A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis

Langerhans cell histiocytosis (LCH) is a rare disease, afflicting approximately 4.6 and 1-2 per 1 million children and adults, respectively. While LCH can involve numerous organ systems such as the lung or bone, it is uncommon for the disease to be limited to the skin. Radiotherapy has an establishe...

Full description

Saved in:
Bibliographic Details
Main Authors: Mark K. Farrugia, Carl Morrison, Francisco Hernandez-Ilizaliturri, Saif Aljabab
Format: Article
Language:English
Published: Wiley 2021-01-01
Series:Case Reports in Oncological Medicine
Online Access:http://dx.doi.org/10.1155/2021/6680635
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1832556869880643584
author Mark K. Farrugia
Carl Morrison
Francisco Hernandez-Ilizaliturri
Saif Aljabab
author_facet Mark K. Farrugia
Carl Morrison
Francisco Hernandez-Ilizaliturri
Saif Aljabab
author_sort Mark K. Farrugia
collection DOAJ
description Langerhans cell histiocytosis (LCH) is a rare disease, afflicting approximately 4.6 and 1-2 per 1 million children and adults, respectively. While LCH can involve numerous organ systems such as the lung or bone, it is uncommon for the disease to be limited to the skin. Radiotherapy has an established role for osseous lesions. However, the efficacy and dose for nonosseous manifestations of the disease are not well described. In the current case report, we detail a 49-year-old adult male with skin-limited LCH requiring palliative radiotherapy (RT) to numerous sites for pain control. The patient was initially diagnosed and treated with single agent cytarabine for approximately 6 months. Despite treatment, he had little symptomatic response of his cutaneous lesions. We delivered a single dose of 8 Gray (Gy) to 3 separate skin lesions, including the bilateral groin, right popliteal region, and right axillary lesion, which resulted in pain reduction and partial response at four-month follow-up. Subsequently, we decided to treat the left axillary untreated lesion to a higher dose of 24 Gy in 12 fractions. At four-month follow-up, the left axilla RT resulted in complete clinical response and improved pain control compared to the right axilla. Following RT treatments, the patient was found to have a BRAF mutation, and vemurafenib was initiated. Further follow-up with positron emissions tomography demonstrated complete metabolic response in numerous disease areas, including both axillae. Based on this case report’s findings, a higher radiotherapy dose may be more effective for treating cutaneous LCH.
format Article
id doaj-art-20d5aa622c1245e4a254819b513e6ade
institution Kabale University
issn 2090-6706
2090-6714
language English
publishDate 2021-01-01
publisher Wiley
record_format Article
series Case Reports in Oncological Medicine
spelling doaj-art-20d5aa622c1245e4a254819b513e6ade2025-02-03T05:44:13ZengWileyCase Reports in Oncological Medicine2090-67062090-67142021-01-01202110.1155/2021/66806356680635A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell HistiocytosisMark K. Farrugia0Carl Morrison1Francisco Hernandez-Ilizaliturri2Saif Aljabab3Department of Radiation Medicine, Roswell Park Comprehensive Cancer Center, USADepartment of Pathology, Roswell Park Comprehensive Cancer Center, USADepartment of Medicine-Lymphoma/Myeloma, Roswell Park Comprehensive Cancer Center, USADepartment of Radiation Medicine, Roswell Park Comprehensive Cancer Center, USALangerhans cell histiocytosis (LCH) is a rare disease, afflicting approximately 4.6 and 1-2 per 1 million children and adults, respectively. While LCH can involve numerous organ systems such as the lung or bone, it is uncommon for the disease to be limited to the skin. Radiotherapy has an established role for osseous lesions. However, the efficacy and dose for nonosseous manifestations of the disease are not well described. In the current case report, we detail a 49-year-old adult male with skin-limited LCH requiring palliative radiotherapy (RT) to numerous sites for pain control. The patient was initially diagnosed and treated with single agent cytarabine for approximately 6 months. Despite treatment, he had little symptomatic response of his cutaneous lesions. We delivered a single dose of 8 Gray (Gy) to 3 separate skin lesions, including the bilateral groin, right popliteal region, and right axillary lesion, which resulted in pain reduction and partial response at four-month follow-up. Subsequently, we decided to treat the left axillary untreated lesion to a higher dose of 24 Gy in 12 fractions. At four-month follow-up, the left axilla RT resulted in complete clinical response and improved pain control compared to the right axilla. Following RT treatments, the patient was found to have a BRAF mutation, and vemurafenib was initiated. Further follow-up with positron emissions tomography demonstrated complete metabolic response in numerous disease areas, including both axillae. Based on this case report’s findings, a higher radiotherapy dose may be more effective for treating cutaneous LCH.http://dx.doi.org/10.1155/2021/6680635
spellingShingle Mark K. Farrugia
Carl Morrison
Francisco Hernandez-Ilizaliturri
Saif Aljabab
A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
Case Reports in Oncological Medicine
title A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
title_full A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
title_fullStr A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
title_full_unstemmed A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
title_short A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis
title_sort dose response relationship to radiotherapy for cutaneous lesions of langerhans cell histiocytosis
url http://dx.doi.org/10.1155/2021/6680635
work_keys_str_mv AT markkfarrugia adoseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT carlmorrison adoseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT franciscohernandezilizaliturri adoseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT saifaljabab adoseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT markkfarrugia doseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT carlmorrison doseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT franciscohernandezilizaliturri doseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis
AT saifaljabab doseresponserelationshiptoradiotherapyforcutaneouslesionsoflangerhanscellhistiocytosis