Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature
Oral and laryngeal cutaneous T-cell lymphoma (CTCL) is rare and usually associated with poor prognosis. Here, we discuss 2 cases of oral CTCL that developed in heavily pretreated patients and provide a review of the literature. The first case is of a 46-year-old African American male with rapidly pr...
| Main Authors: | , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Karger Publishers
2023-07-01
|
| Series: | Case Reports in Dermatology |
| Subjects: | |
| Online Access: | https://beta.karger.com/Article/FullText/530935 |
| _version_ | 1827868380569272320 |
|---|---|
| author | Carolina Alexander-Savino Alexandra Boye-Doe Edith Bowers Anne Beaven |
| author_facet | Carolina Alexander-Savino Alexandra Boye-Doe Edith Bowers Anne Beaven |
| author_sort | Carolina Alexander-Savino |
| collection | DOAJ |
| description | Oral and laryngeal cutaneous T-cell lymphoma (CTCL) is rare and usually associated with poor prognosis. Here, we discuss 2 cases of oral CTCL that developed in heavily pretreated patients and provide a review of the literature. The first case is of a 46-year-old African American male with rapidly progressive disease, presenting with a lesion on his hard palate 6 months after being diagnosed with a CD4+CD8+ CTCL. His cutaneous disease was widespread with tumors on >80% of his body surface area. Unfortunately, the patient died 2 ½ years after his CTCL diagnosis and 7 months after developing the oral CTCL lesion. The second case is of a 38-year-old African American male with stage IIb CD3+CD4+CD30+ mycosis fungoides (MF), who developed a tumor on the hard palate 6 months after diagnosis. He received palliative radiation to the oral lesion and multiple lines of systemic therapy for pulmonary, laryngeal, esophageal, and gastric involvement. Biopsy of the gastric lesions showed a CD30+ T-cell lymphoma with the same clonal peak as in his skin but with large cell transformation. Brentuximab vendoin was started, and the patient is now in complete remission, 30 months later. From the 76 cases of oral CTCL that have been reported in the English language, six were of transformed MF. The most common sites affected were the tongue and palate, and the most common presentation were erythematous or ulcerated tumors, plaques, or nodules associated with dysphagia and pain. Oral CTCL typically occurs years after the initial diagnosis of CTCL and portend a poor prognosis with an average survival of just over 1 year after development of oral lesions. |
| first_indexed | 2024-03-12T15:29:55Z |
| format | Article |
| id | doaj.art-c9dca35c537e48cd976c655a8616bb96 |
| institution | Directory Open Access Journal |
| issn | 1662-6567 |
| language | English |
| last_indexed | 2024-03-12T15:29:55Z |
| publishDate | 2023-07-01 |
| publisher | Karger Publishers |
| record_format | Article |
| series | Case Reports in Dermatology |
| spelling | doaj.art-c9dca35c537e48cd976c655a8616bb962023-08-10T08:11:07ZengKarger PublishersCase Reports in Dermatology1662-65672023-07-011511110.1159/000530935530935Oral Mycosis Fungoides: Report of 2 Cases and Review of the LiteratureCarolina Alexander-Savino0Alexandra Boye-Doe1Edith Bowers2Anne Beaven3Department of Dermatology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USADivision of Hematology, Department of Medicine, University of North Carolina at Chapel Hill, Chapel Hill, NC, USADepartment of Dermatology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USADivision of Hematology, Department of Medicine, University of North Carolina at Chapel Hill, Chapel Hill, NC, USAOral and laryngeal cutaneous T-cell lymphoma (CTCL) is rare and usually associated with poor prognosis. Here, we discuss 2 cases of oral CTCL that developed in heavily pretreated patients and provide a review of the literature. The first case is of a 46-year-old African American male with rapidly progressive disease, presenting with a lesion on his hard palate 6 months after being diagnosed with a CD4+CD8+ CTCL. His cutaneous disease was widespread with tumors on >80% of his body surface area. Unfortunately, the patient died 2 ½ years after his CTCL diagnosis and 7 months after developing the oral CTCL lesion. The second case is of a 38-year-old African American male with stage IIb CD3+CD4+CD30+ mycosis fungoides (MF), who developed a tumor on the hard palate 6 months after diagnosis. He received palliative radiation to the oral lesion and multiple lines of systemic therapy for pulmonary, laryngeal, esophageal, and gastric involvement. Biopsy of the gastric lesions showed a CD30+ T-cell lymphoma with the same clonal peak as in his skin but with large cell transformation. Brentuximab vendoin was started, and the patient is now in complete remission, 30 months later. From the 76 cases of oral CTCL that have been reported in the English language, six were of transformed MF. The most common sites affected were the tongue and palate, and the most common presentation were erythematous or ulcerated tumors, plaques, or nodules associated with dysphagia and pain. Oral CTCL typically occurs years after the initial diagnosis of CTCL and portend a poor prognosis with an average survival of just over 1 year after development of oral lesions.https://beta.karger.com/Article/FullText/530935oral mycosis fungoidesmycosis fungoidescutaneous t-cell lymphomacase report |
| spellingShingle | Carolina Alexander-Savino Alexandra Boye-Doe Edith Bowers Anne Beaven Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature Case Reports in Dermatology oral mycosis fungoides mycosis fungoides cutaneous t-cell lymphoma case report |
| title | Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature |
| title_full | Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature |
| title_fullStr | Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature |
| title_full_unstemmed | Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature |
| title_short | Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature |
| title_sort | oral mycosis fungoides report of 2 cases and review of the literature |
| topic | oral mycosis fungoides mycosis fungoides cutaneous t-cell lymphoma case report |
| url | https://beta.karger.com/Article/FullText/530935 |
| work_keys_str_mv | AT carolinaalexandersavino oralmycosisfungoidesreportof2casesandreviewoftheliterature AT alexandraboyedoe oralmycosisfungoidesreportof2casesandreviewoftheliterature AT edithbowers oralmycosisfungoidesreportof2casesandreviewoftheliterature AT annebeaven oralmycosisfungoidesreportof2casesandreviewoftheliterature |