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Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review

Langerhans cell histiocytosis (LCH) is a rare inflammatory myeloid neoplasm arising from the proliferation of pathologic Langerhans cells. LCH has a spectrum of presentations predominantly affecting male pediatric patients. As LCH is a relatively uncommon diagnosis, there is no standard of care for...

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Autores principales: Etter, Emily, Bosse, Benjamin, Wang, Yi Peng, Hiniker, Susan, Oh, Justin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10279942/
https://www.ncbi.nlm.nih.gov/pubmed/37346879
http://dx.doi.org/10.1002/ccr3.7506
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author Etter, Emily
Bosse, Benjamin
Wang, Yi Peng
Hiniker, Susan
Oh, Justin
author_facet Etter, Emily
Bosse, Benjamin
Wang, Yi Peng
Hiniker, Susan
Oh, Justin
author_sort Etter, Emily
collection PubMed
description Langerhans cell histiocytosis (LCH) is a rare inflammatory myeloid neoplasm arising from the proliferation of pathologic Langerhans cells. LCH has a spectrum of presentations predominantly affecting male pediatric patients. As LCH is a relatively uncommon diagnosis, there is no standard of care for treatment of the disease and treatment is based largely on clinical judgment, lesion characteristics, and symptoms at presentation. Here we present a case of unifocal, isolated orbital LCH in a 19‐year‐old young man treated initially with surgical resection. Follow‐up imaging 2 months later demonstrated significant regrowth of the mass and no other sites of disease. The recurrent orbital disease was treated with stereotactic radiosurgery (SRS) to 7 Gy in one fraction. Near complete resolution of the mass was achieved with no recurrence after 1.5 years of follow‐up. SRS for treatment of orbital LCH is a novel treatment not previously described in the literature which may provide benefit in select cases.
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spelling pubmed-102799422023-06-21 Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review Etter, Emily Bosse, Benjamin Wang, Yi Peng Hiniker, Susan Oh, Justin Clin Case Rep Case Report Langerhans cell histiocytosis (LCH) is a rare inflammatory myeloid neoplasm arising from the proliferation of pathologic Langerhans cells. LCH has a spectrum of presentations predominantly affecting male pediatric patients. As LCH is a relatively uncommon diagnosis, there is no standard of care for treatment of the disease and treatment is based largely on clinical judgment, lesion characteristics, and symptoms at presentation. Here we present a case of unifocal, isolated orbital LCH in a 19‐year‐old young man treated initially with surgical resection. Follow‐up imaging 2 months later demonstrated significant regrowth of the mass and no other sites of disease. The recurrent orbital disease was treated with stereotactic radiosurgery (SRS) to 7 Gy in one fraction. Near complete resolution of the mass was achieved with no recurrence after 1.5 years of follow‐up. SRS for treatment of orbital LCH is a novel treatment not previously described in the literature which may provide benefit in select cases. John Wiley and Sons Inc. 2023-06-19 /pmc/articles/PMC10279942/ /pubmed/37346879 http://dx.doi.org/10.1002/ccr3.7506 Text en © 2023 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd. https://creativecommons.org/licenses/by/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Etter, Emily
Bosse, Benjamin
Wang, Yi Peng
Hiniker, Susan
Oh, Justin
Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title_full Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title_fullStr Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title_full_unstemmed Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title_short Successful treatment of orbital Langerhans cell histiocytosis with stereotactic radiosurgery: A case report and literature review
title_sort successful treatment of orbital langerhans cell histiocytosis with stereotactic radiosurgery: a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10279942/
https://www.ncbi.nlm.nih.gov/pubmed/37346879
http://dx.doi.org/10.1002/ccr3.7506
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