Oral Lichen Planus Pemphigoides: Three Cases of a Rare Entity

Stoopler, Eric T.; Charmelo-Silva, Scarlet; Bindakhil, Mohammed; Alawi, Faizan; Sollecito, Thomas P.

doi:10.1097/dad.0000000000001585

Cited by 7 publications

(5 citation statements)

References 4 publications

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“…13 Oral LPP frequently affects the gingiva and buccal mucosa, presenting as desquamative gingivitis, Wickham striae, ulcerations, and/or bullae. 14,15 The histopathologic features of LPP include lymphocyte-rich subepidermal bullae with occasional neutrophils and eosinophils. 16 Necrotic keratinocytes may be detected in the roof of the blister.…”

Section: Introductionmentioning

confidence: 99%

Lichen Planus Pemphigoides Associated With PD-1 and PD-L1 Inhibitors: A Case Series and Review of the Literature

Boyle

Ashi

Puiu

et al. 2022

The American Journal of Dermatopathology

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Immune checkpoint inhibitors are increasingly being used in the treatment of various solid organ and hematologic malignancies. Dermatologic toxicities associated with programmed cell death protein-1 (PD-1) and programmed death ligand-1 (PD-L1) therapy have been widely reported in the literature. It is important for clinicians to be aware of these toxicities to ensure prompt recognition and treatment. Herein, we present the clinical, histopathologic, and immunofluorescence findings of 3 patients diagnosed with lichen planus pemphigoides (LPP) after treatment with anti-PD-1 inhibitors. We also reviewed the literature and summarize 7 previously reported cases of LPP associated with anti-PD-1 and anti-PD-L1 inhibitors. LPP was diagnosed at a median time of 24.4 weeks (range: 4-78 weeks) after initiation of immunotherapy. Clinical findings included papules, plaques, erosions, vesicles, and bullae on the trunk and extremities. Oral involvement was present in half the cases. Histopathologic features of immunotherapy-induced LPP included lichenoid or vacuolar interface dermatitis, the presence of eosinophils, and subepidermal bullae. Direct immunofluorescence demonstrated linear deposition of immunoglobulin G (IgG) or C3. Indirect immunofluorescence demonstrated linear IgG along basement membrane zone on monkey esophagus in 2 cases and linear IgG on the epidermal side of salt split skin in 3 cases. Serum anti-BP180 was elevated in all cases in which enzyme-linked immunosorbent assay was performed.

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Section: Introductionmentioning

confidence: 99%

Lichen Planus Pemphigoides Associated With PD-1 and PD-L1 Inhibitors: A Case Series and Review of the Literature

Boyle

Ashi

Puiu

et al. 2022

The American Journal of Dermatopathology

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“…The prevalence is uncertain, but while it is rare in adults, it is even more so in children; we found only 18 cases of childhood LPP reported in the literature, 6,7 and ours is the first case with exclusive oral involvement. On the other hand, seven cases with unique oral involvement 8,9 have been reported in adults.…”

Section: Discussionmentioning

confidence: 99%

Multiple longstanding oral ulcers in an adolescent

et al. 2020

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“…19,37 Regarding LPP, topical corticosteroids were usually effective in lesions of the oral mucosa. 9,24,25 However, the majority of cases resolved successfully with systemic corticosteroids, dapsone or a combination of both. 3,9,15,16,37,38 The doses of corticosteroids varied from 10-15 to 1 mg/ kg/day, suggesting that even low-doses could be beneficial.…”

Section: Treatmentmentioning

confidence: 99%

“…Regarding LPP, topical corticosteroids were usually effective in lesions of the oral mucosa 9,24,25 . However, the majority of cases resolved successfully with systemic corticosteroids, dapsone or a combination of both 3,9,15,16,37,38 .…”

Section: Treatmentmentioning

confidence: 99%

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Challenges and pitfalls between lichen planus pemphigoides and bullous lichen planus

et al. 2022

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Lichen planus pemphigoides (LPP) and bullous lichen planus (BLP) are rare dermatoses, which are characterised by blisters and lichenoid lesions. Their clinical presentation is heterogenous, displaying overlapping features or mimicking other dermatological diseases. Therefore, diagnosis can often be challenging, requiring a thorough dermatological examination along with distinctive histological and immunopathological characteristics. Lichenoid degeneration of the basal epidermis exposes various antigens of the dermal-epidermal junction in LPP, resulting in the breakdown of immune tolerance, hence, the production of autoantibodies against type XVII collagen. Conversely, no pathogenic autoantibodies are detected in BLP. However, some cases of mucosal lichen planus might display immunopathological features suggestive of autoimmune blistering diseases. Therefore, a better understanding of the pathophysiology of these two distinct dermatoses is imperative. The aim of this review was to provide a summary of the current knowledge on the clinical hallmarks, diagnosis and available therapeutic options in LPP and BLP.

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Oral Lichen Planus Pemphigoides: Three Cases of a Rare Entity

Cited by 7 publications

References 4 publications

Lichen Planus Pemphigoides Associated With PD-1 and PD-L1 Inhibitors: A Case Series and Review of the Literature

Lichen Planus Pemphigoides Associated With PD-1 and PD-L1 Inhibitors: A Case Series and Review of the Literature

Multiple longstanding oral ulcers in an adolescent

Challenges and pitfalls between lichen planus pemphigoides and bullous lichen planus

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