Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature

A 63-year-old woman, with 11-year history of breast cancer, showed bilateral lacrimal gland enlargement on magnetic resonance imaging. Gallium-67 scintigraphy, as the standard at that time in 2004, demonstrated abnormally high uptake only in bilateral lacrimal glands. The lacrimal glands were extirp...

Full description

Bibliographic Details
Main Authors: Toshihiko Matsuo MD, PhD, Takehiro Tanaka MD, PhD, Kazuya Okada MD, PhD, Kenji Notohara MD, PhD, Keiko Fujii MD, PhD, Nobuharu Fujii MD, PhD
Format: Article
Language:English
Published: SAGE Publishing 2023-07-01
Series:Journal of Investigative Medicine High Impact Case Reports
Online Access:https://doi.org/10.1177/23247096231185483
_version_ 1797786734508900352
author Toshihiko Matsuo MD, PhD
Takehiro Tanaka MD, PhD
Kazuya Okada MD, PhD
Kenji Notohara MD, PhD
Keiko Fujii MD, PhD
Nobuharu Fujii MD, PhD
author_facet Toshihiko Matsuo MD, PhD
Takehiro Tanaka MD, PhD
Kazuya Okada MD, PhD
Kenji Notohara MD, PhD
Keiko Fujii MD, PhD
Nobuharu Fujii MD, PhD
author_sort Toshihiko Matsuo MD, PhD
collection DOAJ
description A 63-year-old woman, with 11-year history of breast cancer, showed bilateral lacrimal gland enlargement on magnetic resonance imaging. Gallium-67 scintigraphy, as the standard at that time in 2004, demonstrated abnormally high uptake only in bilateral lacrimal glands. The lacrimal glands were extirpated and the pathological diagnosis was mantle cell lymphoma (MCL). She underwent bilateral orbital radiation, based on no uptake of gallium-67 in other sites of the body. In a month, bone marrow biopsy revealed the infiltration with MCL, positive for cyclin D1. She showed hepatic lymphadenopathy and splenomegaly, and so received 2 cycles of alternating Hyper-CVAD therapy and high-dose methotrexate with cytarabine, combined with rituximab, in 2 months, leading to complete remission. She underwent autologous peripheral blood stem cell transplantation and was well until the age of 68 years when she showed a recurrent intratracheal submucosal lesion of lymphoma and underwent one course of reduced-dose CHOP combined with rituximab. Next year, the left rib resection revealed the metastasis of breast adenocarcinoma, leading to daily oral letrozole. Further 2 years later, computed tomographic scan demonstrated multiple submucosal nodular lesions in the trachea and bronchi, together with cervical and supraclavicular lymphadenopathy, and intratracheal lesion biopsy and bone marrow biopsy proved the involvement with MCL. She underwent 2 courses of bendamustine and rituximab, resulting in complete remission but died of metastatic breast cancer at the age of 74 years. Clinical features in 48 previous cases with ocular adnexal MCL in the literature were summarized in this study.
first_indexed 2024-03-13T01:13:02Z
format Article
id doaj.art-cca5b587bc924223a0fb08e2fa275545
institution Directory Open Access Journal
issn 2324-7096
language English
last_indexed 2024-03-13T01:13:02Z
publishDate 2023-07-01
publisher SAGE Publishing
record_format Article
series Journal of Investigative Medicine High Impact Case Reports
spelling doaj.art-cca5b587bc924223a0fb08e2fa2755452023-07-05T17:03:24ZengSAGE PublishingJournal of Investigative Medicine High Impact Case Reports2324-70962023-07-011110.1177/23247096231185483Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the LiteratureToshihiko Matsuo MD, PhD0Takehiro Tanaka MD, PhD1Kazuya Okada MD, PhD2Kenji Notohara MD, PhD3Keiko Fujii MD, PhD4Nobuharu Fujii MD, PhD5Okayama University Hospital, JapanOkayama University, JapanKurashiki Central Hospital, JapanKurashiki Central Hospital, JapanOkayama University Hospital, JapanOkayama University Hospital, JapanA 63-year-old woman, with 11-year history of breast cancer, showed bilateral lacrimal gland enlargement on magnetic resonance imaging. Gallium-67 scintigraphy, as the standard at that time in 2004, demonstrated abnormally high uptake only in bilateral lacrimal glands. The lacrimal glands were extirpated and the pathological diagnosis was mantle cell lymphoma (MCL). She underwent bilateral orbital radiation, based on no uptake of gallium-67 in other sites of the body. In a month, bone marrow biopsy revealed the infiltration with MCL, positive for cyclin D1. She showed hepatic lymphadenopathy and splenomegaly, and so received 2 cycles of alternating Hyper-CVAD therapy and high-dose methotrexate with cytarabine, combined with rituximab, in 2 months, leading to complete remission. She underwent autologous peripheral blood stem cell transplantation and was well until the age of 68 years when she showed a recurrent intratracheal submucosal lesion of lymphoma and underwent one course of reduced-dose CHOP combined with rituximab. Next year, the left rib resection revealed the metastasis of breast adenocarcinoma, leading to daily oral letrozole. Further 2 years later, computed tomographic scan demonstrated multiple submucosal nodular lesions in the trachea and bronchi, together with cervical and supraclavicular lymphadenopathy, and intratracheal lesion biopsy and bone marrow biopsy proved the involvement with MCL. She underwent 2 courses of bendamustine and rituximab, resulting in complete remission but died of metastatic breast cancer at the age of 74 years. Clinical features in 48 previous cases with ocular adnexal MCL in the literature were summarized in this study.https://doi.org/10.1177/23247096231185483
spellingShingle Toshihiko Matsuo MD, PhD
Takehiro Tanaka MD, PhD
Kazuya Okada MD, PhD
Kenji Notohara MD, PhD
Keiko Fujii MD, PhD
Nobuharu Fujii MD, PhD
Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
Journal of Investigative Medicine High Impact Case Reports
title Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
title_full Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
title_fullStr Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
title_full_unstemmed Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
title_short Bilateral Lacrimal Gland Mantle Cell Lymphoma in 11-Year Follow-Up: Case Report and Review of 48 Cases With Ocular Adnexal Presentation in the Literature
title_sort bilateral lacrimal gland mantle cell lymphoma in 11 year follow up case report and review of 48 cases with ocular adnexal presentation in the literature
url https://doi.org/10.1177/23247096231185483
work_keys_str_mv AT toshihikomatsuomdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature
AT takehirotanakamdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature
AT kazuyaokadamdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature
AT kenjinotoharamdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature
AT keikofujiimdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature
AT nobuharufujiimdphd bilaterallacrimalglandmantlecelllymphomain11yearfollowupcasereportandreviewof48caseswithocularadnexalpresentationintheliterature