Korean J Hematol.  2007 Jun;42(2):157-161. 10.5045/kjh.2007.42.2.157.

A Case of Bicytopenia Combined with Littoral Cell Angioma

• Song MK
• Seol YM
• Park YE
• An SK
• Choi YJ
• Shin HJ
• Chung JS
• Cho GJ

• KMID: 2083514
• DOI: http://doi.org/10.5045/kjh.2007.42.2.157

Abstract

Littoral cell angioma is a recently described vascular tumor of spleen with an unknown etiology. We present a case in a 74-year-old woman with severe anemia, thrombocytopenia and palpable splenomegaly. The computed tomography of abdomen showed well-defined multiple nodules in spleen. Splenectomy was performed and the histological and immunohistochemical features of splenic tumor were consistent with a littoral cell angioma. The patients had persistent anemia and thrombocytopenia after splenectomy. We suggest hepatic destruction or other autoimmune mechanism contributes persistent bicytopenia. This case illustrated the refractory bicytopenia combined with littoral cell angioma.

Keyword

Littoral cell angioma, Anemia; Thrombocytopenia; Splenomegaly

MeSH Terms

Abdomen
Aged
Anemia
Female
Hemangioma*
Humans
Spleen
Splenectomy
Splenomegaly
Thrombocytopenia

Figure

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  Fig. 1 Peripheral blood smear showed macrocytic anemia and thrombocytopenia. Reticulocyte, tear-drop cell and stomatocyte were occasionally seen.

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  Fig. 2 Computed tomography scan demonstrated hema-tosplenomegaly and several hypodense areas of spleen.

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  Fig. 3 Grossly, multiple nodules intervening spleen parenchyma were found. The spleen was measuring 490g in weight and 17.0×12.0×6.0cm in dimensions. It was entirely replaced by tumor mass (A). The cut surface of the tumor was multinodular and dark brown in color. It was soft and spongy-like in consistency (B).

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  Fig. 4 Immunohistochemical stains of the original tumor. Littoral-cell angioma lining cells expressed membranous immunohistochemistry staining for CD31 (A) and CD68 (B) (×400).

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  Fig. 5 Spleen scan after intravenous injection of erythrocytes labeled with 99m Technetium. No evidence of accessory spleen was shown.

Reference

1). Falk S., Stutte HJ., Frizzera G. Littoral cell angioma.