Academy of Dermatology and Venereology
Letters to the Editor
614
(a)
(c)
60% to 70%.2 Drug patch tests are positive in only 30–50% of patients with suspected drug eruption.3 Therefore, from the present work, we conclude that a careful evaluation of clinical course, and in some cases also a rechallenge test, is very important for diagnosis of drug eruption. K. Igawa,* M. Konishi, Y. Moriyama, K. Fukuyama, H. Yokozeki
(b)
Department of Dermatology, Tokyo Medical and Dental University, Graduate School of Medicine, Tokyo, Japan *Correspondence: K. Igawa. E-mail: [email protected]
References 1 Malkowicz SB. Intravesical therapy for superficial bladder cancer. Semin Urol Oncol 2000; 18: 280–288. 2 Ebo DG, Leysen J, Mayorga C, Rozieres A, Knol EF, Terreehorst I. The in vitro diagnosis of drug allergy: status and perspectives. Allergy 2011; 66: 1275–1286. 3 Barbaud A. Drug patch testing in systemic cutaneous drug allergy. Toxicology 2005; 209: 209–216. DOI: 10.1111/jdv.12415
Figure 1 (a) Physical examination of the patient upon admission. Generalized erythema with scaling was obvious. (b) Histopathology of the patient’s erythema. (c) Generalized erythema induced by rechallenge test with intravesical instillation of mitomycin C.
specimens taken from the right forearm revealed non-specific superficial perivascular infiltration of inflammatory cells without spongiosis (Fig. 1b). Laboratory findings including aspartate aminotransferase and alanine aminotransferase were within normal limits and no eosinophilia was observed. Thus, one of the most apparent differential diagnoses was drug eruption, and all medications, including MMC therapy, were discontinued immediately. After discontinuation of all drugs and starting topical steroid therapy (betamethasone dipropionate; Shionogi & Co., Ltd., Osaka, Japan, for 3 days), the generalized erythema was remarkably improved with no recurrences. Neither patch tests nor druginduced lymphocyte stimulation tests (DLST) for all drugs, including MMC, were positive. However, from his clinical course, intravesical instillation of MMC was the leading candidate for the factor affecting his generalized erythema. Therefore, we performed a rechallenge test with intravesical instillation of MMC (5 mg, or half the previous dosage). After 24 h, generalized erythema had developed (Fig. 1c), and we considered that the rechallenge test was positive and immediately stopped MMC instillation. Topical steroid therapy resulted in prompt recovery to normal skin. In the present case, although DLST and patch test were negative, the patient’s generalized erythema was assumed to be due to MMC treatment, as determined by rechallenge test. In general, the reliability of DLST in drug eruption is relatively low, and positive rates for the test have been reported to range from
JEADV 2015, 29, 607–616
Newly identified phenotypes in a FIP1L1/PDGFRA-associated paediatric HES patient: thrombocytosis, mHPA, young stroke and blindness Editor Hypereosinophilic syndromes (HES) were defined as persistent and marked blood eosinophilia (>1.5 9 109/L, ≥6 months) associated with eosinophil-induced organ damages, where other causes of hypereosinophilia have been excluded.1 Although virtually any tissue at any age can be variably concerned, complications arise most frequently in the skin, heart, lungs and nervous system. A crude incidence of 0.035 per 100,000 mostly at the age of 50, with only 33 paediatric HES cases was reported in literature.2 A 9-year-old girl was admitted to our department with the complaints of malnutrition, fever, cough, diarrhoea and polymorphous pruritic skin eruptions (Fig. 1). Her mother kept work in a chemical factory during pregnancy at the age of 43 years. The patient was born after 43-week pregnancy. She developed erythematous, pruritic papules and plaques on face 1 month after birth, which gradually increased and almost spread onto the whole body surface in 2 years. Bilateral keratomalacia, corneal erosions, ulcerations and perforations occurred
© 2014 European Academy of Dermatology and Venereology
