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Gao T, Wang X, Wei Q, Wang C, Fan Y, Wang Y. Littoral cell angioma of a huge spleen with peripheral blood pancytopenia in a 14-year-old boy: a case report and review of the literature. Front Pediatr 2024; 12:1473930. [PMID: 39544335 PMCID: PMC11560461 DOI: 10.3389/fped.2024.1473930] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/31/2024] [Accepted: 10/09/2024] [Indexed: 11/17/2024] Open
Abstract
Background Splenic littoral cell angioma (LCA) is an exceptionally uncommon malignant potential vascular tumor with infrequent occurrences in pediatric patients. Due to its reliance on histopathological analysis for diagnosis, LCA may be mistakenly identified as other splenic tumors. Patients with LCA may experience anemia or thrombocytopenia, but peripheral blood pancytopenia is infrequent. Case report A 14-year-old boy presented with peripheral blood pancytopenia, necessitating hospitalization after splenomegaly was identified during a physical examination. Following the exclusion of hematological disorders, a splenectomy was conducted; histopathological examination confirmed the diagnoses of LCA. No metastases or recurrences were observed during the 8-month follow-up. To the best of our knowledge, this case represents the first instance of LCA associated with pancytopenia in a pediatric patient. Conclusion LCA can lead to iron-deficiency anemia or thrombocytopenia, with rare occurrences of pancytopenia, potentially resulting in misdiagnosis as a hematological disorder. Surgical intervention remains an effective treatment for LCA.
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Affiliation(s)
| | | | | | | | | | - Yuebin Wang
- Department of General Surgery, The Second Hospital of Lanzhou University, Lanzhou, China
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2
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Gulati S, Jeon H, Vijay A. Littoral cell angiomas: Benign lesion with a penchant for visceral malignancies. Ann Hepatobiliary Pancreat Surg 2023; 27:1-5. [PMID: 36380480 PMCID: PMC9947364 DOI: 10.14701/ahbps.22-062] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/05/2022] [Revised: 08/31/2022] [Accepted: 09/07/2022] [Indexed: 11/17/2022] Open
Abstract
Littoral cell angiomas are rare vascular tumors of the spleen. Because of their rarity, unclear etiopathogenesis, and association with other malignancies, these tumors can pose diagnostic and therapeutic challenges. Due to paucity of published literature on this entity often limited to case reports, relevant data on this topic were procured and synthesized with the aid of a comprehensive Medline search in addition to oncologic, pathologic, radiologic, and surgical literature review on littoral cell angiomas. This article provides an in-depth review into postulated etiopathogenesis, pathology, clinical manifestations, associated malignancies, and prognostic features of littoral cell angiomas.
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Affiliation(s)
- Snigdha Gulati
- Department of Cardio-Thoracic and Neurosurgery, All India Institute of Medical Sciences (AIIMS), New Delhi, India
| | - Hoonbae Jeon
- Division of Hepato-Pancreato-Biliary & Abdominal Transplant Surgery, Tulane University School of Medicine, New Orleans, LA, United States
| | - Adarsh Vijay
- Division of Hepato-Pancreato-Biliary & Abdominal Transplant Surgery, Tulane University School of Medicine, New Orleans, LA, United States,Corresponding author: Adarsh Vijay, MD Division of Hepato-Pancreato-Biliary & Abdominal Transplant Surgery, Tulane Abdominal Transplant Institute, Tulane University School of Medicine, 1415 Tulane Ave., #HC-05, New Orleans, LA 70112-2632, United States Tel: +1-504-988-0794, Fax: +1-504-988-7510, E-mail: ORCID: https://orcid.org/0000-0002-6108-5218
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3
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Arcuri PP, Taglianetti S, Vavalà B, Battaglia C, Laganà D, Manti F. Incidental littoral cell angioma of the spleen: cross-sectional imaging findings and review of the literature. Radiol Case Rep 2022; 17:3545-3550. [PMID: 35923330 PMCID: PMC9340147 DOI: 10.1016/j.radcr.2022.06.063] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/18/2022] [Revised: 06/13/2022] [Accepted: 06/21/2022] [Indexed: 11/24/2022] Open
Abstract
Littoral cell angioma (LCA) is a primary splenic hemangioma found mostly in normal red sinus shore cells of the reticuloendothelial cell system of the spleen. In most cases is benign, but sometimes malignancies have been reported. This tumor displayed epithelial and histiocytic properties based on its cell of origin, splenic littoral cells. In this case report, we will describe a case of a 21-year-male presenting with an incidentally discovered LCA illustrated by cross-sectional imaging techniques, highlighting how the diffusional sequence and the positron emission tomography study, thanks to their greater specificity, have contributed to reaching a correct diagnostic orientation more than dynamic studies with contrast agent in both computed tomography and magnetic resonance.
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4
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Wang W, Qi G, Zhao X, Zhang Y, Zhu R, Liang R, Sun Y. Clinical Landscape of Littoral Cell Angioma in the Spleen Based on a Comprehensive Analysis. Front Oncol 2022; 12:790332. [PMID: 35211400 PMCID: PMC8861295 DOI: 10.3389/fonc.2022.790332] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/06/2021] [Accepted: 01/12/2022] [Indexed: 11/25/2022] Open
Abstract
OBJECTIVE Littoral cell angioma (LCA) is currently considered to be a rare splenic tumor with malignant potential. As the epidemiology, pathogenesis, clinical manifestation, treatment, and prognosis remain unclear, the clinical diagnosis and treatment of LCA have not been standardized. Hence, we performed a comprehensive analysis of 189 observational studies comprising 435 patients to improve the current status of diagnosis and treatment. METHODS PubMed, Embase, WanFang and CNKI were searched from inception to May 2021 to identify LCA studies that were published in English and Chinese. The clinical information of LCA patients were extracted and analyzed. RESULTS The LCA has a male-to-female ratio of 0.90 and a solitary-to-multiple ratio of 0.31. In terms of clinical features, 69.7% of the patients showed splenomegaly, 49.7% were asymptomatic, and 39.2% experienced epigastric discomfort. As the imaging findings of patients with LCA were nonspecific, an image-guided biopsy (10/12) was a safe and effective method for diagnosing in this condition. Notably, results of the prognostic analysis indicated that LCA has a lower risk of recurrence and metastasis. The patient may develop a stable disease or the tumor will grow but will not metastasize. Besides, the novel immunohistochemical pattern of LCA was described as CD31+/ERG+/FVIII Antigen+/CD68+/CD163+/lysozyme+/CD8-/WT1-. CONCLUSION LCA should be reconsidered as a benign primary splenic vascular neoplasm, which is more like an intra-splenic manifestation of abnormal body function. Image-guided biopsy with follow-up might be a beneficial choice for LCA patients. For LCA patients with abdominal discomfort, pathological uncertainty or continuous tumor enlargement, splenectomy remains the preferred treatment.
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Affiliation(s)
- Weijie Wang
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Guangzhao Qi
- Department of Pharmacy, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Xiangtian Zhao
- Department of Radiology, Guangdong Provincial People’s Hospital, Guangdong Academy of Medical Sciences, Guangzhou, China
| | - Yanping Zhang
- Department of Pathology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Rongtao Zhu
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Ruopeng Liang
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Yuling Sun
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China
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5
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Yaney A, Jones D, Perry KA, Jhawar SR. Rare case of littoral cell angioma recognised on CT simulation for adjuvant radiation treatment for early stage breast cancer. BMJ Case Rep 2022; 15:e248167. [PMID: 35131803 PMCID: PMC8823223 DOI: 10.1136/bcr-2021-248167] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/08/2022] [Indexed: 11/04/2022] Open
Abstract
A 63-year-old woman with pathological anatomic/prognostic stage IA ER+/PR+/HER2- multifocal invasive mammary carcinoma with mixed ductal and lobular features of the left breast underwent lumpectomy with sentinel lymph node biopsy. During evaluation of the CT simulation for adjuvant radiation (RT) treatment planning, a liver lesion and splenomegaly were noted. A subsequent MRI abdomen revealed a large, indeterminate splenic lesion with a wide differential and a benign liver cyst. The patient ultimately underwent splenectomy, with pathology consistent with littoral cell angioma, a rare entity associated with several malignancies. At last follow-up, 7 months after completion of RT, she remained cancer free while on anastrazole and was doing well clinically.
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Affiliation(s)
- Alexander Yaney
- Department of Radiation Oncology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
| | - Daniel Jones
- Department of Pathology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
| | - Kyle A Perry
- Department of General Surgery, The Ohio State University Wexner Medical Center, Columbus, OH, USA
| | - Sachin R Jhawar
- Department of Radiation Oncology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
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6
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Li MJ, Zhou X, Cao JY, Zhu CZ, Zhou SS, Zang YJ, Wu LQ. Laparoscopic splenectomy for littoral cell angioma of the spleen: A case report. Medicine (Baltimore) 2019; 98:e14825. [PMID: 30882665 PMCID: PMC6426590 DOI: 10.1097/md.0000000000014825] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022] Open
Abstract
RATIONALE Littoral cell angioma (LCA) is a rare primary vascular neoplasm of the spleen. It can be benign or malignant. Pathology and immunohistochemistry are the gold standards for the diagnosis of LCA. Therefore, splenectomy is recommended for the purpose of diagnosis and treatment, and subsequent follow-up is necessary. There are limited reports about LCA. Here, we present a case of a female patient with LCA undergoing laparoscopic splenectomy in order to provide clinical experience in LCA treatment. PATIENT CONCERNS A 32-year-old female attended the outpatient Department of Hepatobiliary Surgery for follow-up of hepatic hemangiomas. The patient presented with intermittent abdominal distension, which was slightly under no obvious inducement. DIAGNOSIS Physical examination found no signs of abdominal tenderness and rebound tenderness, and liver and spleen were impalpable. The contrast-enhanced computed tomography (CT) showed multiple space-occupying lesions in the spleen, mottled low-density lesions, multiple hypoattenuating nodules with no contrast enhancement on the arterious phase. Delayed contrast-enhanced helical CT scan displayed incomplete filling of hypodense splenic lesions. INTERVENTIONS Given that it was uncertain whether it was a benign or a malignant tumor, a laparoscopic total splenectomy was performed. OUTCOMES The final pathological diagnosis was LCA. Her postsurgical course was uneventful, and no surgery-related complications were found. No signs of recurrence were observed in the 16 months after the operation. LESSONS LCA was a rare primary vascular neoplasm of the spleen, and laparoscopic splenectomy for LCA was safe and feasible, and postoperative course was uneventful. However, regular follow-up and long-time monitoring after splenectomy for LCA is recommended because of its potential malignant biological behavior.
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Affiliation(s)
- Man-Jiang Li
- Medical College of Qingdao University, Qingdao University
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - Xuan Zhou
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - Jing-Yu Cao
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - Cheng-Zhan Zhu
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - San-Shun Zhou
- Medical College of Qingdao University, Qingdao University
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - Yun-Jin Zang
- Medical College of Qingdao University, Qingdao University
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
| | - Li-Qun Wu
- Medical College of Qingdao University, Qingdao University
- Department of Liver Disease Center, Affiliated Hospital of Qingdao University, Qingdao, China
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7
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Anbardar MH, Kumar PV, Forootan HR. Littoral cell angioma of the spleen: Cytological findings and review of the literature. J Cytol 2017; 34:121-124. [PMID: 28469325 PMCID: PMC5398021 DOI: 10.4103/joc.joc_118_15] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/06/2023] Open
Abstract
Littoral cell angioma (LCA) is a unique lesion of the spleen that arises from the cells lining the venous sinuses of the splenic red pulp and shows the features of combined endothelial and histiocytic differentiation. Several cases of LCA have been reported in the literature; however, the cytological findings have been described for only a few cases. We report the case of an 11-year-old boy with anemia, epigastric abdominal pain, and splenomegaly. The splenic lesions showed anastomosing vascular channels with cyst-like spaces filled by many sloughed endothelial cells, which were positive for CD68 and CD31 and negative for CD34. Scraping cytology revealed isolated and clusters of three-dimensional bland looking, epithelioid foamy tumoral cells with low nuclear cytoplasmic ratio, which mostly contained intracytoplasmic hemosiderin pigment. Although the fine needle aspiration cytology of splenic lesions is uncommon and LCA is a rare splenic lesion, it must be noted in the differential diagnosis of any splenic vascular neoplasm.
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Affiliation(s)
- Mohammad H Anbardar
- Department of Pathology, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Perikala V Kumar
- Department of Pathology, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Hamid R Forootan
- Department of Pediatric Surgery, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
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8
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Kumar B. Left-Sided Flank Pain, Fevers, and Fatigue in a 26-Year-Old Male With Seropositive Rheumatoid Arthritis. Arthritis Care Res (Hoboken) 2016; 68:1714-1720. [PMID: 26414970 DOI: 10.1002/acr.22735] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/30/2015] [Revised: 08/11/2015] [Accepted: 09/15/2015] [Indexed: 11/05/2022]
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9
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Peckova K, Michal M, Hadravsky L, Suster S, Damjanov I, Miesbauerova M, Kazakov DV, Vernerova Z, Michal M. Littoral cell angioma of the spleen: a study of 25 cases with confirmation of frequent association with visceral malignancies. Histopathology 2016; 69:762-774. [PMID: 27374010 DOI: 10.1111/his.13026] [Citation(s) in RCA: 31] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/01/2016] [Revised: 06/28/2016] [Accepted: 06/30/2016] [Indexed: 12/20/2022]
Abstract
AIMS Littoral cell angioma (LCA) is a rare primary splenic tumour that is frequently associated with internal malignancies. Immunohistochemistry can demonstrate a distinct hybrid endothelial-histiocytic phenotype of littoral cells, and is a helpful adjunct for making the correct diagnosis. The aims of this study were to present a series of 25 LCAs, with an emphasis on the frequent association of the neoplasm with visceral malignancies, and to provide a detailed immunohistochemical analysis by employing new markers. METHODS AND RESULTS All 25 cases with available tissue blocks were immunohistochemically stained for endothelial and histiocytic markers. Clinical and follow-up data were retrieved from the respective institutions. The tumours were obtained from 16 males and nine females, whose age ranged from 32 to 86 years (mean 56.2 years). Clinical information was available for 24 of 25 patients, and follow-up for 11 of 25 patients (range 2-19 years; mean 11.6 years). Immunohistochemically, all cases were positive for LYVE-1, factor VIII, FLI-1, vascular endothelial growth factor receptor (VEGFR)-2, VEGFR-3, claudin-5, ERG, LMO2, CD31, CD163, lysozyme, and CD4, but negative for D2-40, CD8, and factor XIIIa. Fifteen of 25 cases were associated with various malignancies, including epithelial, mesenchymal and haematological tumours. CONCLUSIONS The cohort of 25 patients is the largest series of LCAs published to date. By using antibodies against recently introduced endothelial markers, we have expanded the immunoprofile of LCA. We have further highlighted the clinical significance of LCA, as more than half of the patients in this study also harboured a coexisting visceral malignancy. Therefore, we conclude that the finding of splenic LCA mandates a thorough clinical evaluation for a concomitant malignancy.
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Affiliation(s)
- Kvetoslava Peckova
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic.
| | - Michael Michal
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic.,Biomedical Centre, Faculty of Medicine in Plzen and Charles University Hospital Plzen, Pilsen, Czech Republic
| | - Ladislav Hadravsky
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic
| | - Saul Suster
- Department of Pathology and MCW Cancer Center, Medical College of Wisconsin, Milwaukee, WI, USA
| | - Ivan Damjanov
- Department of Pathology and Laboratory Medicine, The University of Kansas School of Medicine, Kansas City, KS, USA
| | - Marketa Miesbauerova
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic
| | - Dmitry V Kazakov
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic
| | - Zdenka Vernerova
- Department of Pathology, Charles University, Third Medical Faculty and Charles University Hospital Kralovske Vinohrady, Prague, Czech Republic
| | - Michal Michal
- Department of Pathology, Charles University, Medical Faculty and Charles University Hospital Plzen, Pilsen, Czech Republic
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de Ridder GG, Galeotti J, Carney J, Wang E. Persistent thrombocytopaenia in a young man with splenomegaly, rebound thrombocytosis after splenectomy and subsequent pulmonary embolism: splenic littoral cell angioma and associated events. BMJ Case Rep 2015; 2015:bcr-2015-212882. [PMID: 26604237 DOI: 10.1136/bcr-2015-212882] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/18/2022] Open
Abstract
Littoral cell angioma (LCA) is a rare endothelial cell neoplasm in the spleen. Although many cases of LCA are asymptomatic, some present with signs and symptoms related to splenomegaly, whereas others manifest with haematological abnormalities, including anaemia and/or thrombocytopaenia (ie, hypersplenism). We report a case of LCA presenting with chronic thrombocytopaenia, probably due to splenic sequestration of platelets or phagocytosis of platelets by neoplastic cells. Following therapeutic splenectomy, the patient suffered from a marked rebound thrombocytosis and subsequently developed pulmonary embolisms. He was treated with anticoagulant therapy combined with antiplatelet therapy, and his symptoms were quickly resolved. This case emphasises an exclusion of primary splenic disorders in patients with chronic thrombocytopaenia, especially in those with splenomegaly and the contemplation of thromboembolism prophylaxis postsplenectomy.
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Affiliation(s)
| | - Jonathan Galeotti
- Department of Pathology, Duke University Medical Center, Durham, North Carolina, USA
| | - John Carney
- Department of Pathology, Duke University Medical Center, Durham, North Carolina, USA
| | - Endi Wang
- Department of Pathology, Duke University Medical Center, Durham, North Carolina, USA
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11
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Marzetti A, Messina F, Prando D, Verza LA, Vacca U, Azabdaftari A, Rubinato L, Reale D, Favat M, Barbujani M, Agresta F. Laparoscopic splenectomy for a littoral cell angioma of the spleen: Case report. World J Clin Cases 2015; 3:951-955. [PMID: 26601099 PMCID: PMC4644898 DOI: 10.12998/wjcc.v3.i11.951] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/08/2015] [Revised: 06/27/2015] [Accepted: 08/07/2015] [Indexed: 02/05/2023] Open
Abstract
A littoral cell angioma (LCA) is a primary vascular tumor of the spleen, that can have malignant potential and may present association with other malignancies. This is a case of LCA that was discovered incidentally in a 79-year-old woman who presented with a polycythemia at the time of consultation. The neoplasm was evaluated by ultrasound and computed tomography. The patient underwent a splenectomy that revealed LCA by pathological evaluation. The post-operative outcome was favorable with no complications or recurrent disease. This case presentation, clinical, radiographic, and pathological features of an uncommon splenic tumor can be studied in order to advance our knowledge in our understanding of LCA.
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12
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Bailey A, Vos J, Cardinal J. Littoral cell angioma: A case report. World J Clin Cases 2015; 3:894-899. [PMID: 26488026 PMCID: PMC4607808 DOI: 10.12998/wjcc.v3.i10.894] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/14/2015] [Revised: 06/21/2015] [Accepted: 08/07/2015] [Indexed: 02/05/2023] Open
Abstract
Primary splenic lesions are rare entities among which littoral cell angioma (LCA) is a recently described, uncommon vascular lesion that is unique to the spleen. It has heretofore been described primarily in pathologic series and has been found mostly to behave as a benign entity. A few reports of malignant variants have been reported. We present a case report of a solitary LCA discovered after splenectomy for an incidentally discovered splenic lesion, along with a literature review.
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13
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YANG LING, ZHAO SHUANG, LIU RONGBO. Unicentric mesenteric Castleman's disease with littoral cell angioma, anemia, growth retardation and amenorrhea: A case report. Oncol Lett 2015; 9:1779-1781. [PMID: 25789041 PMCID: PMC4356425 DOI: 10.3892/ol.2015.2933] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2014] [Accepted: 01/08/2015] [Indexed: 02/05/2023] Open
Abstract
Castleman's disease (CD) is a rare lymphoproliferative disorder of unknown origin, and littoral cell angioma (LCA) is a rare vascular tumor of the spleen with an unknown etiology. The current study reports the case of a 28-year-old female who presented with anemia, growth retardation and amenorrhea. Physical examination revealed a mass in the mesentery, splenomegaly with multiple small nodules, hepatomegaly and an infantile uterus. Histopathological analysis of the resected mass and spleen confirmed the diagnosis of hyaline-vascular CD and LCA. The patient's anemia resolved, and menstruation and breast development also commenced following surgery. To the best of our knowledge, this is the first report of CD accompanied by littoral cell angioma, anemia, growth retardation and amenorrhea.
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Affiliation(s)
- LING YANG
- Department of Radiology, West China Hospital, Sichuan University, Chengdu, Sichuan 610000, P.R. China
| | - SHUANG ZHAO
- Department of Radiology, West China Hospital, Sichuan University, Chengdu, Sichuan 610000, P.R. China
| | - RONG-BO LIU
- Department of Radiology, West China Hospital, Sichuan University, Chengdu, Sichuan 610000, P.R. China
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14
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Littoral cell angioma in a patient with Crohn's disease. Case Rep Gastrointest Med 2015; 2015:474969. [PMID: 25705528 PMCID: PMC4326338 DOI: 10.1155/2015/474969] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/24/2014] [Revised: 01/19/2015] [Accepted: 01/20/2015] [Indexed: 12/15/2022] Open
Abstract
Littoral cell angioma is a rare vascular tumor of the spleen. The pathogenesis is unknown but the lesion is associated with several malignancies and immunological disorders. The diagnosis requires histopathological examination. The malignant potential of this lesion is unknown, which is why splenectomy is recommend for all cases. Symptomatic cases generally suffer from hypersplenism and pyrexia. A previously healthy 20-year-old female was diagnosed with colonic Crohn's disease; as part of the work-up a magnetic resonance enterography was performed which showed multiple signal changes of the spleen. The patient reported chronic abdominal pain in the left upper quadrant, malaise, and fever. The unknown splenic lesions prompted a laparoscopic splenectomy; pathology revealed a littoral cell angioma. The abdominal pain and malaise remitted but the fever persisted one year despite adequate treatment of the patient's Crohn's disease. Littoral cell angioma is associated with immune-dysregulation including Crohn's disease with several reported cases. Signs and symptoms of hypersplenism and splenic lesions on imaging should raise suspicion of littoral cell angioma in patients with Crohn's disease. Magnetic resonance enterography to assess disease severity in Crohn's disease may provide an opportunity to study the prevalence and natural history of this rare splenic tumor.
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15
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Bedir R, Sehitoǧlu I, Calapoǧlu AS, Yurdakul C. A rare case of splenic littoral cell angioma in a child. J Lab Physicians 2014; 6:117-20. [PMID: 25328338 PMCID: PMC4196359 DOI: 10.4103/0974-2727.141511] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022] Open
Abstract
Littoral 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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Affiliation(s)
- Recep Bedir
- Department of Pathology, Recep Tayyip Erdogan University of Medical Faculty, Rize, Turkey
| | - Ibrahim Sehitoǧlu
- Department of Pathology, Recep Tayyip Erdogan University of Medical Faculty, Rize, Turkey
| | - Ahmet Salih Calapoǧlu
- Department of Pediatric Surgery, Recep Tayyip Erdogan University of Medical Faculty, Rize, Turkey
| | - Cüneyt Yurdakul
- Department of Pathology, Recep Tayyip Erdogan University of Medical Faculty, Rize, Turkey
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Schlitter MC, Rühl H, Thiesler T, Penner AH, Berens C, Wolf D, Mayer K. Paraneoplastic disseminated intravascular coagulation caused by splenic littoral cell angioma. Ann Hematol 2014; 93:1795-6. [PMID: 24789322 DOI: 10.1007/s00277-014-2060-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/28/2014] [Accepted: 03/15/2014] [Indexed: 11/29/2022]
Affiliation(s)
- Marie Caroline Schlitter
- Medical Clinic III for Oncology, Haematology and Rheumatology, University Hospital Bonn (UKB), Sigmund-Freud-Straße 25, 53127, Bonn, Germany
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17
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Littoral cell angiosarcoma in a dog. J Comp Pathol 2013; 149:221-4. [PMID: 23453493 DOI: 10.1016/j.jcpa.2013.01.003] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/22/2012] [Revised: 11/12/2012] [Accepted: 01/13/2013] [Indexed: 02/06/2023]
Abstract
This report describes the microscopical and immunohistochemical characteristics of littoral cell angiosarcoma in a 12-year-old, neutered female, beagle dog. The dog succumbed to metastatic disease 3 months after diagnosis of a mid-splenic mass. The tumour was characterized by two histological patterns: anastomosing microvascular channels and microvascular papillary fronds. The neoplastic cells expressed both endothelial and histiocytic markers and were erythrophagocytic. Immunohistochemical findings consistent with malignancy were CD34 expression and high Ki67 nuclear immunoreactivity.
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18
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Larsen BT, Bishop MC, Hunter GC, Renner SW. Low-Grade, Metastasizing Splenic Littoral Cell Angiosarcoma Presenting With Hepatic Cirrhosis and Splenic Artery Aneurysm. Int J Surg Pathol 2013; 21:618-26. [DOI: 10.1177/1066896913476737] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
The behavior of littoral cell neoplasms ranges from benign (littoral cell angioma, LCA) to highly malignant (angiosarcoma). Two unusual cases of low-grade metastatic littoral cell angiosarcoma (LCAS) have been reported with late recurrence and bulky metastases. We present the third case of this rare neoplasm in a 38-year-old man with cirrhosis and a large splenic artery aneurysm, without extrasplenic masses. The spleen showed nodules resembling LCA, immunoreactive for CD31, factor VIII, CD68, and CD163 but not CD8 or CD34. Also present were solid areas of immunophenotypically identical bland spindle cells, although lighter CD31 immunostaining distinguished them from LCA-like angiomatous channels. Similar cells diffusely infiltrated the cirrhotic liver. After splenectomy, pancytopenia resolved, and he is asymptomatic 19 months later. Low-grade LCAS is a previously unreported cause of cirrhosis and may metastasize without forming masses. In cases of LCA, CD31 immunohistochemistry may facilitate detection of LCAS and indicate metastatic potential.
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Affiliation(s)
- Brandon T. Larsen
- University of Arizona, Tucson, AZ, USA
- Southern Arizona Veterans Affairs Health Care System, Tucson, AZ, USA
| | - Maria C. Bishop
- Southern Arizona Veterans Affairs Health Care System, Tucson, AZ, USA
| | - Glenn C. Hunter
- Southern Arizona Veterans Affairs Health Care System, Tucson, AZ, USA
| | - Stephen W. Renner
- Southern Arizona Veterans Affairs Health Care System, Tucson, AZ, USA
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19
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Littoral cell angioma and angiosarcoma of the spleen: report of two cases in siblings and review of the literature. J Gastrointest Surg 2012; 16:863-7. [PMID: 22068970 DOI: 10.1007/s11605-011-1773-6] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/28/2011] [Accepted: 10/26/2011] [Indexed: 01/31/2023]
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20
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Melzer N, Barth PJ, Müller KM, Foss HD, Krug U, Schilling M, Marziniak M, Grauer OM, Wiendl H. Rapidly progressive B-cell dominated inflammatory neuropathy and littoral cell angioma of the spleen associated with plasmablastic B-cell lymphoma. Leuk Lymphoma 2012; 53:1242-4. [DOI: 10.3109/10428194.2011.640677] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Affiliation(s)
- Nico Melzer
- Inflammatory Disorders of the Nervous System and Neurooncology, Department of Neurology
| | | | | | | | - Utz Krug
- Hematology and Oncology, Department of Medicine A
| | | | - Martin Marziniak
- Inflammatory Disorders of the Nervous System and Neurooncology, Department of Neurology
| | - Oliver Martin Grauer
- Inflammatory Disorders of the Nervous System and Neurooncology, Department of Neurology
| | - Heinz Wiendl
- Inflammatory Disorders of the Nervous System and Neurooncology, Department of Neurology
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21
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Liang W, Lu J, Qin M, Sang X. Littoral cell angioma mimicking hepatic tumor. Acta Radiol Short Rep 2012; 1:10.1258_arsr.2012.120031. [PMID: 23986846 PMCID: PMC3738355 DOI: 10.1258/arsr.2012.120031] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/21/2012] [Accepted: 07/04/2012] [Indexed: 11/18/2022] Open
Abstract
Littoral cell angioma is a rare vascular tumor of the spleen that was described by Falk et al. in 1991. Because of the limited number, untypical imaging manifestations, and lack of knowledge on this tumor type, these tumors are often misdiagnosed. In most cases, the tumor presents with multiple small hypoattenuating nodules in the spleen with delayed enhancement. However, solitary littoral cell angiomas have not been well described. We present the CT features of an unusual littoral cell angioma mimicking hepatic tumor.
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22
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Hu ZQ, A YJ, Sun QM, Li W, Li L. The splenic Littoral cell angioma in China: a case report and review. World J Surg Oncol 2011; 9:168. [PMID: 22172167 PMCID: PMC3271992 DOI: 10.1186/1477-7819-9-168] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/20/2011] [Accepted: 12/15/2011] [Indexed: 01/18/2023] Open
Abstract
Littoral cell angioma (LCA) is a rare splenic vascular neoplasm that arises from the cells lining the red pulp sinuses. It is deemed to be a benign and incidental lesion. The earliest literature report of littoral cell angioma has been described by Falk. The examination of samples after splenectomy reveals similar pathological change and its change rule is summarized. However, many recent reports have described it to be a malignant tumor with congenital and immunological associations. Generally speaking, the definitive diagnosis can only be made after histological and immunohistochemical profiles. In this case report, we presented the case of a 48-year-old woman with multiple splenic LCAs. Initially, the patient was characteristics of abdominal distension, weakness and fatigue. Multiple hemangiomas were observed in the spleen through abdominal ultrasonic diagnosis. Computed tomography (CT) scans revealed the splenomegaly with multiple round and hyperdense lesions. The patient subsequently underwent splenectomy. Postoperative histological and immunohistochemical studies confirmed the diagnosis of LCA. Based on the presentation of this case, clinical, radiographic and pathological results of LCA as well as recent advances in our understanding of this uncommon splenic lesion were reviewed. LCA is an uncommon splenic tumor diagnosed in patients with or without abdominal discomfort. Only a few case reports regarding this kind of tumor have been published as inconsistent results. In the present paper, we have reported a case of LCA and reviewed the literature.
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Affiliation(s)
- Zong-Qiang Hu
- Ganmei Affiliated Hospital of Kunming Medical University, Kunming, China
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23
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Splenic littoral cell hemangioendothelioma in a patient with Crohn's disease previously treated with immunomodulators and anti-TNF agents: a rare tumor linked to deep immunosuppression. Am J Gastroenterol 2011; 106:1863-5. [PMID: 21979217 DOI: 10.1038/ajg.2011.204] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
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24
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Nagarajan P, Cai G, Padda MS, Selbst M, Kowalski D, Proctor DD, Chhieng D, Aslanian HR, Harigopal M. Littoral cell angioma of the spleen diagnosed by endoscopic ultrasound-guided fine-needle aspiration biopsy. Diagn Cytopathol 2011; 39:318-22. [PMID: 21488173 DOI: 10.1002/dc.21384] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
Littoral cell angiomas are uncommon primary vascular neoplasms that arise from the sinusoidal lining or littoral cells of the splenic red pulp, and hence are unique to the spleen. We report a case of littoral cell angioma in 34-year-old woman, which was diagnosed by endoscopic ultrasound-guided fine needle aspiration biopsy (EUS-FNAB). The cytologic features of littoral cell angiomas have been described only in three previous case reports, one of which was a bench-top aspirate. In our case, we have utilized the fine-needle aspiration samples obtained by a linear endoscopic ultrasound examination for establishing the diagnosis. The characteristic cytologic features identified on the smears along with immunohistochemical analysis performed on the compact cellblock prepared from the aspirate aided in the confirmation of the diagnosis. We suggest that EUS-FNAB is a safe and reliable method in the diagnosis of vascular lesions of the spleen.
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Affiliation(s)
- Priyadharsini Nagarajan
- Department of Pathology, Cytopathology, Yale University, School of Medicine, New Haven, CT, USA
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25
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Benetatos N, Filobbos R, Ammori B. Laparoscopic partial splenectomy for littoral cell angioma. J Surg Case Rep 2011; 2011:4. [PMID: 24950042 PMCID: PMC3649272 DOI: 10.1093/jscr/2011.7.4] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023] Open
Abstract
Littoral cell angioma (LCA) is a rare benign splenic vascular neoplasm. We report a case of a 58-year-old man with a 5.5-cm lesion in the upper pole of the spleen. A laparoscopic partial splenectomy was performed. He was discharged on the second postoperative day. Pathology revealed LCA. Laparoscopic partial splenectomy can be performed safely in selected cases and is an appropriate option in patients with benign conditions as it enables preservation of the immunologic function of the spleen. Long-term follow up in patients with LCA is important given its high association with other malignancies.
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Affiliation(s)
- N Benetatos
- Departments of Surgery and Radiology, North Manchester General Hospital, Manchester, UK
| | - R Filobbos
- Departments of Surgery and Radiology, North Manchester General Hospital, Manchester, UK
| | - B Ammori
- Departments of Surgery and Radiology, North Manchester General Hospital, Manchester, UK
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26
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Bhavsar T, Wang C, Huang Y, Karachristos A, Inniss S. Littoral cell angiomas of the spleen associated with solid pseudopapillary tumor of the pancreas. World J Gastrointest Pathophysiol 2011; 2:53-6. [PMID: 21860837 PMCID: PMC3158892 DOI: 10.4291/wjgp.v2.i3.53] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/21/2010] [Revised: 03/31/2011] [Accepted: 04/07/2011] [Indexed: 02/06/2023] Open
Abstract
Littoral cell angiomas (LCA) of the spleen are vascular tumors of unknown etiology arising from the littoral cells of the splenic red pulp sinuses. Usually a benign and incidental finding, LCA have been repeatedly reported in association with a variety of visceral malignancies and hold the potential for dissemination per se. We encountered a case of a 30 year old female who was diagnosed with solid pseudopapillary tumor of the head and distal pancreas by fine needle aspiration cytology. A distal pancreatectomy with splenectomy was performed in addition to a pylorus-preserving Whipple’s procedure and cholecystectomy. Histopathological examination confirmed solid pseudopapillary tumor of the pancreas and showed multiple well-circumscribed anastomosing vascular channels in the spleen. The diagnosis of LCA of the spleen was confirmed by immunohistochemistry that revealed co-expression of endothelial cell marker, CD31 and CD34, along with histiocytic marker, CD68 by the vascular lining cells. LCA has been previously reported in association with colorectal and pancreatic adenocarcinoma, malignant lymphoma, myelodysplasia and autoimmune disorders. We report the first case of LCA associated with solid pseudopapillary tumor of the pancreas.
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27
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Pilz JB, Sperschneider T, Lutz T, Loosli B, Maurer CA. Littoral cell angioma in main and accessory intrapancreatic spleen presenting as splenic rupture. Am J Surg 2010; 201:e15-7. [PMID: 20409532 DOI: 10.1016/j.amjsurg.2009.11.013] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/01/2009] [Revised: 11/24/2009] [Accepted: 11/24/2009] [Indexed: 01/13/2023]
Abstract
We report the incidental finding of a nodular mass in the pancreatic tail on a contrast-enhanced computed tomography scan preinterventional to emergency laparotomy for splenic rupture. Because of the past surgical history and radiologic appearance, differential diagnosis included atypical lymphoma in the spleen and regional lymph node, pancreatic adenocarcinoma with splenic metastasis, and intrapancreatic metastase of malignant melanoma; the patient underwent both splenectomy and pancreatic tail resection. A diagnosis of littoral cell angioma in main and accessory intrapancreatic spleen was made. To our knowledge, this is the first description of littoral cell angioma of the spleen involving both main and accessory organ presenting as splenic rupture.
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Affiliation(s)
- Julia B Pilz
- Department of Gastroenterology, Liestal Hospital, Switzerland
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28
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Berman E, Ikpatt F, Wang D, Dembner A, Zauber NP. Rapid progression of littoral cell angioma of the spleen in a man with multiple infections. Rare Tumors 2010; 2:e17. [PMID: 21139945 PMCID: PMC2994494 DOI: 10.4081/rt.2010.e17] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/15/2009] [Revised: 03/04/2010] [Accepted: 03/05/2010] [Indexed: 01/04/2023] Open
Abstract
Littoral cell angioma is an uncommon primary vascular neoplasm of the spleen. It frequently follows a benign course, but cases with aggressive behavior have been described. We present a case of this rare disease highlighting radiological examinations showing a rapid increase in the size of the spleen as well as an increase in the number and size of existing nodules. We also discuss the relationship of this disorder to chronic infections and its possible association with Epstein Barr virus.
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Affiliation(s)
- Errol Berman
- Saint Barnabas Medical Center, Livingston, New Jersey, USA
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29
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Koh YW, Park BH, Lee SR, Lee H, Huh JR. Splenic Littoral Cell Angioma with Hepatitis C Associated Liver Cirrhosis. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2010. [DOI: 10.4174/jkss.2010.78.3.203] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Affiliation(s)
- Young Wha Koh
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Bong-Hee Park
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sang-Ryung Lee
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Heejin Lee
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Joo Ryung Huh
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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30
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Koh YW, Lee H, Choi G, Kwon GY, Kim EJ, Huh J. Primary Splenic Vascular Lesions: A Clinicopathologic, Immunophenotypic and Radiopathologic Correlation Study of 40 Cases. KOREAN JOURNAL OF PATHOLOGY 2010. [DOI: 10.4132/koreanjpathol.2010.44.5.502] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Affiliation(s)
- Young Wha Koh
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Heejin Lee
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Gawon Choi
- Department of Pathology, Mizmedi Hospital, Seoul, Korea
| | - Gui Young Kwon
- Department of Pathology, Chung-Ang University Hospital, Chung-Ang School of Medicine, Seoul, Korea
| | - Eun-Ju Kim
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Jooryung Huh
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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31
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Abstract
Littoral cell angioma (LCA) is a rare primary benign vascular neoplasm of the spleen. The case of a 52-year-old female with LCA is reported. The patient did not have any symptoms or a significant past medical history. Computed tomography (CT) and ultrasound (US) imaging studies showed multiple lesions in the spleen and gallbladder stones. The tumour was removed successfully by laparoscopic splenectomy and simultaneously cholecystectomy was conducted for gallbladder stones. The patient recovered uneventfully. The tumour lining cells were positive for CD31/CD68 markers, and negative for CD34. This is the first report of a LCA combined with gallbladder stones.
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Affiliation(s)
- Ya-Jun Wang
- Department of General Surgery, Xuanwu Hospital of Capital Medical University, Beijing, China.
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32
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Bierenbaum J, Alapat DV, Godinez C, Park AE, Zhao XF, Baer MR. Littoral cell angioma: a correctable cause of progressive pancytopenia in a patient with myelodysplastic syndrome. Leuk Res 2009; 34:e117-9. [PMID: 19853914 DOI: 10.1016/j.leukres.2009.09.030] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/21/2009] [Revised: 09/23/2009] [Accepted: 09/23/2009] [Indexed: 10/20/2022]
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33
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Tee M, Vos P, Zetler P, Wiseman SM. Incidental littoral cell angioma of the spleen. World J Surg Oncol 2008; 6:87. [PMID: 18713469 PMCID: PMC2527567 DOI: 10.1186/1477-7819-6-87] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/14/2008] [Accepted: 08/19/2008] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Littoral cell angioma (LCA) is a recently described primary vascular neoplasm of the spleen that may be associated with other malignancies and may itself also have malignant potential. CASE PRESENTATION We present a case of LCA that was discovered incidentally in a 52-year-old woman who presented with biliary colic at the time of consultation for cholecystectomy. This vascular neoplasm was evaluated by ultrasound, CT, MRI, Tc-99m labelled red blood cell scintigraphy, and core biopsy. A splenectomy revealed LCA by pathological evaluation. Post-operative outcome was favourable with no evidence of complication or recurrent disease. Following this case presentation, clinical, radiographic, and pathological features of LCA will be reviewed as well as recent advances in our understanding of this uncommon splenic lesion. CONCLUSION LCA is a rare, generally benign, primary vascular tumour of the spleen that typically is discovered incidentally. Individuals diagnosed with this tumour must be carefully evaluated to exclude primary, secondary, and synchronous malignancies.
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Affiliation(s)
- May Tee
- Faculty of Medicine, University of British Columbia, Vancouver, British Columbia, Canada.
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