Littoral-cell angioma of the spleen: A case report

Abstract

Littoral-cell angioma (LCA) is a primary splenic vascular tumor that arises from the normal littoral cells lining the sinus channels of the splenic red pulp. We report a case of LCA of the spleen, which has been infrequently communicated in the literature. A 76-year-old man with a 2-wk history of weight loss, abdominal pain and changes in bowel habits was admitted to our hospital. Imaging studies (CT and MRI) showed multiple lesions in the spleen. Splenectomy was performed. Lining cells were positive for CD31/CD68 markers. Our case was associated with a serrated colonic adenoma. LCA is a benign vascular tumor of the spleen that needs to be included in the differential diagnosis of multiple splenic nodules.

Is littoral cell angioma of the spleen as rare as previously believed in the pediatric population?

“…As in our first case, LCA may be completely asymptomatic and represent an incidental finding made during imaging [1,[9][10][11], or the patient may conversely present with abdominal pain -as described in our second and third examples, along with the possible symptoms of hypersplenism (anemia, thrombocytopenia and splenomegaly). This presents a diagnostic dilemma, since the differential diagnosis of such a lesion is thus made inadvertently complex [7].…”

“…Atypical cells are absent, and mitotic activity is usually very low. High-field magnification typically reveals tall and plump sinuslining cells with little mitosis, and no cytologic atypia to suggest malignancy [11,[16][17][18][19][20].…”

Vasoformative Disorder, Resembling Littoral Cell Angioma, of the Spleen in a Geriatric Japanese Macaque(Macaca fuscata)

“…21 Pathologic diagnosis is based on neoplastic proliferation of anastomosing vascular channels rimmed by cells reacting immunohistochemically to both endothelial cell and macrophage markers. 3,5,6,21 The present macaque splenic lesion was composed of diffuse proliferation of vague vascular channels, whose structures closely resembled splenic sinuses. Therefore, there seemed to be gradual transition from normal-appearing red-pulp sinuses to vasformative lesions.…”

“…6 The littoral cells are considered to be unique lining cells possessing both vascular endothelial and macrophage-like properties. 3,5,6,11,21 Splenic hemangiomatosis consists of vascular endothelial cells forming cavernous and capillary vessels but lacks a macrophagelike nature of the neoplastic cells. 4,16 Here, we describe a macaque case of a vasoformative lesion of the spleen, the pathologic characteristics of which bore a resemblance to those of littoral cell angiomas of the spleen in humans.…”

“…4,16 Here, we describe a macaque case of a vasoformative lesion of the spleen, the pathologic characteristics of which bore a resemblance to those of littoral cell angiomas of the spleen in humans. 3,5,6,21 In veterinary literature, there have been no reports of littoral cell angiomas in domestic and experimental animals. 7,19 Splenic vasoformative disorders in humans are often accompanied by a disturbance of blood coagulation.…”