A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation
A 57-year-old male had been suffering from an itchy map-shaped symmetrical erosive erythema with a crust that was attached to his upper arm and buttock, and occasionally he suffered from spiking fever. Laboratory examinations showed neither anti-desmoglein 1/3 antibodies nor anti-BP 180 antibodies,...
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| Format: | Article |
| Language: | English |
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Karger Publishers
2014-03-01
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| Series: | Case Reports in Dermatology |
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| Online Access: | http://www.karger.com/Article/FullText/360980 |
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| author | Saori Itoi Atsushi Tanemura Chie Tsuji Shun Kitaba Akinori Yokomi Ichiro Katayama Chiharu Tateishi Daisuke Tsuruta |
| author_facet | Saori Itoi Atsushi Tanemura Chie Tsuji Shun Kitaba Akinori Yokomi Ichiro Katayama Chiharu Tateishi Daisuke Tsuruta |
| author_sort | Saori Itoi |
| collection | DOAJ |
| description | A 57-year-old male had been suffering from an itchy map-shaped symmetrical erosive erythema with a crust that was attached to his upper arm and buttock, and occasionally he suffered from spiking fever. Laboratory examinations showed neither anti-desmoglein 1/3 antibodies nor anti-BP 180 antibodies, and he fulfilled the criteria for a diagnosis of systemic lupus erythematosus (SLE). Histologically, there was eosinophilic necrosis of keratinocytes, liquefaction and degradation with severe lymphocyte infiltration into the epidermis and subepidermal blister formation, suggestive of a variant of SLE, bullous lupus erythematosus (BLE). One month after remission of BLE, peculiar annular hypopigmentation appeared on the peripheral borders. An immunohistochemical analysis showed a decrease in Melan A-positive melanocytes and concomitant pigment incontinentia, with dense infiltration of CD8+ T cells and IL-17A+ Th17 cells. An ultrastructural analysis revealed a decrease, but not a complete disappearance, of both melanocytes and melanosomes, and no impairment in melanosomal transfer. In this case report, we would like to introduce the development of annular depigmentation complicated with BLE, and discuss the effects of lupus condition on melanocyte damage based on immunohistological and electromicroscopic findings of those vitiliginous lesions. |
| first_indexed | 2024-12-17T13:54:18Z |
| format | Article |
| id | doaj.art-97d25769cd33468ea803a6ff6788eb3c |
| institution | Directory Open Access Journal |
| issn | 1662-6567 |
| language | English |
| last_indexed | 2024-12-17T13:54:18Z |
| publishDate | 2014-03-01 |
| publisher | Karger Publishers |
| record_format | Article |
| series | Case Reports in Dermatology |
| spelling | doaj.art-97d25769cd33468ea803a6ff6788eb3c2022-12-21T21:45:58ZengKarger PublishersCase Reports in Dermatology1662-65672014-03-0161919710.1159/000360980360980A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular HypopigmentationSaori ItoiAtsushi TanemuraChie TsujiShun KitabaAkinori YokomiIchiro KatayamaChiharu TateishiDaisuke TsurutaA 57-year-old male had been suffering from an itchy map-shaped symmetrical erosive erythema with a crust that was attached to his upper arm and buttock, and occasionally he suffered from spiking fever. Laboratory examinations showed neither anti-desmoglein 1/3 antibodies nor anti-BP 180 antibodies, and he fulfilled the criteria for a diagnosis of systemic lupus erythematosus (SLE). Histologically, there was eosinophilic necrosis of keratinocytes, liquefaction and degradation with severe lymphocyte infiltration into the epidermis and subepidermal blister formation, suggestive of a variant of SLE, bullous lupus erythematosus (BLE). One month after remission of BLE, peculiar annular hypopigmentation appeared on the peripheral borders. An immunohistochemical analysis showed a decrease in Melan A-positive melanocytes and concomitant pigment incontinentia, with dense infiltration of CD8+ T cells and IL-17A+ Th17 cells. An ultrastructural analysis revealed a decrease, but not a complete disappearance, of both melanocytes and melanosomes, and no impairment in melanosomal transfer. In this case report, we would like to introduce the development of annular depigmentation complicated with BLE, and discuss the effects of lupus condition on melanocyte damage based on immunohistological and electromicroscopic findings of those vitiliginous lesions.http://www.karger.com/Article/FullText/360980Bullous lupus erythematosusAnnular hypopigmentationUltrastructural analysis |
| spellingShingle | Saori Itoi Atsushi Tanemura Chie Tsuji Shun Kitaba Akinori Yokomi Ichiro Katayama Chiharu Tateishi Daisuke Tsuruta A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation Case Reports in Dermatology Bullous lupus erythematosus Annular hypopigmentation Ultrastructural analysis |
| title | A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation |
| title_full | A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation |
| title_fullStr | A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation |
| title_full_unstemmed | A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation |
| title_short | A Rare Case of Male Bullous Lupus Erythematosus Complicated with Subsequent Annular Hypopigmentation |
| title_sort | rare case of male bullous lupus erythematosus complicated with subsequent annular hypopigmentation |
| topic | Bullous lupus erythematosus Annular hypopigmentation Ultrastructural analysis |
| url | http://www.karger.com/Article/FullText/360980 |
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