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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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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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Jia F, Lin H, Li YL, Zhang JL, Tang L, Lu PT, Wang YQ, Cui YF, Yang XH, Lu ZY. Early postsurgical lethal outcome due to splenic littoral cell angioma: A case report. World J Clin Cases 2023; 11:1188-1197. [PMID: 36874427 PMCID: PMC9979300 DOI: 10.12998/wjcc.v11.i5.1188] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/14/2022] [Revised: 12/09/2022] [Accepted: 01/16/2023] [Indexed: 02/14/2023] Open
Abstract
BACKGROUND Littoral cell angioma (LCA) is a rare benign vascular tumor of the spleen. Given its rarity, standard diagnostic and therapeutic recommendations have yet to be developed for reported cases. Splenectomy is the only method of obtaining a pathological diagnosis and providing treatment to obtain a favorable prognosis.
CASE SUMMARY A 33-year-old female presented with abdominal pain for one month. Computed tomography and ultrasound revealed splenomegaly with multiple lesions and two accessory spleens. The patient underwent laparoscopic total splenectomy and accessory splenectomy, and splenic LCA was confirmed by pathology. Four months after surgery, the patient presented with acute liver failure, was readmitted, rapidly progressed to multiple organ dysfunction syndrome and died.
CONCLUSION Preoperative diagnosis of LCA is challenging. We systematically reviewed online databases to identify the relevant literature and found a close relationship between malignancy and immunodysregulation. When a patient suffers from both splenic tumors and malignancy or immune-related disease, LCA is possible. Due to potential malignancy, total splenectomy (including accessory spleen) and regular follow-up after surgery are recommended. If LCA is diagnosed after surgery, a comprehensive postoperative examination is needed.
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Affiliation(s)
- Fan Jia
- Department of Abdominal Ultrasound, First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Han Lin
- Department of Hepatic Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Yi-Long Li
- Department of Pancreatic and Biliary Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Jin-Ling Zhang
- Department of Radiology and Nuclear Medicine, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Liang Tang
- Department of Radiology and Nuclear Medicine, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Peng-Tian Lu
- Department of Pathology, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Yu-Qing Wang
- Department of Hematopathology, First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Yi-Feng Cui
- Department of Hepatic Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Xiu-Hua Yang
- Department of Abdominal Ultrasound, First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Zhao-Yang Lu
- Department of Hepatic Surgery, Key Laboratory of Hepatosplenic Surgery, Ministry of Education, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
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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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Vladimirov M, Bausch D, Stein HJ, Keck T, Wellner U. Hybrid Laparoscopic Versus Open Pancreatoduodenectomy. A Meta-Analysis. World J Surg 2022; 46:901-915. [PMID: 35043246 PMCID: PMC8885482 DOI: 10.1007/s00268-021-06372-1] [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] [Accepted: 10/07/2021] [Indexed: 11/30/2022]
Abstract
INTRODUCTION Hybrid laparoscopic techniques have been proposed as a good transition from open to complete minimally invasive approach especially in complex surgical procedures. This meta-analysis aimed to compare the outcomes of hybrid laparoscopic pancreatoduodenectomy versus open pancreatoduodenectomy. METHODS A systematic literature research was performed according to PRISMA guidelines. A broad search strategy with terms "laparoscopy" and "pancreatoduodenectomy" was used. Included studies were analyzed by quantitative meta-analysis using the metafor package for R software. RESULTS Of 655 identified articles, 627 were excluded and 28 articles fully assessed, including 14 comparative studies, 8 case series and 6 case reports. Extracted data included intraoperative variables and postoperative outcome parameters. The predefined inclusion criteria were met by 14 comparative studies, and 371 patients were pooled in the meta-analysis. Hybrid laparoscopic pacreatoduodenectomy was associated with significantly longer operative time (I2 0%, p = 0,01, Mean HPD 494,6 min, Mean OPD 421,6 min, WMD 67 min, 95% CI 14-120 min). For all other postoperative outcome parameters, no statistically significant differences were found. A nonsignificant reduction in intraoperative transfusion rate (I2 20%, p = 0,2, proportion HPD 2%, proportion OPD 1,6%, OR 0,44, 95% CI 0,16-1,27) and blood loss (I2 95%, p = 0,1, Mean HPD 397,2 ml, Mean OPD 1017,8 ml, MD - 601 ml, 95% CI - 1311-108) was observed for hybrid pancreatoduodenectomy in comparison to open surgery. CONCLUSIONS This meta-analysis demonstrates significantly increased operation time for hybrid laparoscopic compared to open pancreatoduodenectomy. Intraoperative variables as well as postoperative parameters and major morbidity were comparable for both techniques. Overall results of this meta-analysis demonstrated the hybrid technique as a safe procedure in high-volume centers offering aspects of a safe transition to fully laparoscopic pancreatoduodenectomy.
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Affiliation(s)
- Miljana Vladimirov
- Klinik für Allgemein, Viszeral- und Thoraxchirurgie, PMU Nürnberg, Nuremberg, Deutschland
| | - Dirk Bausch
- Klinik für Chirurgie, Universitätsklinikum Schleswig-Holstein, Campus Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Deutschland
| | - Hubert J Stein
- Klinik für Allgemein, Viszeral- und Thoraxchirurgie, PMU Nürnberg, Nuremberg, Deutschland
| | - Tobias Keck
- Klinik für Chirurgie, Universitätsklinikum Schleswig-Holstein, Campus Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Deutschland.
| | - Ulrich Wellner
- Klinik für Chirurgie, Universitätsklinikum Schleswig-Holstein, Campus Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Deutschland
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Case report: Infrequent littoral cell angioma of the spleen. Int J Surg Case Rep 2021; 85:106242. [PMID: 34333257 PMCID: PMC8346658 DOI: 10.1016/j.ijscr.2021.106242] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/19/2021] [Revised: 07/21/2021] [Accepted: 07/24/2021] [Indexed: 12/30/2022] Open
Abstract
Introduction and importance Littoral cell angioma is a rare solid spleen tumor with uncertain malignant potential. It is usually asymptomatic; therefore, its diagnosis is usually incidental. There are approximately 150 cases reported in the medical literature, but none of them in the Hispanic population. Case presentation We present a case of a 54-year-old woman who presented to our clinic with nonspecific abdominal pain. Imaging studies show a splenic mass with littoral cell angioma characteristics. The patient underwent an open splenectomy with subsequent histopathologic and immunohistochemical studies that confirmed the presence of a littoral cell angioma of a diameter of 8 × 4.5 × 3.5 cm. The patient was discharged after an uneventful postoperative recovery and was referred to the outpatient clinic for follow up. Clinical discussion This case report highlights the close relationship between the littoral cell angioma, neoplasias, and autoimmune diseases. Even though LCA has a good prognosis, there is still the possibility of malignant transformation, especially when the spleen weighs 1500 g; our patient's sample pointed towards a benign pathology. LCA has a positive IHC for endothelial and histiocyte tissues. The IHC results of our patient were positive for CD34+ and CD68+, confirming the LCA diagnosis. Conclusion Within red pulp spleen tumors, LCA should be highly considered as a differential diagnosis in all types of populations. In the case of a confirmed LCA, routine screening for neoplasias and autoimmune diseases should be performed.
LCA is a benign tumor with endothelial and histiocytic differentiation. LCA has a close relationship with neoplasias and autoimmune diseases. LCA is more prevalent in Asia, but it can also be seen in South America.
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