A Rare Case of Splenic Littoral Cell Angioma in a Child

ABSTRACTLittoral cell angioma (LCA) is a rare, benign primary vascular neoplasm of the spleen. The tumor originates from the littoral cells lining the sinuses of the red pulp of the spleen. Preoperative distinction of this tumor from other benign or malign splenic lesions is difficult. Radiologically most cases present as multiple nodules. Definitive diagnosis can only be made histopathologically and immunohistochemically following splenectomy. This clinical situation can coexist with various malignancies and autoimmune disorders. Even though, it is mostly benign, since it has the potential to become malignant after splenectomy, long-term follow-up is required. We present an LCA case, which appeared as a solitary mass in the spleen of an 11-year-old girl with abdominal pain admitted to our hospital.

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Multi-Focal Splenic Tumour in a Belgian Patient and a Brief Review of the Literature on Littoral Cell Angioma

European Journal of Case Reports in Internal Medicine ◽

10.12890/2020_001863 ◽

2020 ◽

Author(s):

Maaike Ramael ◽

Patrick Schoeters ◽

Karl De Pooter ◽

Frederik Van Sonhoven ◽

Hilde Van Steelandt ◽

...

Keyword(s):

Vascular Tumour ◽

Review Of The Literature ◽

Littoral Cell ◽

Littoral Cell Angioma ◽

First Case ◽

Littoral Cells ◽

Splenic Red Pulp ◽

Underlying Pathology ◽

Red Pulp

We describe the case of a 66-year-old woman with littoral cell angioma (LCA) confirmed by histopathology and immunohistochemistry, to our knowledge the first case in Belgium. LCA is an extremely rare primary vascular tumour of the splenic red pulp, probably originating from littoral cells. If a splenic mass and nodules are incidentally identified on imaging and the patient has no associated signs or symptoms, LCA should be suspected. Histopathology and adjacent techniques are mandatory for definitive diagnosis. Splenectomy followed by adequate follow-up is necessary to exclude underlying pathology.

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Littoral Cell Angioma of the Spleen: An Additional Report of Four Cases with Emphasis on the Association with Visceral Organ Cancers

Tumori Journal ◽

10.1177/030089169808400516 ◽

1998 ◽

Vol 84 (5) ◽

pp. 595-599 ◽

Cited By ~ 46

Author(s):

Michele Bisceglia ◽

Joshua Z. Sickel ◽

Felice Giangaspero ◽

Vito Gomes ◽

Mostafà Amini ◽

...

Keyword(s):

Vascular Tumor ◽

Clinical Syndrome ◽

Vascular Tumors ◽

Littoral Cell ◽

Littoral Cell Angioma ◽

Tentorial Meningioma ◽

Clinical Observations ◽

Littoral Cells ◽

Tumors Of The Liver

Aims and background Littoral cell angioma (LCA) is an uncommon vascular tumor of the spleen recently described and interpreted as the tumoral counterpart of the normally present littoral cells lining the splenic sinus channels of red pulp. The diagnosis of LCA is suggested by a quite characteristic morphology and confirmed by the demonstration of a hybrid endothelial/histiocytic phenotype. Methods Four original and previously unreported cases of LCA are presented. All four splenic vascular tumors were investigated by light microscopy and immunohistochemistry for endothelial and histiocytic markers. Results All four cases were associated with visceral epithelial malignancies (colorectal adenocarcinoma in two cases, renal and pancreatic adenocarcinoma in one case each). One case was also associated with an intracranial tentorial meningioma. Conclusions We consider our findings as a novelty and signal the possible existence of a clinical syndrome. Five of a total of 21 previously reported cases in the literature were also described as being associated with other cancers (non-Hodgkin's lymphoma in two cases, two not further specified tumors of the liver and brain, an epithelial ovarian cancer, and a non-small cell lung cancer in one case each). Close follow-up and careful investigation in search of a second visceral neoplasm are strongly recommended in cases of LCA, but further clinical observations and more in-depth genetic and molecular studies are needed before any valid conclusions can be drawn.

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Littoral cell angioma mimicking metastatic tumors

Current Issues in Pharmacy and Medical Sciences ◽

10.1515/cipms-2015-0081 ◽

2015 ◽

Vol 28 (4) ◽

pp. 247-249

Author(s):

Justyna Szumilo ◽

Anna Ostrowska ◽

Malgorzata Zdunek ◽

Slawomir Rudzki ◽

Tomasz Chroscicki ◽

...

Keyword(s):

Histopathological Examination ◽

Vascular Tumor ◽

Littoral Cell ◽

Littoral Cell Angioma ◽

Littoral Cells ◽

Upper Abdominal ◽

Splenic Red Pulp ◽

Tomography Appearance ◽

Red Pulp

Abstract Littoral cell angioma is a rare primary, vascular tumor thought to originate from the endothelial cells lining the sinuses of the splenic red pulp (the “littoral cells”). It is a benign, usually asymptomatic lesion diagnosed incidentally. Ultrasound and tomography appearance is not characteristic and histopathological examination is required. This work provides a case-study of littoral cell angioma which was seen in a 55-year-old female who complained of non-specific upper abdominal pain. Computed tomography revealed multiple hypo-attenuated splenic lesions suggestive for metastasis. A splenectomy was performed and routine microscopic examination supported by immunohistochemistry reactions with CD68, CD34 and CD31 showed littoral cell angioma.

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Atipical immunophenotype in a littoral cell angioma

Vojnosanitetski pregled ◽

10.2298/vsp0901063c ◽

2009 ◽

Vol 66 (1) ◽

pp. 63-65 ◽

Cited By ~ 2

Author(s):

Radoje Colovic ◽

Nada Suvajdzic ◽

Nikica Grubor ◽

Natasa Colovic ◽

Tatjana Terzic

Keyword(s):

Differential Diagnosis ◽

Case Report ◽

Final Diagnosis ◽

Vascular Tumor ◽

Littoral Cell ◽

Littoral Cell Angioma ◽

Negative Case ◽

Splenic Neoplasm ◽

Littoral Cells ◽

Red Pulp

Background. Littoral-cell angioma (LCA) is a recently described benign vascular tumor of the spleen, whose imaging and pathologic characteristics have been discussed only by a few authors. The tumor is characterized by a mixture of papillary and cystic areas lined by neoplastic cells deriving from normal splenic lining - littoral cells. The neoplastic LCA cells express both endothelial and histiocytic antigens associated with CD8 negativity, compared with the normal endothelium of the venous sinuses of the spleen red pulp that only expresses endothelial antigens and CD8 positivity. Therefore, the typical and characteristic immunohistochemical pattern of the LCA is as follows: CD31, CD68, CD163, CD21, FVIII antigen positive; CD34, CD8 negative. Case report. We reported a 60-year-old male with moderate nodular splenomegaly with one large hypoechogenic solid lesion and mild thrombocytopenia in whom the diagnosis of LCA was made after the elective splenectomy. Namely, histopathological and immunohistochemical data allowed a final diagnosis of classical LCA in spite of CD21 negativity. As far as we know this is the first reported CD21-negative LCA patient. Histological specimens were presented and differential diagnoses discussed. Conclusion. Littoral-cell angioma is a very rare benign splenic neoplasm that should be considered in the differential diagnosis of multinodular splenomegaly, particularly if the patient has the signs of hypersplenism.

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A Rare Tumor of Nasal Cavity: Glomangiopericytoma

Case Reports in Otolaryngology ◽

10.1155/2014/282958 ◽

2014 ◽

Vol 2014 ◽

pp. 1-3 ◽

Cited By ~ 1

Author(s):

Aysegul Verim ◽

Cigdem Kalaycik Ertugay ◽

Cigdem Tepe Karaca ◽

Pembegul Gunes ◽

Shahrouz Sheidaei ◽

...

Keyword(s):

Nasal Cavity ◽

Facial Pain ◽

Who Classification ◽

Left Nasal Cavity ◽

Vascular Neoplasm ◽

Long Term Follow Up ◽

Thin Walled ◽

Systemic Examination

Glomangiopericytoma is a rare vascular neoplasm characterized by a pattern of prominent perivascular growth. A 72-year-old woman was admitted to our clinic complaining of nasal obstruction, frequent epistaxis, and facial pain. A reddish tumor filling the left nasal cavity was observed on endoscopy and treated with endoscopic excision. Microscopically, closely packed cells interspersed with numerous thin-walled, branching staghorn vessels were seen. Glomangiopericytoma is categorized as a borderline low malignancy tumor by WHO classification. Long-term follow-up with systemic examination is necessary due to high risk of recurrence.

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Therapy and prevention for mental health: What if mental diseases are mostly not brain disorders?

Behavioral and Brain Sciences ◽

10.1017/s0140525x1800105x ◽

2019 ◽

Vol 42 ◽

Cited By ~ 2

Author(s):

John P. A. Ioannidis

Keyword(s):

Mental Health ◽

Mental Disease ◽

Brain Disorders ◽

Social Stressors ◽

Labor Education ◽

Mental Diseases ◽

Long Term Follow Up ◽

Political Decisions

AbstractNeurobiology-based interventions for mental diseases and searches for useful biomarkers of treatment response have largely failed. Clinical trials should assess interventions related to environmental and social stressors, with long-term follow-up; social rather than biological endpoints; personalized outcomes; and suitable cluster, adaptive, and n-of-1 designs. Labor, education, financial, and other social/political decisions should be evaluated for their impacts on mental disease.

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