Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report
Ameloblastoma is a rare tumor arising from odontogenic cells that is benign, yet locally aggressive. Metastasizing ameloblastoma (METAM) is an ultra-rare ameloblastoma variant in which both primary and secondary tumors have histological features of benign ameloblastoma. This is a case report of a pa...
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| Format: | Article |
| Language: | English |
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SAGE Publishing
2025-12-01
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| Series: | Rare Tumors |
| Online Access: | https://doi.org/10.1177/20363613241309961 |
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| author | Meghan M Lynch Pedro Hermida-Viveiros Sean Stencel Hannah Knott Rusul Al-Maryati Farres Obeidin Borislav A Alexiev Susan Abbinanti Senthil Damodaran Mark Agulnik Seth M Pollack |
| author_facet | Meghan M Lynch Pedro Hermida-Viveiros Sean Stencel Hannah Knott Rusul Al-Maryati Farres Obeidin Borislav A Alexiev Susan Abbinanti Senthil Damodaran Mark Agulnik Seth M Pollack |
| author_sort | Meghan M Lynch |
| collection | DOAJ |
| description | Ameloblastoma is a rare tumor arising from odontogenic cells that is benign, yet locally aggressive. Metastasizing ameloblastoma (METAM) is an ultra-rare ameloblastoma variant in which both primary and secondary tumors have histological features of benign ameloblastoma. This is a case report of a patient who presented with a jaw mass and subsequent lung metastases, later diagnosed as METAM. Initial treatments, including carboplatin, etoposide, and taxane-based chemotherapy, were ineffective. Molecular profiling revealed mutations including PIK3CA H1047R and BRAF V600E. The patient was enrolled in a tumor-agnostic trial and began treatment with copanlisib, a PI3K inhibitor, which resulted in a partial response and durable disease regression. After 76 cycles, she continues to tolerate therapy well with minimal adverse events. This case highlights the potential of targeted therapies such as copanlisib for treating METAM, providing a promising therapeutic option for patients with PIK3CA mutations. |
| format | Article |
| id | doaj-art-dc52a83725924b098e777dbacdaef723 |
| institution | OA Journals |
| issn | 2036-3613 |
| language | English |
| publishDate | 2025-12-01 |
| publisher | SAGE Publishing |
| record_format | Article |
| series | Rare Tumors |
| spelling | doaj-art-dc52a83725924b098e777dbacdaef7232025-08-20T02:26:46ZengSAGE PublishingRare Tumors2036-36132025-12-011710.1177/20363613241309961Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case reportMeghan M LynchPedro Hermida-ViveirosSean StencelHannah KnottRusul Al-MaryatiFarres ObeidinBorislav A AlexievSusan AbbinantiSenthil DamodaranMark AgulnikSeth M PollackAmeloblastoma is a rare tumor arising from odontogenic cells that is benign, yet locally aggressive. Metastasizing ameloblastoma (METAM) is an ultra-rare ameloblastoma variant in which both primary and secondary tumors have histological features of benign ameloblastoma. This is a case report of a patient who presented with a jaw mass and subsequent lung metastases, later diagnosed as METAM. Initial treatments, including carboplatin, etoposide, and taxane-based chemotherapy, were ineffective. Molecular profiling revealed mutations including PIK3CA H1047R and BRAF V600E. The patient was enrolled in a tumor-agnostic trial and began treatment with copanlisib, a PI3K inhibitor, which resulted in a partial response and durable disease regression. After 76 cycles, she continues to tolerate therapy well with minimal adverse events. This case highlights the potential of targeted therapies such as copanlisib for treating METAM, providing a promising therapeutic option for patients with PIK3CA mutations.https://doi.org/10.1177/20363613241309961 |
| spellingShingle | Meghan M Lynch Pedro Hermida-Viveiros Sean Stencel Hannah Knott Rusul Al-Maryati Farres Obeidin Borislav A Alexiev Susan Abbinanti Senthil Damodaran Mark Agulnik Seth M Pollack Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report Rare Tumors |
| title | Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report |
| title_full | Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report |
| title_fullStr | Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report |
| title_full_unstemmed | Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report |
| title_short | Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report |
| title_sort | durable disease regression with copanlisib treatment in pi3k mutated metastasizing ameloblastoma a case report |
| url | https://doi.org/10.1177/20363613241309961 |
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