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      Multifocal sclerosing angiomatoid nodular transformation of the spleen: a case report and review of literature

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          Abstract

          Sclerosing angiomatoid nodular transformation (SANT) is a relatively new entity in the spleen, which usually presents in the form of single nodule. Only 5 multifocal SANT cases have been reported in English literature. The present case is the first report of a 38-years-old male patient with SANT in the form of multiple nodules, who has been cured via laparoscope. In comparison to solitary SANT, multifocal SANT occurs more likely in males than females and association with malignant neoplasm has not been described yet. Multifocal SANT as well as solitary SANT show some relationships with IgG4-related sclerosing disease.

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          The online version of this article (doi:10.1186/s13000-015-0312-2) contains supplementary material, which is available to authorized users.

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          Sclerosing angiomatoid nodular transformation (SANT): report of 25 cases of a distinctive benign splenic lesion.

          Juan Rosai, Raphael Chan, Luciano Lombardi … (2004)
          Twenty-five cases of a morphologically distinctive vascular lesion of the spleen are described. The patients were 17 women and 8 men, ranging in age from 22 to 74 years (mean, 48.4 years; median, 56 years). The most common presentations were incidental finding of an asymptomatic splenic mass (13 patients), abdominal pain or discomfort (6 patients), and splenomegaly (4 patients). None of the patients had evidence of recurrent disease after splenectomy. The splenic lesion was solitary, measuring 3 to 17 cm, and sharply demarcated from the surrounding parenchyma. The cut surface revealed a mass of coalescing red-brown nodules embedded in a dense fibrous stroma. All cases showed a remarkably consistent multinodular appearance at low-power examination. The individual nodules had an angiomatoid appearance, in the sense that they were composed of slit-like, round or irregular-shaped vascular spaces lined by plump endothelial cells and interspersed by a population of spindly or ovoid cells. Some of the nodules (particularly the smaller ones) were surrounded by concentric rings of collagen fibers. Numerous red blood cells were present, as well as scattered inflammatory cells. Nuclear atypia was minimal, mitotic figures were extremely rare, and necrosis was consistently absent. The internodular stroma consisted of variably myxoid to dense fibrous tissue with scattered plump myofibroblasts, plasma cells, lymphocytes, and siderophages. Immunostaining revealed 3 distinct types of vessels in the angiomatoid nodules: CD34+/CD8-/CD31+ capillaries, CD34-/CD8+/CD31+ sinusoids, and CD34-/CD8-/CD31+ small veins, recapitulating the composition of the normal splenic red pulp. These features are therefore different from those of littoral cell angioma, conventional hemangioma, and hemangioendothelioma of the spleen. We interpret these angiomatoid nodules as altered red pulp tissue that had been entrapped by a nonneoplastic stromal proliferative process. The characteristic morphologic appearance, immunophenotype, and benign clinical course suggest that this is a distinctive nonneoplastic vascular lesion of the spleen that we propose to designate as sclerosing angiomatoid nodular transformation (SANT).
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            Sclerosing Angiomatoid Nodular Transformation (SANT) of the spleen: Case report and review of the literature.

            Gavin Falk, Nishank Nooli, Gareth Morris-Stiff … (2012)
            Sclerosing Angiomatoid Nodular Transformation of the spleen (SANT) is a rare benign vascular lesion of the spleen with extensive sclerosis and unknown etiology. We report a new case of SANT of the spleen found in a 53-year-old female following detection of a splenic mass on a routine computed tomography (CT). The patient underwent an uncomplicated laparoscopic splenectomy and the specimen was sent for histopathologic examination. A review of the 97 reported cases of SANT found in the literature was undertaken. There were 43 males and 54 females with a median age of 46 years (range: 11-82 years). SANT is classically considered to be a female predominant disease, however 44.3% of reported case were male and the gender predilection may soon be neutralized as more cases are reported. 65 of the 97 (67%) patients were in 30-60 year age group. The majority of lesions (n=50) were incidentally found on imaging, and for those patients presenting with symptoms, abdominal pain (n=18) was the predominant symptom. The diagnosis of SANT should be considered in any patient presenting with a splenic lesion that contains an angiomatoid or inflammatory component. As the differential diagnosis for SANT includes malignant pathologies, and currently no reliable diagnostic radiological feature has been identified to differentiate between these conditions, SANT will continue to be diagnosed on the basis of surgical histopathology. Copyright © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved.
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              Image-guided percutaneous splenic biopsy and drainage.

              Owen O’Connor, Michael Maher, Lee Crush … (2012)
              Percutaneous splenic biopsy and drainage are relatively safe and accurate procedures. The risk of major complication (1.3%) following percutaneous splenic biopsy does not exceed that of other solid intra-abdominal organ biopsies, and it has less morbidity and mortality than splenectomy. Both computed tomography and ultrasound can be used to provide image guidance for biopsy and drainage. The safety profile of fine-needle aspiration cytology is better than core needle biopsy, but core biopsy has superior diagnostic accuracy.
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                Author and article information

                Contributors
                Jianfeng Li: 13658608819 , lijianfeng@hotmail.com
                Journal
                Journal ID (nlm-ta): Diagn Pathol
                Journal ID (iso-abbrev): Diagn Pathol
                Title: Diagnostic Pathology
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1746-1596
                Publication date (Electronic): 11 July 2015
                Publication date PMC-release: 11 July 2015
                Publication date Collection: 2015
                Volume: 10
                Electronic Location Identifier: 95
                Affiliations
                [ ]Department of Otorhinolaryngology Head and Neck Surgery, Shandong Provincial Hospital Affiliated to Shandong University, Jinan, P.R. China; Shandong Provincial Key Laboratory of Otology, NO 4, Duanxing Road (West), HuaiYin District, Jinan, P.R. China
                [ ]Department of Pathology, Shandong Provincial Hospital Affiliated to Shandong University, Jinan City, Shandong Prov China
                Article
                Publisher ID: 312
                DOI: 10.1186/s13000-015-0312-2
                PMC ID: 4498527
                PubMed ID: 26159169
                SO-VID: f5227438-9165-48de-8266-fbbf700fbd96
                Copyright © © Cao et al. 2015
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 22 March 2015
                Date accepted : 3 June 2015
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2015

                ScienceOpen disciplines: Pathology
                Keywords: sclerosing angiomatoid nodular transformation,multiple nodules,laparoscopic splenectomy,igg4-related sclerosing disease
                Data availability:
                ScienceOpen disciplines: Pathology
                Keywords: sclerosing angiomatoid nodular transformation, multiple nodules, laparoscopic splenectomy, igg4-related sclerosing disease

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