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Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review
BACKGROUND: Langerhans cell histiocytosis (LCH) is a neoplastic transformation of myeloid precursors that commonly presents as an osteolytic lesion of the long or flat bones in children. Aneurysmal bone cysts (ABC) are benign neoplasms that frequently affect the metaphysis of long bones and the spin...
Autores principales: | , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Springer Berlin Heidelberg
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10643349/ https://www.ncbi.nlm.nih.gov/pubmed/37522932 http://dx.doi.org/10.1007/s00381-023-06108-7 |
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author | Mo, Jonathan Tomonaga Darrow, Morgan Angus Sharma, Julia Devi |
author_facet | Mo, Jonathan Tomonaga Darrow, Morgan Angus Sharma, Julia Devi |
author_sort | Mo, Jonathan Tomonaga |
collection | PubMed |
description | BACKGROUND: Langerhans cell histiocytosis (LCH) is a neoplastic transformation of myeloid precursors that commonly presents as an osteolytic lesion of the long or flat bones in children. Aneurysmal bone cysts (ABC) are benign neoplasms that frequently affect the metaphysis of long bones and the spine, often revealing a rapidly expansile lesion with fluid-fluid levels. LCH with secondary ABC-like changes is a rare condition that has only been reported five times, with two presentations in the cranium. The aim of this paper is to review the etiology, clinical and radiographic presentations, and treatment of this condition, as well as to present a novel case on the topic. CASE DESCRIPTION: We describe a 5-year-old boy with a rapidly growing head mass and eye pain resulting in a diagnosis of LCH with secondary ABC-like changes. Radiography demonstrated an expansile, lytic lesion of the left parietal bone with fluid-fluid levels. A confirmatory diagnosis was made through histopathology, demonstrating an inflammatory, histiocytic infiltrate staining positive for CD1a, CD68, CD207 (Langerin), and S-100. The lesion was surgically excised, and the patient recovered without any complications. CONCLUSION: We present a novel case of LCH with secondary ABC-like changes managed with surgical excision. While a radiographic workup with multiple imaging modalities is helpful for diagnosis, a thorough immunohistochemical analysis is essential as imaging characteristics are variable and nonspecific. Furthermore, surgical excision should be considered first-line treatment for lesions involving the skull in surgically accessible areas as it is curative, alleviates symptoms, and allows for histopathological diagnosis. |
format | Online Article Text |
id | pubmed-10643349 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Springer Berlin Heidelberg |
record_format | MEDLINE/PubMed |
spelling | pubmed-106433492023-11-14 Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review Mo, Jonathan Tomonaga Darrow, Morgan Angus Sharma, Julia Devi Childs Nerv Syst Case-based Review BACKGROUND: Langerhans cell histiocytosis (LCH) is a neoplastic transformation of myeloid precursors that commonly presents as an osteolytic lesion of the long or flat bones in children. Aneurysmal bone cysts (ABC) are benign neoplasms that frequently affect the metaphysis of long bones and the spine, often revealing a rapidly expansile lesion with fluid-fluid levels. LCH with secondary ABC-like changes is a rare condition that has only been reported five times, with two presentations in the cranium. The aim of this paper is to review the etiology, clinical and radiographic presentations, and treatment of this condition, as well as to present a novel case on the topic. CASE DESCRIPTION: We describe a 5-year-old boy with a rapidly growing head mass and eye pain resulting in a diagnosis of LCH with secondary ABC-like changes. Radiography demonstrated an expansile, lytic lesion of the left parietal bone with fluid-fluid levels. A confirmatory diagnosis was made through histopathology, demonstrating an inflammatory, histiocytic infiltrate staining positive for CD1a, CD68, CD207 (Langerin), and S-100. The lesion was surgically excised, and the patient recovered without any complications. CONCLUSION: We present a novel case of LCH with secondary ABC-like changes managed with surgical excision. While a radiographic workup with multiple imaging modalities is helpful for diagnosis, a thorough immunohistochemical analysis is essential as imaging characteristics are variable and nonspecific. Furthermore, surgical excision should be considered first-line treatment for lesions involving the skull in surgically accessible areas as it is curative, alleviates symptoms, and allows for histopathological diagnosis. Springer Berlin Heidelberg 2023-07-31 2023 /pmc/articles/PMC10643349/ /pubmed/37522932 http://dx.doi.org/10.1007/s00381-023-06108-7 Text en © The Author(s) 2023 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 licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence 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 licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . |
spellingShingle | Case-based Review Mo, Jonathan Tomonaga Darrow, Morgan Angus Sharma, Julia Devi Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title | Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title_full | Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title_fullStr | Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title_full_unstemmed | Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title_short | Langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
title_sort | langerhans cell histiocytosis with aneurysmal bone cyst-like changes: a case-based literature review |
topic | Case-based Review |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10643349/ https://www.ncbi.nlm.nih.gov/pubmed/37522932 http://dx.doi.org/10.1007/s00381-023-06108-7 |
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