Sclerosing angiomatoid nodular transformation of the spleen presenting rapid growth after adrenalectomy: Report of a case

Nagai, Yurie; Satoh, Daisuke; Matsukawa, Hiroyoshi; Shiozaki, Shigehiro

doi:10.1016/j.ijscr.2016.11.054

Cited by 10 publications

(12 citation statements)

References 16 publications

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“…The etiopathogenesis is still unknown, but several causes are hypothesized: Epstein-Barr virus association [ 16 ], red pulp abnormal transformation due to stromal proliferation, or hamartomas/inflammatory pseudoneoplasm final stage [ 6 , 17 ]. Finally, in recent studies [ 18 ], it is noted that SANT proliferation could be associated to the typical sclerosing injuries of the disorder related to immunoglobulin G4 (IgG4).…”

Section: Discussionmentioning

confidence: 99%

Sclerosing Angiomatoid Nodular Transformation: Laparoscopic Splenectomy as Therapeutic and Diagnostic Approach at the Same Time

Cipolla

Florena

Ferrara

et al. 2018

Case Reports in Surgery

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Introduction Sclerosing angiomatoid nodular transformation (SANT) of the spleen is a rare benign vascular lesion with unknown etiopathogenesis and with definite features of imaging, histopathology, and immunohistochemistry. It was first described by Martel et al. in 2004, and to date, only 151 cases have been reported. Case Description We report a case of SANT of the spleen detected in a 66-year-old Caucasian, without comorbidities, presented to our department with epigastric pain. We, also, presented a review of the literature. Conclusions SANT is a benign incidentally vascular condition in the majority of cases. The wide age and gender distribution in our review is in accordance with that in previous studies in English literature. In our opinion, splenectomy is the choice treatment because it is at the same time diagnostic and therapeutic in a definitive way.

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Section: Discussionmentioning

confidence: 99%

Sclerosing Angiomatoid Nodular Transformation: Laparoscopic Splenectomy as Therapeutic and Diagnostic Approach at the Same Time

Cipolla

Florena

Ferrara

et al. 2018

Case Reports in Surgery

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“…The curative treatment for SANT is splenectomy [ 46 , [48] , [49] , [50] , [51] , [52] , [53] , [55] , [56] , [57] , [58] , [59] , [60] ]. Open splenectomy or minimally invasive splenectomy are both options for surgical treatment [ [62] , [63] , [64] , [65] , [66] , [67] , [68] , [70] , [71] , [72] , [73] , [74] , [75] , [76] , [77] ].…”

Section: Discussionmentioning

confidence: 99%

Sclerosing angiomatoid nodular transformation presenting with thrombocytopenia after laparoscopic splenectomy - Case report and systematic review of 230 patients

Aziret

Yılmaz

Kalpakci

et al. 2020

Annals of Medicine and Surgery

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Background Sclerosing angiomatoid vascular transformation (SANT) is a rare vascular disease of the spleen, which is difficult to diagnose due to its pre-intervention appearance of malignancy. Case Report: An 85-year-old male was transferred to our clinic for thrombocytopenia and splenic mass. A contrast enhanced abdominal CT and MRI showed nodular lesions, the largest 50mm in diameter, and several areas of heterogeneous contrast field involvement in the spleen parenchyma. Laparoscopic splenectomy was performed with normal range of platelet level. The patient's postoperative course was uneventful and he was discharged on the 6th postoperative day. Histopathology revealed SANT. The patient is now in the 18 th month of remission with platelet levels within normal range and with no recurrence. Results Between 2004 and April 2020, a total of 230 SANT patients who underwent laparoscopic or open splenectomy or biopsy were reported in the literature. Most patients were female (52.1%), and the median age was 46 years (9 weeks-85 years). Most patients were asymptomatic (56%). Open splenectomy was performed on 166 patients (72.1%),laparoscopic splenectomy on 35 patients (15.2%) and laparoscopic partial splenectomy on 15 patients (6.5%). The median operation time and spleen weight were 143 minutes (88-213) and 260gr (68-2,720), respectively. Median follow-up time was 12 months (0-166). No recurrence was seen in patients undergoing total splenectomy. Conclusion SANT is an unusual disease of the spleen. In the light of this systematic review, a minimally invasive method for total or partial splenectomy,specifically laparoscopy, can be preferred as the treatment of choice.

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“…[10–11] Microscopically, some SANT lesions contain dense IgG4+ plasma cells [12–13] within fibrous stroma and erythrocyte extrasation, thus sharing features with IgG4-related sclerosing lesions. Recently, Nagai et al [14] described a rapidly growing SANT after adrenalectomy, and this phenomena may be due to the decrease in glucocorticoid concentrations, thus mimicking steroid-responsive IgG4-related sclerosing lesions. These data raise the possibility that SANT is an IgG4-related sclerosing lesion.…”

Section: Discussionmentioning

confidence: 99%

Sclerosing angiomatoid nodular transformation in the spleen

Cao¹,

Wang²,

Wang³

2019

Medicine

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To assess the clinicopathologic features, differential diagnosis, and pathogenesis of sclerosing angiomatoid nodular transformation (SANT) of the spleen.The clinical characteristics of 7 cases with SANT were retrospectively analyzed. Histochemical and immunohistochemical (EnVision method) examinations were performed. Moreover, quantitative assessment of IgG4 and IgG-positive cells was performed.The 7 SANT patients included 1 female and 6 males, with ages ranging from 7 to 60 years (mean 33.4 years). They showed no specific clinical manifestations. Macroscopically, the surface of the excised masses was gray-white, and vague nodularity was observed. Mass sizes ranged from 3.0 to 7.0 cm (mean 5.5 cm). Microscopically, all cases were characterized based on multiple angiomatoid nodules of various sizes embedded in a fibrosclerotic stroma. The nodules were round and sometimes convoluted. Moreover, the nodules were composed of slit-like, irregularly-shaped, or slightly dilated vascular spaces lined by plump endothelial cells, and interspersed with a population of spindly or ovoid cells. Immunohistochemical examination showed a heterogeneous staining pattern, with the lining cells of small capillaries expressing CD34 and those of sinusoid-like structures expressing CD8. CD31 was stained in the lining and interspersed cells, thus resulting in a complex meshwork. Additionally, the lining cells were focally positive for CD68. Vimentin and smooth muscle antibody were expressed in all 7 cases, whereas no desmin or CD21 was detected. IgG4-positive cells accounted for 2 to 5 per high-power field (mean 3.7). Furthermore, the IgG-positive cells accounted for 2 to 8 per high-power field (mean 4.2).SANT is a rare primary benign tumor-like lesion of the spleen, and has characteristic histopathological features and immunohistochemical profiles. SANT should be distinguished from other angiomatoid tumors and tumor-like lesions. Moreover, SANT could be treated by splenectomy, with favorable prognosis. The relationship between SANT and IgG4-related sclerosing lesions remains to be clarified.

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Sclerosing angiomatoid nodular transformation of the spleen presenting rapid growth after adrenalectomy: Report of a case

Abstract: HighlightsSANT displayed a rapid growth in size from 20 mm to 70 mm during 3 years after adrenalectomy.Splenectomy was performed by hand assisted laparoscopic surgery (HALS).Corticosteroids might be useful for treating SANT.

Cited by 10 publications

References 16 publications

Sclerosing Angiomatoid Nodular Transformation: Laparoscopic Splenectomy as Therapeutic and Diagnostic Approach at the Same Time

Sclerosing Angiomatoid Nodular Transformation: Laparoscopic Splenectomy as Therapeutic and Diagnostic Approach at the Same Time

Sclerosing angiomatoid nodular transformation presenting with thrombocytopenia after laparoscopic splenectomy - Case report and systematic review of 230 patients

Sclerosing angiomatoid nodular transformation in the spleen

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