Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 23/01/2017.
Histopathological examination of a papule showed orthokeratotic hyperkeratosis, hypergranulosis and acanthosis of the epidermis and a lymphocytic inflammatory infiltrate in the upper dermis (figure 1E). Histology of a bullous lesion revealed a subepidermal blister with a predominant eosinophilic infiltrate in the dermis (figure 1F). Direct immunofluorescence of peribullous skin showed a linear band of C3 at the dermal-epidermal junction (figure 1G). ELISA identified high titres (457 U/mL) of autoantibodies against bullous pemphigoid antigen 180 (BP180). The remaining laboratory investigation was normal, including 17-estradiol, progesterone and testosterone levels. Serologies for HIV, hepatitis B and C virus were negative. Pelvic ultrasound was normal. The correlation between clinical, histopathological and immunopathological features allowed the diagnosis of LPP. Hormone therapy was discontinued and oral prednisolone (0.5 mg/kg/day) was initiated. No recurrence of lichenoid or bullous lesions was seen after 4 weeks of therapy. The dose of prednisolone was tapered gradually and the patient completed a total of 6 months of therapy without relapse after another 6 months. ELISA repeated after 6 and 12 months of hormone therapy withdrawal showed a marked decrease in circulating antibodies. Hormone therapy was the possible cause of LPP in our patient, given the development of LPP 2 weeks after the initiation of therapy and the excellent response to withdrawal of gestodene and ethinylestradiol. LPP is more common in adults and females, with an incidence peak between the fifth and sixth decade [1]. There have been no reported cases of simultaneous hormone replacement therapy in women. The pathogenesis of drug-induced LPP remains unknown but an epitope spreading phenomenon appears to be involved. Drugs can induce basement membrane destruction with the exposure of hidden antigens leading to the formation of autoantibodies and subepidermal blisters [1, 6]. Zillikens demonstrated a secondary autoimmune response in LPP against a novel and unique epitope of region 4 of the NC16A domain of the BP180 antigen [6]. Our patient had increased levels of serum anti-BP180 antibodies at presentation that correlated with disease activity as they decreased after hormone therapy cessation. As far as we know, this is the first reported case of LPP possibly induced by the association of gestodene and ethinylestradiol. Disclosure. Financial support: none. Conflict of interest: none. 1 Department of Dermatology and Venereology, Hospital de Curry Cabral – Centro Hospitalar de Lisboa Central, Rua da Beneficência, no 8, 1069-166 Lisbon, Portugal 2 Department of Dermatology, Instituto Português de Oncologia Francisco Gentil, Lisbon, Portugal
André LAUREANO1 Margarida RAFAEL2 Gabriela MARQUES PINTO1 Jorge CARDOSO1
1. Zaraa I, Mahfoudh A, Kallel S, et al. Lichen planus pemphigoides: four new cases and a review of the literature. Int J Dermatol 2013; 52: 406-12.
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2. Swalle VJ, Black MM, Bhogal BS. Lichen planus pemphigoides: two case reports. Clin Exp Dermatol 1998; 23: 132-5. 3. Ben Salem C, Chenguel L, Denguezli M, et al. Captoprilinduced lichen planus pemphigoides. Pharmacoepidemiol Drug Saf 2008; 17: 722-4. 4. Conde-Fernandes I, Pinto-Almeida T, Mendes I, et al. Lichen planus pemphigoides in a child. Eur J Dermatol 2012; 22: 570-1. 5. Duong B, Shellie M, Samt Naveed, Theos A. Lichen planus pemphigoides in a 2-year old girl: Response to treatment with methotrexate. Am J Dermatol 2012; 67: e154-6. 6. Zilikens D, Caux F, Mascaro JM, et al. Autoantibodies in lichen planus pemphigoides react with a novel epitope within the C-terminal NC16A domain of BP180. J Invest Dermatol 1999; 113: 117-21. doi:10.1684/ejd.2013.2180
Two cases of lichen planus pigmentosus inversus: possible causative role of tightly fitting underclothes Lichen planus (LP) shows various clinical appearances and LP pigmentosus inversus (LPPI) is a rare variant, as only 18 cases have been reported in the literature [1-3]. LPPI presents as asymptomatic or mildly pruritic, linear or oval, violaceous or brown macules with the long axis following the flexural fold, most commonly on the axillae and groin. Thus, LPPI shares predilection sites with intertrigo but the clinical appearance is different. It is suspected that the moist microenvironment, sweat content or other flexor-associated external stimuli are involved in its etiology [2]. However, its causes remain unclearly elucidated. Here, we describe two cases of LPPI in which the contact with tightly fitting underclothes was possibly causative. Case 1 was a 65-year-old Japanese man, referred to us in 2011 for evaluation of a 1-year-history of asymptomatic pigmented macules in the intertriginous areas. For several years, he had used long johns and a long-sleeved undershirt, which were worn directly next to the skin and perfectly fitted to the body. His inguinal and axillar regions were thus tightly fastened by these underclothes. He had no medical history of contact dermatitis to underclothes, rubber or metals. On examination, multiple, dark-colored erythematous lesions were observed on the bilateral inguinal regions (figure 1A), axillae (figure 1B) and popliteal fossae (figure 1C). The individual lesions had an ellipsoid to annular configuration and some were annularly enlarged with a centrally healing part (figure 1A). A biopsy specimen from an inguinal lesion revealed a band-like lymphocytic infiltrate in the upper dermis (figure 1F) with abundant colloid bodies, pigmentary incontinence and melanophagia in the papillary dermis (figure 1G), indicating LP. Betamethasone butylate propionate was topically applied for 3 months with a limited therapeutic effect. We then advised the patient to discontinue wearing the tight underclothes. The lesions began to regress and disappeared 6 months later. Case 2 was a 64-year-old Japanese man who consulted us in January 2012 with a 2-month history of slightly pruritic, linear, pigmented patches, confined to the bilateral inguinal regions (figure 1D). The eruption appeared in the winter season of 2011 and for the preceding several months he had EJD, vol. 23, n◦ 6, November-December 2013
Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 23/01/2017.
A
B
C
D
E
after discontinuation of the tight underclothes, suggesting their causative role in our cases. Lichenoid interface dermatitis or pigmented contact dermatitis is induced by various chemicals, such as naphthol AS, benzyl salicylate, fragrance and various other cosmetics [5-7]. Since LP seems not to be simply induced by mechanical stimuli, such chemicals contained in the clothes might elicit a lichenoid tissue reaction. Our patch tests could not reveal the causative chemical materials in the present case. We followed these two patients over a short period. It is currently unknown whether the LPPI in our cases was caused by a particular substance recently prevalent in the market. Further accumulation of LPPI cases might clarify this issue. Disclosure. Financial support: none. Conflict of interest: none. 1
F
G
Figure 1. Case 1: Linear or oval, violaceous or brown macules with the long axis following the flexural fold, such as the right inguinal (A), left axillar (B), and right popliteal (C) regions. Case 2: Pigmented patches in the left inguinal region before (D) and after (E) discontinuation of underclothes. Histopathology of case 1: Band-like lymphohistiocytic infiltrate (F) with abundant colloid bodies and pigmentary incontinence as well as melanophagia in the papillary dermis (G).
used tightly fitting long johns, which were worn directly next to the skin. Histologically, the eruption had characteristic features of LP, including a band-like lymphocytic infiltration in the upper dermis, liquefaction degeneration of the basal layer and dermal melanophages. The underclothes were discontinued and alclometasone dipropionate ointment was initiated. All lesions were cleared in 4 months (figure 1E). He avoided using the tight underwear in the next winter, and the eruption did not occur. Patch tests to the materials contained in the underclothes were performed in case 2. All the patch tests to mercapto mix 2%, thiuram mix 1.25%, dithiocabamate mix 2%, p-tertiary-buthylphenol formaldehyde resin 1%, epoxy resin 1%, formaldehyde 1% (Brial Allergen GmbH, Greven, Germany), nickel, cobalt, ferrum, and chrome (Torii pharmacy, Tokyo, Japan) were negative. We diagnosed both patients as having LPPI. Because of its characteristic predilection sites and pigmentation tendency, LPPI is thought to be a distinct entity among LP. Very similar cases have been reported as annular LP [4]. The eruptions occurred strikingly in the intertriginous areas, suggesting that local external stimuli were causative rather than systemic factors. This hypothesis is further supported by the lack of oral and nail LP lesions, the absence of recent drug use and the negative serological tests for HIV and hepatitis B and C viruses. Intriguingly, the skin lesions disappeared EJD, vol. 23, n◦ 6, November-December 2013
Department of Dermatology, Shizuoka General Hospital, 4-27-1 Kita Ando Aoi-ku, Shizuoka, 420-8527, Japan 2 Department of Dermatology, Hamamatsu School of Medicine, Hamamatsu, Japan
Yuta MAJIMA1 Hiroaki YAGI1 Yu IKEDA1 Taisuke ITO2 Yoshiki TOKURA2
1. Bennàssar A, Mas A, Julià M, Iranzo P, Ferrando J. Annular plaques in the skin folds: 4 cases of lichen planus pigmentosus-inversus. Actas Dermosifiliogr 2009; 100: 602-5. 2. Ohshima N, Shirai A, Saito I, Asahina A. Lichen planus pigmentosus-inversus occurring extensively in multiple intertriginous areas. J Dermatol 2012; 39: 412-4. 3. Gaertner E, Elstein W. Lichen planus pigmentosus-inversus: case report and review of an unusual entity. Dermatol Online J 2012; 18: 11. 4. Reich HL, Nguyen JT, James WD. Annular lichen planus: a case series of 20 patients. J Am Acad Dermatol 2004; 50: 595-9. 5. Le Coz CJ, Lepoittevin JP. Clothing dermatitis from Naphthol AS. Contact Dermatitis 2001; 44: 366-75. 6. Alagappan U, Tay YK, Lim SP. Pigmented contact dermatitis secondary to benzyl salicylate. Acta Derm Venereol 2013 Mar 25. doi: 10.2340/00015555-1542. [Epub ahead of print] 7. Parish DH, Wolf R. Avoiding unnecessary fragrance-induced pigmented contact dermatitis: the ‘pulse point’ myth. Dermatology 2008; 216: 85-6. doi:10.1684/ejd.2013.2182
Recurrent verruciform xanthoma on a skin autograft of the penis Verruciform xanthoma (VX) is an uncommon benign lesion that affects the oral mucosa, the anogenital region or other body sites [1]. Its etiopathogenesis remains unclear. Several factors have been suggested in its formation [1, 2]. Reactive VX following surgery is rare, considered secondary to chronic inflammation [2]. On the other hand, complete surgical excision is, as a rule, curative; recurrence of VX after complete excision is unusual [3, 4]. We report a case of penile VX recurring after radical excision of a previous
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Histopathological examination of a papule showed orthokeratotic hyperkeratosis, hypergranulosis and acanthosis of the epidermis and a lymphocytic inflammatory infiltrate in the upper dermis (figure 1E). Histology of a bullous lesion revealed a subepidermal blister with a predominant eosinophilic infiltrate in the dermis (figure 1F). Direct immunofluorescence of peribullous skin showed a linear band of C3 at the dermal-epidermal junction (figure 1G). ELISA identified high titres (457 U/mL) of autoantibodies against bullous pemphigoid antigen 180 (BP180). The remaining laboratory investigation was normal, including 17-estradiol, progesterone and testosterone levels. Serologies for HIV, hepatitis B and C virus were negative. Pelvic ultrasound was normal. The correlation between clinical, histopathological and immunopathological features allowed the diagnosis of LPP. Hormone therapy was discontinued and oral prednisolone (0.5 mg/kg/day) was initiated. No recurrence of lichenoid or bullous lesions was seen after 4 weeks of therapy. The dose of prednisolone was tapered gradually and the patient completed a total of 6 months of therapy without relapse after another 6 months. ELISA repeated after 6 and 12 months of hormone therapy withdrawal showed a marked decrease in circulating antibodies. Hormone therapy was the possible cause of LPP in our patient, given the development of LPP 2 weeks after the initiation of therapy and the excellent response to withdrawal of gestodene and ethinylestradiol. LPP is more common in adults and females, with an incidence peak between the fifth and sixth decade [1]. There have been no reported cases of simultaneous hormone replacement therapy in women. The pathogenesis of drug-induced LPP remains unknown but an epitope spreading phenomenon appears to be involved. Drugs can induce basement membrane destruction with the exposure of hidden antigens leading to the formation of autoantibodies and subepidermal blisters [1, 6]. Zillikens demonstrated a secondary autoimmune response in LPP against a novel and unique epitope of region 4 of the NC16A domain of the BP180 antigen [6]. Our patient had increased levels of serum anti-BP180 antibodies at presentation that correlated with disease activity as they decreased after hormone therapy cessation. As far as we know, this is the first reported case of LPP possibly induced by the association of gestodene and ethinylestradiol. Disclosure. Financial support: none. Conflict of interest: none. 1 Department of Dermatology and Venereology, Hospital de Curry Cabral – Centro Hospitalar de Lisboa Central, Rua da Beneficência, no 8, 1069-166 Lisbon, Portugal 2 Department of Dermatology, Instituto Português de Oncologia Francisco Gentil, Lisbon, Portugal
André LAUREANO1 Margarida RAFAEL2 Gabriela MARQUES PINTO1 Jorge CARDOSO1
1. Zaraa I, Mahfoudh A, Kallel S, et al. Lichen planus pemphigoides: four new cases and a review of the literature. Int J Dermatol 2013; 52: 406-12.
904
2. Swalle VJ, Black MM, Bhogal BS. Lichen planus pemphigoides: two case reports. Clin Exp Dermatol 1998; 23: 132-5. 3. Ben Salem C, Chenguel L, Denguezli M, et al. Captoprilinduced lichen planus pemphigoides. Pharmacoepidemiol Drug Saf 2008; 17: 722-4. 4. Conde-Fernandes I, Pinto-Almeida T, Mendes I, et al. Lichen planus pemphigoides in a child. Eur J Dermatol 2012; 22: 570-1. 5. Duong B, Shellie M, Samt Naveed, Theos A. Lichen planus pemphigoides in a 2-year old girl: Response to treatment with methotrexate. Am J Dermatol 2012; 67: e154-6. 6. Zilikens D, Caux F, Mascaro JM, et al. Autoantibodies in lichen planus pemphigoides react with a novel epitope within the C-terminal NC16A domain of BP180. J Invest Dermatol 1999; 113: 117-21. doi:10.1684/ejd.2013.2180
Two cases of lichen planus pigmentosus inversus: possible causative role of tightly fitting underclothes Lichen planus (LP) shows various clinical appearances and LP pigmentosus inversus (LPPI) is a rare variant, as only 18 cases have been reported in the literature [1-3]. LPPI presents as asymptomatic or mildly pruritic, linear or oval, violaceous or brown macules with the long axis following the flexural fold, most commonly on the axillae and groin. Thus, LPPI shares predilection sites with intertrigo but the clinical appearance is different. It is suspected that the moist microenvironment, sweat content or other flexor-associated external stimuli are involved in its etiology [2]. However, its causes remain unclearly elucidated. Here, we describe two cases of LPPI in which the contact with tightly fitting underclothes was possibly causative. Case 1 was a 65-year-old Japanese man, referred to us in 2011 for evaluation of a 1-year-history of asymptomatic pigmented macules in the intertriginous areas. For several years, he had used long johns and a long-sleeved undershirt, which were worn directly next to the skin and perfectly fitted to the body. His inguinal and axillar regions were thus tightly fastened by these underclothes. He had no medical history of contact dermatitis to underclothes, rubber or metals. On examination, multiple, dark-colored erythematous lesions were observed on the bilateral inguinal regions (figure 1A), axillae (figure 1B) and popliteal fossae (figure 1C). The individual lesions had an ellipsoid to annular configuration and some were annularly enlarged with a centrally healing part (figure 1A). A biopsy specimen from an inguinal lesion revealed a band-like lymphocytic infiltrate in the upper dermis (figure 1F) with abundant colloid bodies, pigmentary incontinence and melanophagia in the papillary dermis (figure 1G), indicating LP. Betamethasone butylate propionate was topically applied for 3 months with a limited therapeutic effect. We then advised the patient to discontinue wearing the tight underclothes. The lesions began to regress and disappeared 6 months later. Case 2 was a 64-year-old Japanese man who consulted us in January 2012 with a 2-month history of slightly pruritic, linear, pigmented patches, confined to the bilateral inguinal regions (figure 1D). The eruption appeared in the winter season of 2011 and for the preceding several months he had EJD, vol. 23, n◦ 6, November-December 2013
Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 23/01/2017.
A
B
C
D
E
after discontinuation of the tight underclothes, suggesting their causative role in our cases. Lichenoid interface dermatitis or pigmented contact dermatitis is induced by various chemicals, such as naphthol AS, benzyl salicylate, fragrance and various other cosmetics [5-7]. Since LP seems not to be simply induced by mechanical stimuli, such chemicals contained in the clothes might elicit a lichenoid tissue reaction. Our patch tests could not reveal the causative chemical materials in the present case. We followed these two patients over a short period. It is currently unknown whether the LPPI in our cases was caused by a particular substance recently prevalent in the market. Further accumulation of LPPI cases might clarify this issue. Disclosure. Financial support: none. Conflict of interest: none. 1
F
G
Figure 1. Case 1: Linear or oval, violaceous or brown macules with the long axis following the flexural fold, such as the right inguinal (A), left axillar (B), and right popliteal (C) regions. Case 2: Pigmented patches in the left inguinal region before (D) and after (E) discontinuation of underclothes. Histopathology of case 1: Band-like lymphohistiocytic infiltrate (F) with abundant colloid bodies and pigmentary incontinence as well as melanophagia in the papillary dermis (G).
used tightly fitting long johns, which were worn directly next to the skin. Histologically, the eruption had characteristic features of LP, including a band-like lymphocytic infiltration in the upper dermis, liquefaction degeneration of the basal layer and dermal melanophages. The underclothes were discontinued and alclometasone dipropionate ointment was initiated. All lesions were cleared in 4 months (figure 1E). He avoided using the tight underwear in the next winter, and the eruption did not occur. Patch tests to the materials contained in the underclothes were performed in case 2. All the patch tests to mercapto mix 2%, thiuram mix 1.25%, dithiocabamate mix 2%, p-tertiary-buthylphenol formaldehyde resin 1%, epoxy resin 1%, formaldehyde 1% (Brial Allergen GmbH, Greven, Germany), nickel, cobalt, ferrum, and chrome (Torii pharmacy, Tokyo, Japan) were negative. We diagnosed both patients as having LPPI. Because of its characteristic predilection sites and pigmentation tendency, LPPI is thought to be a distinct entity among LP. Very similar cases have been reported as annular LP [4]. The eruptions occurred strikingly in the intertriginous areas, suggesting that local external stimuli were causative rather than systemic factors. This hypothesis is further supported by the lack of oral and nail LP lesions, the absence of recent drug use and the negative serological tests for HIV and hepatitis B and C viruses. Intriguingly, the skin lesions disappeared EJD, vol. 23, n◦ 6, November-December 2013
Department of Dermatology, Shizuoka General Hospital, 4-27-1 Kita Ando Aoi-ku, Shizuoka, 420-8527, Japan 2 Department of Dermatology, Hamamatsu School of Medicine, Hamamatsu, Japan
Yuta MAJIMA1 Hiroaki YAGI1 Yu IKEDA1 Taisuke ITO2 Yoshiki TOKURA2
1. Bennàssar A, Mas A, Julià M, Iranzo P, Ferrando J. Annular plaques in the skin folds: 4 cases of lichen planus pigmentosus-inversus. Actas Dermosifiliogr 2009; 100: 602-5. 2. Ohshima N, Shirai A, Saito I, Asahina A. Lichen planus pigmentosus-inversus occurring extensively in multiple intertriginous areas. J Dermatol 2012; 39: 412-4. 3. Gaertner E, Elstein W. Lichen planus pigmentosus-inversus: case report and review of an unusual entity. Dermatol Online J 2012; 18: 11. 4. Reich HL, Nguyen JT, James WD. Annular lichen planus: a case series of 20 patients. J Am Acad Dermatol 2004; 50: 595-9. 5. Le Coz CJ, Lepoittevin JP. Clothing dermatitis from Naphthol AS. Contact Dermatitis 2001; 44: 366-75. 6. Alagappan U, Tay YK, Lim SP. Pigmented contact dermatitis secondary to benzyl salicylate. Acta Derm Venereol 2013 Mar 25. doi: 10.2340/00015555-1542. [Epub ahead of print] 7. Parish DH, Wolf R. Avoiding unnecessary fragrance-induced pigmented contact dermatitis: the ‘pulse point’ myth. Dermatology 2008; 216: 85-6. doi:10.1684/ejd.2013.2182
Recurrent verruciform xanthoma on a skin autograft of the penis Verruciform xanthoma (VX) is an uncommon benign lesion that affects the oral mucosa, the anogenital region or other body sites [1]. Its etiopathogenesis remains unclear. Several factors have been suggested in its formation [1, 2]. Reactive VX following surgery is rare, considered secondary to chronic inflammation [2]. On the other hand, complete surgical excision is, as a rule, curative; recurrence of VX after complete excision is unusual [3, 4]. We report a case of penile VX recurring after radical excision of a previous
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