Splenic hamartoma with bizarre stromal cells: a case report and literature review

Abstract Background Splenic hamartoma is a rare benign vascular proliferative lesion composed of unorganized sinusoid-like channels lined with plump or flat endothelial cells and characterized by a CD8-positive immunophenotype of the lining cells. Scattered bizarre stromal cells can be found in some...

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Main Authors: Na Cheng, Jianning Chen, Yuhang Pan, Ye Jiang, Jing Zhou, Chunkui Shao
Format: Article
Language:English
Published: BMC 2018-01-01
Series:Diagnostic Pathology
Subjects:
Online Access:http://link.springer.com/article/10.1186/s13000-018-0687-y
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author Na Cheng
Jianning Chen
Yuhang Pan
Ye Jiang
Jing Zhou
Chunkui Shao
author_facet Na Cheng
Jianning Chen
Yuhang Pan
Ye Jiang
Jing Zhou
Chunkui Shao
author_sort Na Cheng
collection DOAJ
description Abstract Background Splenic hamartoma is a rare benign vascular proliferative lesion composed of unorganized sinusoid-like channels lined with plump or flat endothelial cells and characterized by a CD8-positive immunophenotype of the lining cells. Scattered bizarre stromal cells can be found in some splenic hamartomas. The presence of splenic hamartoma with bizarre stromal cells is extremely rare and these bizarre cells make it possible to be regarded as a malignancy. Recognition of this rare histologic variant will help to avoid diagnostic confusion and overtreatment of this benign entity. Case presentation We report a case of a 40-year-old man with occasional left-sided waist back pain. A splenic space-occupying lesion was detected by ultrasound and magnetic resonance imaging. Microscopically bizarre large cells were scattered throughout the splenic hamartoma. The cells exhibited atypical nuclei, scarcely visible cytoplasm, and vesicular chromatin, and they did not form expansile clusters and lacked mitotic activity. An immunohistochemical panel was performed. The bizarre cells strongly expressed vimentin, and the Ki-67 index was very low. The lesion was diagnosed as a splenic hamartoma with bizarre stromal cells. Conclusions To the best of our knowledge, this is the first systematic review on a splenic hamartoma with bizarre stromal cells; only six cases have been described in the literature. Proper identification is important to secure adequate treatment.
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spelling doaj.art-eac1d26e916341169a4dbdba71f3904d2022-12-21T19:07:56ZengBMCDiagnostic Pathology1746-15962018-01-011311710.1186/s13000-018-0687-ySplenic hamartoma with bizarre stromal cells: a case report and literature reviewNa Cheng0Jianning Chen1Yuhang Pan2Ye Jiang3Jing Zhou4Chunkui Shao5Department of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityDepartment of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityDepartment of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityDepartment of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityDepartment of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityDepartment of Pathology, The Third Affiliated Hospital, Sun Yat-sen UniversityAbstract Background Splenic hamartoma is a rare benign vascular proliferative lesion composed of unorganized sinusoid-like channels lined with plump or flat endothelial cells and characterized by a CD8-positive immunophenotype of the lining cells. Scattered bizarre stromal cells can be found in some splenic hamartomas. The presence of splenic hamartoma with bizarre stromal cells is extremely rare and these bizarre cells make it possible to be regarded as a malignancy. Recognition of this rare histologic variant will help to avoid diagnostic confusion and overtreatment of this benign entity. Case presentation We report a case of a 40-year-old man with occasional left-sided waist back pain. A splenic space-occupying lesion was detected by ultrasound and magnetic resonance imaging. Microscopically bizarre large cells were scattered throughout the splenic hamartoma. The cells exhibited atypical nuclei, scarcely visible cytoplasm, and vesicular chromatin, and they did not form expansile clusters and lacked mitotic activity. An immunohistochemical panel was performed. The bizarre cells strongly expressed vimentin, and the Ki-67 index was very low. The lesion was diagnosed as a splenic hamartoma with bizarre stromal cells. Conclusions To the best of our knowledge, this is the first systematic review on a splenic hamartoma with bizarre stromal cells; only six cases have been described in the literature. Proper identification is important to secure adequate treatment.http://link.springer.com/article/10.1186/s13000-018-0687-ySpleenSplenic hamartomaBizarre stromal cellsImmunohistochemistry
spellingShingle Na Cheng
Jianning Chen
Yuhang Pan
Ye Jiang
Jing Zhou
Chunkui Shao
Splenic hamartoma with bizarre stromal cells: a case report and literature review
Diagnostic Pathology
Spleen
Splenic hamartoma
Bizarre stromal cells
Immunohistochemistry
title Splenic hamartoma with bizarre stromal cells: a case report and literature review
title_full Splenic hamartoma with bizarre stromal cells: a case report and literature review
title_fullStr Splenic hamartoma with bizarre stromal cells: a case report and literature review
title_full_unstemmed Splenic hamartoma with bizarre stromal cells: a case report and literature review
title_short Splenic hamartoma with bizarre stromal cells: a case report and literature review
title_sort splenic hamartoma with bizarre stromal cells a case report and literature review
topic Spleen
Splenic hamartoma
Bizarre stromal cells
Immunohistochemistry
url http://link.springer.com/article/10.1186/s13000-018-0687-y
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AT yejiang splenichamartomawithbizarrestromalcellsacasereportandliteraturereview
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