Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
Cutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who present...
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Frontiers Media S.A.
2023-02-01
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| Series: | Frontiers in Immunology |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fimmu.2023.1121380/full |
| _version_ | 1811167250605932544 |
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| author | Tong Li Tong Li Hongjie Liu Hongjie Liu Wei Li Wei Li |
| author_facet | Tong Li Tong Li Hongjie Liu Hongjie Liu Wei Li Wei Li |
| author_sort | Tong Li |
| collection | DOAJ |
| description | Cutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who presented with both flaccid and tense vesicles and bullae. Direct immunofluorescence revealed IgA autoantibody deposits in the basement membrane zone (BMZ) and intercellular space of the epidermis, which displayed an atypical autoantibody deposition pattern. The patient showed rapid disease progression and died during follow-up. We performed a literature review of autoimmune bullous diseases (AIBDs) associated with MM or its precursors and found 17 previously reported cases. Together with the present case, cutaneous involvement of the skin folds was frequently reported, and mucous membranes were barely affected. IgA pemphigus, with consistent IgA monoclonality, was observed in half of the cases. Five patients displayed atypical autoantibody deposition patterns in the skin; the prognosis of these patients appeared to be poorer than that of other patients. We aim to increase our understanding of AIBDs associated with MM or its precursors. |
| first_indexed | 2024-04-10T16:06:09Z |
| format | Article |
| id | doaj.art-9171941a84094d05b78ec8a4006e3278 |
| institution | Directory Open Access Journal |
| issn | 1664-3224 |
| language | English |
| last_indexed | 2024-04-10T16:06:09Z |
| publishDate | 2023-02-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Immunology |
| spelling | doaj.art-9171941a84094d05b78ec8a4006e32782023-02-10T04:48:50ZengFrontiers Media S.A.Frontiers in Immunology1664-32242023-02-011410.3389/fimmu.2023.11213801121380Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature reviewTong Li0Tong Li1Hongjie Liu2Hongjie Liu3Wei Li4Wei Li5Department of Dermatology, Rare Diseases Center, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaLaboratory of Dermatology, Clinical Institute of Inflammation and Immunology, Frontiers Science Center for Disease-related Molecular Network, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaDepartment of Dermatology, Rare Diseases Center, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaLaboratory of Dermatology, Clinical Institute of Inflammation and Immunology, Frontiers Science Center for Disease-related Molecular Network, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaDepartment of Dermatology, Rare Diseases Center, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaLaboratory of Dermatology, Clinical Institute of Inflammation and Immunology, Frontiers Science Center for Disease-related Molecular Network, West China Hospital, Sichuan University, Chengdu, Sichuan, ChinaCutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who presented with both flaccid and tense vesicles and bullae. Direct immunofluorescence revealed IgA autoantibody deposits in the basement membrane zone (BMZ) and intercellular space of the epidermis, which displayed an atypical autoantibody deposition pattern. The patient showed rapid disease progression and died during follow-up. We performed a literature review of autoimmune bullous diseases (AIBDs) associated with MM or its precursors and found 17 previously reported cases. Together with the present case, cutaneous involvement of the skin folds was frequently reported, and mucous membranes were barely affected. IgA pemphigus, with consistent IgA monoclonality, was observed in half of the cases. Five patients displayed atypical autoantibody deposition patterns in the skin; the prognosis of these patients appeared to be poorer than that of other patients. We aim to increase our understanding of AIBDs associated with MM or its precursors.https://www.frontiersin.org/articles/10.3389/fimmu.2023.1121380/fullmultiple myelomaautoimmune bullous diseaseIgA autoantibodiesmonoclonal gammopathydirect immunofluorescence |
| spellingShingle | Tong Li Tong Li Hongjie Liu Hongjie Liu Wei Li Wei Li Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review Frontiers in Immunology multiple myeloma autoimmune bullous disease IgA autoantibodies monoclonal gammopathy direct immunofluorescence |
| title | Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review |
| title_full | Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review |
| title_fullStr | Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review |
| title_full_unstemmed | Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review |
| title_short | Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review |
| title_sort | case report vesiculobullous eruption with an atypical iga deposition pattern in a patient with multiple myeloma a case report and literature review |
| topic | multiple myeloma autoimmune bullous disease IgA autoantibodies monoclonal gammopathy direct immunofluorescence |
| url | https://www.frontiersin.org/articles/10.3389/fimmu.2023.1121380/full |
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