Skip to main content
Top
Published in: Diagnostic Pathology 1/2018

Open Access 01-12-2018 | Case Report

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

Authors: Na Cheng, Jianning Chen, Yuhang Pan, Ye Jiang, Jing Zhou, Chunkui Shao

Published in: Diagnostic Pathology | Issue 1/2018

Login to get access

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.
Literature
1.
go back to reference Falk S, Stutte HJ. Hamartomas of the spleen: a study of 20 biopsy cases. Histopathology. 1989;14(6):603–12.CrossRefPubMed Falk S, Stutte HJ. Hamartomas of the spleen: a study of 20 biopsy cases. Histopathology. 1989;14(6):603–12.CrossRefPubMed
2.
go back to reference Lee H, Maeda K. Hamartoma of the spleen. Arch Pathol Lab Med. 2009;133(1):147–51.PubMed Lee H, Maeda K. Hamartoma of the spleen. Arch Pathol Lab Med. 2009;133(1):147–51.PubMed
3.
go back to reference Ali TZ, Beyer G, Taylor M, Volpe C, Papadimitriou JC. Splenic hamartoma: immunohistochemical and ultrastructural profile of two cases. Int J Surg Pathol. 2005;13(1):103–11.CrossRefPubMed Ali TZ, Beyer G, Taylor M, Volpe C, Papadimitriou JC. Splenic hamartoma: immunohistochemical and ultrastructural profile of two cases. Int J Surg Pathol. 2005;13(1):103–11.CrossRefPubMed
5.
go back to reference Cheuk W, Lee AK, Arora N, Ben-Arie Y, Chan JK. Splenic hamartoma with bizarre stromal cells. Am J Surg Pathol. 2005;29(1):109–14.CrossRefPubMed Cheuk W, Lee AK, Arora N, Ben-Arie Y, Chan JK. Splenic hamartoma with bizarre stromal cells. Am J Surg Pathol. 2005;29(1):109–14.CrossRefPubMed
6.
go back to reference Lam KY, Yip KH, Peh WC. Splenic vascular lesions: unusual features and a review of the literature. Aust N Z J Surg. 1999;69(6):422–5.CrossRefPubMed Lam KY, Yip KH, Peh WC. Splenic vascular lesions: unusual features and a review of the literature. Aust N Z J Surg. 1999;69(6):422–5.CrossRefPubMed
7.
go back to reference Zukerberg LR, Kaynor BL, Silverman ML, Harris NL. Splenic hamartoma and capillary hemangioma are distinct entities: immunohistochemical analysis of CD8 expression by endothelial cells. Hum Pathol. 1991;22(12):1258–61.CrossRefPubMed Zukerberg LR, Kaynor BL, Silverman ML, Harris NL. Splenic hamartoma and capillary hemangioma are distinct entities: immunohistochemical analysis of CD8 expression by endothelial cells. Hum Pathol. 1991;22(12):1258–61.CrossRefPubMed
8.
go back to reference Ramdall RB, Alasio TM, Cai G, Yang GC. Primary vascular neoplasms unique to the spleen: littoral cell angioma and splenic hamartoma diagnosis by fine-needle aspiration biopsy. Diagn Cytopathol. 2007;35(3):137–42.CrossRefPubMed Ramdall RB, Alasio TM, Cai G, Yang GC. Primary vascular neoplasms unique to the spleen: littoral cell angioma and splenic hamartoma diagnosis by fine-needle aspiration biopsy. Diagn Cytopathol. 2007;35(3):137–42.CrossRefPubMed
9.
go back to reference Conlon S, Royston D, Murphy P. Splenic hamartoma. Cytopathology. 2007;18(3):200–2.PubMed Conlon S, Royston D, Murphy P. Splenic hamartoma. Cytopathology. 2007;18(3):200–2.PubMed
10.
go back to reference Arber DA, Strickler JG, Chen YY, Weiss LM. Splenic vascular tumors: a histologic, immunophenotypic, and virologic study. Am J Surg Pathol. 1997;21(7):827–35.CrossRefPubMed Arber DA, Strickler JG, Chen YY, Weiss LM. Splenic vascular tumors: a histologic, immunophenotypic, and virologic study. Am J Surg Pathol. 1997;21(7):827–35.CrossRefPubMed
11.
go back to reference Levy AD, Abbott RM, Abbondanzo SL. Littoral cell angioma of the spleen: CT features with clinicopathologic comparison. Radiology. 2004;230(2):485–90.CrossRefPubMed Levy AD, Abbott RM, Abbondanzo SL. Littoral cell angioma of the spleen: CT features with clinicopathologic comparison. Radiology. 2004;230(2):485–90.CrossRefPubMed
12.
go back to reference Abramowsky C, Alvarado C, Wyly JB, Ricketts R. "Hamartoma" of the spleen (splenoma) in children. Pediatr Dev Pathol. 2004;7(3):231–6.CrossRefPubMed Abramowsky C, Alvarado C, Wyly JB, Ricketts R. "Hamartoma" of the spleen (splenoma) in children. Pediatr Dev Pathol. 2004;7(3):231–6.CrossRefPubMed
13.
go back to reference Darden JW, Teeslink R, Parrish A. Hamartoma of the spleen: a manfestation of tuberous sclerosis. Am Surg. 1975;41(9):564–6.PubMed Darden JW, Teeslink R, Parrish A. Hamartoma of the spleen: a manfestation of tuberous sclerosis. Am Surg. 1975;41(9):564–6.PubMed
14.
go back to reference Van Heerden JA, Longo MF, Cardoza F, Farrow GM. The abdominal mass in the patient with tuberous sclerosis. Surgical implications and report of a case. Arch Surg. 1967;95(2):317–9.CrossRefPubMed Van Heerden JA, Longo MF, Cardoza F, Farrow GM. The abdominal mass in the patient with tuberous sclerosis. Surgical implications and report of a case. Arch Surg. 1967;95(2):317–9.CrossRefPubMed
15.
go back to reference Laskin WB, Alasadi R, Variakojis D. Splenic hamartoma. Am J Surg Pathol. 2005;29(8):1114–5.PubMed Laskin WB, Alasadi R, Variakojis D. Splenic hamartoma. Am J Surg Pathol. 2005;29(8):1114–5.PubMed
16.
go back to reference Yigit N, Covey S, Tam W. Massive splenic hamartoma with bizarre stromal cells. Int J Hematol. 2015;101(4):315–6.CrossRefPubMed Yigit N, Covey S, Tam W. Massive splenic hamartoma with bizarre stromal cells. Int J Hematol. 2015;101(4):315–6.CrossRefPubMed
17.
go back to reference Collins BT, Phillips NJ, Hsueh EC. Fine needle aspiration biopsy of splenic hamartoma with bizarre stromal cells: a case report. Acta Cytol. 2008;52(3):347–50.CrossRefPubMed Collins BT, Phillips NJ, Hsueh EC. Fine needle aspiration biopsy of splenic hamartoma with bizarre stromal cells: a case report. Acta Cytol. 2008;52(3):347–50.CrossRefPubMed
18.
go back to reference Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol. 1995;19(8):859–72.CrossRefPubMed Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol. 1995;19(8):859–72.CrossRefPubMed
19.
go back to reference Coffin CM, Hornick JL, Fletcher CD. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol. 2007;31(4):509–20.CrossRefPubMed Coffin CM, Hornick JL, Fletcher CD. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol. 2007;31(4):509–20.CrossRefPubMed
20.
go back to reference Cessna MH, Zhou H, Sanger WG, Perkins SL, Tripp S, Pickering D, et al. Expression of ALK1 and p80 in inflammatory myofibroblastic tumor and its mesenchymal mimics: a study of 135 cases. Mod Pathol. 2002;15(9):931–8.CrossRefPubMed Cessna MH, Zhou H, Sanger WG, Perkins SL, Tripp S, Pickering D, et al. Expression of ALK1 and p80 in inflammatory myofibroblastic tumor and its mesenchymal mimics: a study of 135 cases. Mod Pathol. 2002;15(9):931–8.CrossRefPubMed
21.
go back to reference Grogg KL, Macon WR, Kurtin PJ, Nascimento AG. A survey of clusterin and fascin expression in sarcomas and spindle cell neoplasms: strong clusterin immunostaining is highly specific for follicular dendritic cell tumor. Mod Pathol. 2005;18(2):260–6.CrossRefPubMed Grogg KL, Macon WR, Kurtin PJ, Nascimento AG. A survey of clusterin and fascin expression in sarcomas and spindle cell neoplasms: strong clusterin immunostaining is highly specific for follicular dendritic cell tumor. Mod Pathol. 2005;18(2):260–6.CrossRefPubMed
22.
go back to reference Chan JK, Fletcher CD, Nayler SJ, Cooper K. Follicular dendritic cell sarcoma. Clinicopathologic analysis of 17 cases suggesting a malignant potential higher than currently recognized. Cancer. 1997;79(2):294–313.CrossRefPubMed Chan JK, Fletcher CD, Nayler SJ, Cooper K. Follicular dendritic cell sarcoma. Clinicopathologic analysis of 17 cases suggesting a malignant potential higher than currently recognized. Cancer. 1997;79(2):294–313.CrossRefPubMed
23.
go back to reference Abbott RM, Levy AD, Aguilera NS, Gorospe L, Thompson WM. From the archives of the AFIP: primary vascular neoplasms of the spleen: radiologic-pathologic correlation. Radiographics. 2004;24(4):1137–63.CrossRefPubMed Abbott RM, Levy AD, Aguilera NS, Gorospe L, Thompson WM. From the archives of the AFIP: primary vascular neoplasms of the spleen: radiologic-pathologic correlation. Radiographics. 2004;24(4):1137–63.CrossRefPubMed
24.
go back to reference Giovagnoni A, Giorgi C, Goteri G. Tumours Of the spleen. Cancer Imaging 2005;5:73-77. Giovagnoni A, Giorgi C, Goteri G. Tumours Of the spleen. Cancer Imaging 2005;5:73-77.
25.
go back to reference Harada S, Kalla H, Balasubramanian M, Brodsky I, Gladstone D, Hou JS. Classical Hodgkin lymphoma concurrently evolving in a patient with marginal zone B-cell lymphoma of the spleen. Ann Diagn Pathol. 2008;12(3):212–6.CrossRefPubMed Harada S, Kalla H, Balasubramanian M, Brodsky I, Gladstone D, Hou JS. Classical Hodgkin lymphoma concurrently evolving in a patient with marginal zone B-cell lymphoma of the spleen. Ann Diagn Pathol. 2008;12(3):212–6.CrossRefPubMed
Metadata
Title
Splenic hamartoma with bizarre stromal cells: a case report and literature review
Authors
Na Cheng
Jianning Chen
Yuhang Pan
Ye Jiang
Jing Zhou
Chunkui Shao
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Diagnostic Pathology / Issue 1/2018
Electronic ISSN: 1746-1596
DOI
https://doi.org/10.1186/s13000-018-0687-y

Other articles of this Issue 1/2018

Diagnostic Pathology 1/2018 Go to the issue