Refractory Bullous Systemic Lupus Erythematosus Successfully Treated with Rituximab: A Case Report and Literature Review
Issued Date
2023-01-01
Resource Type
eISSN
11787015
Scopus ID
2-s2.0-85153864661
Journal Title
Clinical, Cosmetic and Investigational Dermatology
Volume
16
Start Page
883
End Page
890
Rights Holder(s)
SCOPUS
Bibliographic Citation
Clinical, Cosmetic and Investigational Dermatology Vol.16 (2023) , 883-890
Suggested Citation
Ratanapokasatit Y., Seree-Aphinan C., Chanprapaph K. Refractory Bullous Systemic Lupus Erythematosus Successfully Treated with Rituximab: A Case Report and Literature Review. Clinical, Cosmetic and Investigational Dermatology Vol.16 (2023) , 883-890. 890. doi:10.2147/CCID.S403866 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/82492
Title
Refractory Bullous Systemic Lupus Erythematosus Successfully Treated with Rituximab: A Case Report and Literature Review
Author(s)
Author's Affiliation
Other Contributor(s)
Abstract
Bullous systemic lupus erythematosus (BSLE) is a rare blistering skin manifestation of systemic lupus erythematosus (SLE). Dapsone is reported to be helpful in mild-to-moderate BSLE cases; however, its use may be limited or prohibited due to particular complications such as drug hypersensitivity, dose-dependent hemolytic anemia, and other significant hematologic abnormalities. Rituximab, an anti-CD20 monoclonal antibody, has been reported with off-label use in BSLE patients, but data are still limited. Hence, our objective is to explore the efficacy of rituximab among these patients. Herein, we report a 21-year-old Thai woman presented with blistering eruption on the oral cavity, scalp, trunk, and extremities for 1 month. The investigations revealed a positive direct Coomb’s test, an elevated erythrocyte sedimentation rate (ESR), and a positive antinuclear antibody (ANA). Skin biopsy showed focal interface dermatitis. Direct immunofluorescence (DIF) illustrated mixed linear and granular deposition of immunoglobulin (Ig)G, IgM, IgA, and C3 along the dermo-epidermal junction (DEJ). Enzyme-linked immunosorbent assay (ELISA) showed circulating antibodies to type VII collagen. She was diagnosed with severe BSLE and autoimmune hemolytic anemia (AIHA) refractory to several oral immunosuppressants but was successfully treated with rituximab. The authors also performed a review of the literature on prior BSLE cases managed with rituximab.