A Rare Case of Extensive Oral Involvement in Bullous Systemic Lupus Erythematosus: Diagnostic and Therapeutic Challenges

Tiara Resmi,1 Oki Suwarsa,2 Irna Sufiawati3 1Oral Medicine Residency Programme, Faculty of Dentistry, Universitas Padjadjaran, Bandung, Indonesia; 2Department of Dermatology and Venereology, Faculty of Medicine, Universitas Padjadjaran, Bandung, Indonesia; 3Department of Oral Medicine, Faculty of Dentistry, Universitas Padjadjaran, Bandung, IndonesiaCorrespondence: Irna Sufiawati, Department of Oral Medicine, Faculty of Dentistry, Universitas Padjadjaran, Jalan Sekeloa Selatan I, Bandung, 40132, Indonesia, Email [email protected]: Bullous systemic lupus erythematosus (BSLE) is a rare autoantibody-mediated subepidermal blistering disease that affects fewer than 5% of systemic lupus erythematosus (SLE) patients. While oral involvement is generally mild, extensive oral lesions as the predominant manifestation are exceptionally uncommon and present significant diagnostic and therapeutic challenges. This report describes a unique case of BSLE with dominant and extensive oral involvement, a presentation rarely documented in the literature. A 29-year-old female presented to the Oral Medicine Clinic with severe, diffuse oral ulcerations of one week’s duration (VAS pain score 8/10), which impaired swallowing and were accompanied by fever, without any history of new medication or food exposure. A 29-year-old female presented to the Oral Medicine Clinic with severe diffuse oral ulcerations for one week (VAS pain score 8/10) that impaired swallowing and were accompanied by fever, without any history of new medications or food exposure. Clinical examination identified widespread erythematous bullae and erosions across the oral mucosa, as well as non-scrapable white plaques on the dorsum and lateral tongue. Histopathological analysis revealed subepidermal separation with predominant neutrophilic infiltration, confirming BSLE and distinguishing it from other vesiculobullous disorders in the absence of direct immunofluorescence (DIF) testing. Management consisted of a multimodal approach, including topical therapy (dexamethasone ointment, antiseptic rinses, saline compresses, and protective emollients) and systemic therapy with high-dose intravenous methylprednisolone and oral cyclosporine, supported by antibiotics and analgesics under dermatology supervision. Notably, all oral lesions resolved completely within four weeks without the need for more aggressive systemic treatment. This case highlights the diagnostic value of a multidisciplinary, clinicopathological approach in confirming BSLE with dominant oral involvement, even when advanced immunofluorescence testing is unavailable, and demonstrates the effectiveness of a tailored, minimally invasive therapeutic strategy that achieved complete healing without the need for aggressive systemic therapy.Keywords: bullous systemic lupus erythematosus, oral ulcerations, autoimmune blistering disease, non-communicable diseases, case report

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SHILAP Revista de lepidopterología
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2025-12-01
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Autoimmune Bullous Skin Diseases
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article

A Rare Case of Extensive Oral Involvement in Bullous Systemic Lupus Erythematosus: Diagnostic and Therapeutic Challenges

C. T. Resmi, O Suwarsa, Sufiawati I
SHILAP Revista de lepidopterología
Autoimmune Bullous Skin Diseases
article

A Rare Case of Extensive Oral Involvement in Bullous Systemic Lupus Erythematosus: Diagnostic and Therapeutic Challenges

C. T. Resmi, O Suwarsa, Sufiawati I
article en

Abstract

Tiara Resmi,1 Oki Suwarsa,2 Irna Sufiawati3 1Oral Medicine Residency Programme, Faculty of Dentistry, Universitas Padjadjaran, Bandung, Indonesia; 2Department of Dermatology and Venereology, Faculty of Medicine, Universitas Padjadjaran, Bandung, Indonesia; 3Department of Oral Medicine, Faculty of Dentistry, Universitas Padjadjaran, Bandung, IndonesiaCorrespondence: Irna Sufiawati, Department of Oral Medicine, Faculty of Dentistry, Universitas Padjadjaran, Jalan Sekeloa Selatan I, Bandung, 40132, Indonesia, Email [email protected]: Bullous systemic lupus erythematosus (BSLE) is a rare autoantibody-mediated subepidermal blistering disease that affects fewer than 5% of systemic lupus erythematosus (SLE) patients. While oral involvement is generally mild, extensive oral lesions as the predominant manifestation are exceptionally uncommon and present significant diagnostic and therapeutic challenges. This report describes a unique case of BSLE with dominant and extensive oral involvement, a presentation rarely documented in the literature. A 29-year-old female presented to the Oral Medicine Clinic with severe, diffuse oral ulcerations of one week’s duration (VAS pain score 8/10), which impaired swallowing and were accompanied by fever, without any history of new medication or food exposure. A 29-year-old female presented to the Oral Medicine Clinic with severe diffuse oral ulcerations for one week (VAS pain score 8/10) that impaired swallowing and were accompanied by fever, without any history of new medications or food exposure. Clinical examination identified widespread erythematous bullae and erosions across the oral mucosa, as well as non-scrapable white plaques on the dorsum and lateral tongue. Histopathological analysis revealed subepidermal separation with predominant neutrophilic infiltration, confirming BSLE and distinguishing it from other vesiculobullous disorders in the absence of direct immunofluorescence (DIF) testing. Management consisted of a multimodal approach, including topical therapy (dexamethasone ointment, antiseptic rinses, saline compresses, and protective emollients) and systemic therapy with high-dose intravenous methylprednisolone and oral cyclosporine, supported by antibiotics and analgesics under dermatology supervision. Notably, all oral lesions resolved completely within four weeks without the need for more aggressive systemic treatment. This case highlights the diagnostic value of a multidisciplinary, clinicopathological approach in confirming BSLE with dominant oral involvement, even when advanced immunofluorescence testing is unavailable, and demonstrates the effectiveness of a tailored, minimally invasive therapeutic strategy that achieved complete healing without the need for aggressive systemic therapy.Keywords: bullous systemic lupus erythematosus, oral ulcerations, autoimmune blistering disease, non-communicable diseases, case report

SHILAP Revista de lepidopterología
University of Dental Medicine (MM), Family Medicine Residency of Idaho (US)
Openalex Percentile: Top 28%
Autoimmune Bullous Skin Diseases
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