Cargando…

Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma

BACKGROUND: Ocular medulloepithelioma (diktyoma) is a rare and potentially malignant paediatric tumour of the non-pigmented ciliary epithelium. Adjuvant chemotherapy can be given in advanced cases, but the indications and regimens remain to be defined. The aim was to identify whether adjuvant chemot...

Descripción completa

Detalles Bibliográficos
Autores principales: Sheriff, Ibrahim H. N., Karaa, Esin K., Chowdhury, Tanzina, Scheimberg, Irene, Duncan, Catriona, Reddy, M. Ashwin, Sagoo, Mandeep S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10049972/
https://www.ncbi.nlm.nih.gov/pubmed/35105935
http://dx.doi.org/10.1038/s41433-022-01936-4
_version_ 1785014578111840256
author Sheriff, Ibrahim H. N.
Karaa, Esin K.
Chowdhury, Tanzina
Scheimberg, Irene
Duncan, Catriona
Reddy, M. Ashwin
Sagoo, Mandeep S.
author_facet Sheriff, Ibrahim H. N.
Karaa, Esin K.
Chowdhury, Tanzina
Scheimberg, Irene
Duncan, Catriona
Reddy, M. Ashwin
Sagoo, Mandeep S.
author_sort Sheriff, Ibrahim H. N.
collection PubMed
description BACKGROUND: Ocular medulloepithelioma (diktyoma) is a rare and potentially malignant paediatric tumour of the non-pigmented ciliary epithelium. Adjuvant chemotherapy can be given in advanced cases, but the indications and regimens remain to be defined. The aim was to identify whether adjuvant chemotherapy offers treatment benefit in advanced ocular medulloepithelioma. METHODS: This was a retrospective case series of subjects referred to a single specialist ocular oncology centre for advanced ocular medulloepithelioma subsequently treated with enucleation, including those needing adjuvant systemic vincristine, etoposide and carboplatin. A case-note review was performed for included subjects meeting referral criteria. The outcomes were histopathology characteristics, recurrence, metastases and survival. RESULTS: Between March 2010 and June 2017, four male patients (mean age 31 months) underwent enucleation for ocular medulloepithelioma. Adjuvant chemotherapy was commenced in 3 patients (75%) due to malignant histopathological features. With a mean follow-up time of 81.5 months (median 71 months, range 49–135 months) none of the patients have had recurrence, metastases or death from the tumour. CONCLUSIONS: This series is unique in reporting the management of advanced malignant ocular medulloepithelioma with adjuvant systemic vincristine, etoposide and carboplatin for advanced tumours with malignant features. This regimen appears to be safe and may be effective in preventing metastatic spread.
format Online
Article
Text
id pubmed-10049972
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Nature Publishing Group UK
record_format MEDLINE/PubMed
spelling pubmed-100499722023-03-30 Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma Sheriff, Ibrahim H. N. Karaa, Esin K. Chowdhury, Tanzina Scheimberg, Irene Duncan, Catriona Reddy, M. Ashwin Sagoo, Mandeep S. Eye (Lond) Article BACKGROUND: Ocular medulloepithelioma (diktyoma) is a rare and potentially malignant paediatric tumour of the non-pigmented ciliary epithelium. Adjuvant chemotherapy can be given in advanced cases, but the indications and regimens remain to be defined. The aim was to identify whether adjuvant chemotherapy offers treatment benefit in advanced ocular medulloepithelioma. METHODS: This was a retrospective case series of subjects referred to a single specialist ocular oncology centre for advanced ocular medulloepithelioma subsequently treated with enucleation, including those needing adjuvant systemic vincristine, etoposide and carboplatin. A case-note review was performed for included subjects meeting referral criteria. The outcomes were histopathology characteristics, recurrence, metastases and survival. RESULTS: Between March 2010 and June 2017, four male patients (mean age 31 months) underwent enucleation for ocular medulloepithelioma. Adjuvant chemotherapy was commenced in 3 patients (75%) due to malignant histopathological features. With a mean follow-up time of 81.5 months (median 71 months, range 49–135 months) none of the patients have had recurrence, metastases or death from the tumour. CONCLUSIONS: This series is unique in reporting the management of advanced malignant ocular medulloepithelioma with adjuvant systemic vincristine, etoposide and carboplatin for advanced tumours with malignant features. This regimen appears to be safe and may be effective in preventing metastatic spread. Nature Publishing Group UK 2022-02-01 2023-04 /pmc/articles/PMC10049972/ /pubmed/35105935 http://dx.doi.org/10.1038/s41433-022-01936-4 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Article
Sheriff, Ibrahim H. N.
Karaa, Esin K.
Chowdhury, Tanzina
Scheimberg, Irene
Duncan, Catriona
Reddy, M. Ashwin
Sagoo, Mandeep S.
Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title_full Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title_fullStr Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title_full_unstemmed Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title_short Systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
title_sort systemic adjuvant chemotherapy for advanced malignant ocular medulloepithelioma
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10049972/
https://www.ncbi.nlm.nih.gov/pubmed/35105935
http://dx.doi.org/10.1038/s41433-022-01936-4
work_keys_str_mv AT sheriffibrahimhn systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT karaaesink systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT chowdhurytanzina systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT scheimbergirene systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT duncancatriona systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT reddymashwin systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma
AT sagoomandeeps systemicadjuvantchemotherapyforadvancedmalignantocularmedulloepithelioma