Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature
Abstract Purpose The purpose of this study was to describe the case of a pediatric patient diagnosed with mucous membrane pemphigoid (MMP) with exclusive ocular involvement presenting at diagnosis with peripheral ulcerative keratitis and provide a review of the literature. Methods A 12-year-old girl...
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| Format: | Article |
| Language: | English |
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SpringerOpen
2025-05-01
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| Series: | Journal of Ophthalmic Inflammation and Infection |
| Online Access: | https://doi.org/10.1186/s12348-025-00480-y |
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| author | Eugenia M. Ramos-Dávila Raul E. Ruiz-Lozano Alejandro Rodríguez-García Carlos Álvarez-Guzmán |
| author_facet | Eugenia M. Ramos-Dávila Raul E. Ruiz-Lozano Alejandro Rodríguez-García Carlos Álvarez-Guzmán |
| author_sort | Eugenia M. Ramos-Dávila |
| collection | DOAJ |
| description | Abstract Purpose The purpose of this study was to describe the case of a pediatric patient diagnosed with mucous membrane pemphigoid (MMP) with exclusive ocular involvement presenting at diagnosis with peripheral ulcerative keratitis and provide a review of the literature. Methods A 12-year-old girl presented with cicatricial conjunctivitis and peripheral ulcerative keratitis (PUK). A conjunctival biopsy and direct immunofluorescence revealed linear deposits of IgG, IgM, and C3 at the basement membrane zone, confirming a diagnosis of ocular MMP. Results The patient was initially treated with dapsone 25 mg/day and prednisone 40 mg/day along with topical treatment including cyclosporine 0.05%, loteprednol etabonate 0.05%, and sodium hyaluronate 0.15% and trehalose 3%. Inflammation persisted as an increase in the extension of symblepharon was documented. Subsequently, dapsone was switched to oral methotrexate 15 mg/week and prednisone was successfully tapered to 5 mg/day. After three years of follow-up, disease activity remained quiescent. Conclusions Pediatric mucous membrane pemphigoid with ocular involvement is a rare condition of which few reports have been published, resulting in scarce information regarding its clinical course and response to treatment. We report the first case observed in a Hispanic patient, opening with peripheral ulcerative keratitis, and responding successfully to methotrexate. |
| format | Article |
| id | doaj-art-dd1b815c8e894d8c9ca196c9006d1d39 |
| institution | DOAJ |
| issn | 1869-5760 |
| language | English |
| publishDate | 2025-05-01 |
| publisher | SpringerOpen |
| record_format | Article |
| series | Journal of Ophthalmic Inflammation and Infection |
| spelling | doaj-art-dd1b815c8e894d8c9ca196c9006d1d392025-08-20T03:22:11ZengSpringerOpenJournal of Ophthalmic Inflammation and Infection1869-57602025-05-011511710.1186/s12348-025-00480-yChildhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literatureEugenia M. Ramos-Dávila0Raul E. Ruiz-Lozano1Alejandro Rodríguez-García2Carlos Álvarez-Guzmán3Tecnologico de Monterrey, Escuela de Medicina y Ciencias de la SaludBascom Palmer Eye Institute, University of MiamiTecnologico de Monterrey, Escuela de Medicina y Ciencias de la SaludTecnologico de Monterrey, Escuela de Medicina y Ciencias de la SaludAbstract Purpose The purpose of this study was to describe the case of a pediatric patient diagnosed with mucous membrane pemphigoid (MMP) with exclusive ocular involvement presenting at diagnosis with peripheral ulcerative keratitis and provide a review of the literature. Methods A 12-year-old girl presented with cicatricial conjunctivitis and peripheral ulcerative keratitis (PUK). A conjunctival biopsy and direct immunofluorescence revealed linear deposits of IgG, IgM, and C3 at the basement membrane zone, confirming a diagnosis of ocular MMP. Results The patient was initially treated with dapsone 25 mg/day and prednisone 40 mg/day along with topical treatment including cyclosporine 0.05%, loteprednol etabonate 0.05%, and sodium hyaluronate 0.15% and trehalose 3%. Inflammation persisted as an increase in the extension of symblepharon was documented. Subsequently, dapsone was switched to oral methotrexate 15 mg/week and prednisone was successfully tapered to 5 mg/day. After three years of follow-up, disease activity remained quiescent. Conclusions Pediatric mucous membrane pemphigoid with ocular involvement is a rare condition of which few reports have been published, resulting in scarce information regarding its clinical course and response to treatment. We report the first case observed in a Hispanic patient, opening with peripheral ulcerative keratitis, and responding successfully to methotrexate.https://doi.org/10.1186/s12348-025-00480-y |
| spellingShingle | Eugenia M. Ramos-Dávila Raul E. Ruiz-Lozano Alejandro Rodríguez-García Carlos Álvarez-Guzmán Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature Journal of Ophthalmic Inflammation and Infection |
| title | Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature |
| title_full | Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature |
| title_fullStr | Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature |
| title_full_unstemmed | Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature |
| title_short | Childhood-onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis: a case report and review of the literature |
| title_sort | childhood onset ocular mucous membrane pemphigoid presenting with peripheral ulcerative keratitis a case report and review of the literature |
| url | https://doi.org/10.1186/s12348-025-00480-y |
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