脾脏硬化性血管瘤样结节性转化临床病理特征分析并文献复习
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摘要
目的探讨脾硬化性血管瘤样结节性转化(SANT)的临床病理特征。方法分析8例SANT临床表现、病理形态学特征及免疫组化特点,并结合文献进行讨论。其中6例无特异性临床表现。男性5例,女性3例。年龄38～57岁,中位年龄48岁。大体检查:病变呈结节状,灰白色、灰红色至灰黄色,边界清楚或稍模糊。最大直径介于3～6 cm,平均4.8 cm。镜下,病变由多个血管瘤样结节构成,结节中央可见裂隙样和窦样血管腔,管壁显著增厚,结节周围可见呈向心性分布的纤维细胞或肌纤维母细胞伴淋巴细胞、浆细胞浸润。免疫组化示:病变结节内小血管CD34(+),网格状血管内皮细胞CD31(+),部分窦状腔隙细胞CD8(+)。结论 SANT比较少见,是原发于脾脏的良性血管瘤样病变,无特征性临床表现及影像学表现,确诊需手术病理,应注意与其他肿瘤及瘤样病变,尤其是恶性肿瘤的鉴别。脾切除可治愈,预后良好。
Objective To investigate the clinicopathologic features of sclerosing angiomatoid nodular transformation of the spleen(SANT). Methods Clinical manifestation, pathomorphological characteristics and immunohistochemical features of 8 cases of SANT were analyzed, and relevant literatures were reviewed. Results Six of the cases showed no specific clinical manifestation. There were 5 males and 3 females in this study, whose age ranged from 38-57 years(mean 48 years). Grossly, SANT showed a well or not circumscribed but non-encapsulated bosselated mass, with multiple individual or confluent tan white, red brown or gray yellow nodules. The mass size ranged from 3-6 cm(mean 4.8 cm). Microscopically, the mass was composed of multiple angiomatoid nodules, with crack and sinus like vascular cavity in the middle. The wall of cavity was thick. The nodules were separated by fibrous or fibrosclerotic stroma, and a scattered lympho-plasmacytic infiltrate. Immunohistochemical stain showed small blood vessel was positive for CD34, mesh-like vascular endothelial cell was positive for CD31 and splenic sinusoids were positive for CD8. Conclusion SANT is a rare benign splenic vascular lesion, with no specific clinical manifestations and imaging findings. It can be only diagnosed on the basis of surgical histopathology and should be distinguished from other tumor or tumor-like lesions, especially malignant tumor. Fortunately, it can be cured by resection and the prognosis is good.
Objective To investigate the clinicopathologic features of sclerosing angiomatoid nodular transformation of the spleen(SANT). Methods Clinical manifestation, pathomorphological characteristics and immunohistochemical features of 8 cases of SANT were analyzed, and relevant literatures were reviewed. Results Six of the cases showed no specific clinical manifestation. There were 5 males and 3 females in this study, whose age ranged from 38-57 years(mean 48 years). Grossly, SANT showed a well or not circumscribed but non-encapsulated bosselated mass, with multiple individual or confluent tan white, red brown or gray yellow nodules. The mass size ranged from 3-6 cm(mean 4.8 cm). Microscopically, the mass was composed of multiple angiomatoid nodules, with crack and sinus like vascular cavity in the middle. The wall of cavity was thick. The nodules were separated by fibrous or fibrosclerotic stroma, and a scattered lympho-plasmacytic infiltrate. Immunohistochemical stain showed small blood vessel was positive for CD34, mesh-like vascular endothelial cell was positive for CD31 and splenic sinusoids were positive for CD8. Conclusion SANT is a rare benign splenic vascular lesion, with no specific clinical manifestations and imaging findings. It can be only diagnosed on the basis of surgical histopathology and should be distinguished from other tumor or tumor-like lesions, especially malignant tumor. Fortunately, it can be cured by resection and the prognosis is good.
引文
[1] Martel M,Cheuk W,Lombardi L,et al.Sclerosing angiomatoid nodular transformation (SANT):report of 25 cases of a distinctive benign splenic lesion.American Journal of Surgical Pathology,2004,28(10):1268-1279.
[2] ?nder S,Kosemehmetoglu K,Himmetoglu ?.Firat P and Uner A:Sclerosing angiomatoid nodular transformation (SANT) of spleen:A case report describing cytology,histology,immunoprofile and differential diagnosis[J].Cytopathology,2012,23(2):129-132.
[3] Thipphavong S,Duigenan S,Schindera ST,Gee MS and Philips S:Nonneoplastic,benign,and malignant splenic diseases:Cross_sectional imaging findings and rare disease entities[J].AJR Am J Roentgenol,2014,203(2):315-322.
[4] Bamboat ZM,Masiakos PT.Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain[J].J Pediatr Surg,2010,45(7):E13-E16.
[5] Falk GA,Nooli NP,Morris-Stiff G,et al.Sclerosing Angiomatoid Nodular Transformation (SANT) of the spleen:case report and review of the literature[J].Int J Surg Case Rep,2012,3(10):492-500.
[6] Wang HL,Li KW,Wang J.Sclerosing angiomatoid nodular transformation of the spleen:report of five cases and review of literature[J].Chin Med J (Engl),2012,125(13):2386-2389.
[7] Diebold J,Le Tourneau A,Marmey B,et al:Is sclerosing angiomatoid nodular transformation (SANT) of the splenic red pulp identical to inflammatory pseu-dotumour?Report of 16 cases[J].Histopathology,2008,53(3):299-310.
[8] Lewis RB,Lattin GE Jr,Nandedkar M and Aguilera NS:Sclerosing angiomatoid nodular transformation of the spleen:CT and MRI features with pathologic correlation[J].AJR Am J Roentgenol,2013,200(4):W353-360.
[9] Karaosmanoglu DA,Karcaaltincaba M,Akata D.CT and MRI findings of sclerosing angiomatoid nodular transformation of the spleen:Spoke wheel pattern[J].Korean J Radiol 9(Suppl),2008,9(增刊):S52-S55.
[10] Raman SP,Singhi A,Horton KM,et al.Sclerosing angiomatoid nodular transformation of the spleen (SANT):Multimodality imaging appearance of five cases with radiology_pathology correlation[J].Abdom Imaging,2013,38(4):827-834.
[11] Agrawal M,Uppin SG,Bh S,et al.Sclerosing angiomatoid nodular transformation of the spleen:A new entity or a new name?[J].Turk Patoloji Derg,Apr 9 2014 (Epub ahead of print).
[12] Tian Bao Wang,Bao Guang Hu,Da Wei Liu,et al.Sclerosing angiomatoid nodular transformation of the spleen:A case report and literature review[J].ONCOLOGY LETTERS,2016,12(2):928-932.
[13] Awamleh AA,Perez-Ordonez B.Sclerosing angiomatoid nodular transformation of the spleen[J].Arch Pathol Lab Med,2007,131(6):974-978.
[14] Kim HH,Hur YH,Koh YS,et al.Sclerosing angiomatoid nodular transfor-mation of the spleen related to IgG4_associated disease:Report of a case[J].Surg Today,2013,43(8):930-936.
[15] Di Blasi A,Boscaino A.De Dominicis G and MarinoMarsilia G.Splenic hamartoma[J].Pathologica,2005,93(3):124-129,
[16] Lee H,Maeda K.Hamartoma of the spleen[J].Arch Pathol Lab Med,2009,133(1):147-151.
[17] Emir S,Sozen S,Yazar MF,et al.Arslan Solmaz O and Ozkan Z:Littoralcell angioma of the spleen[J].Arch Iran Med,2013,16(3):189-191.
[18] Lee SH.Fine-needle aspiration cytology of splenic hamartoma[J].Diagn Cytopathol,2003,28(2):82-85.
[19] Giovagnoni A,Giorgi C,Goteri G.Tumours of the spleen[J].Cancer Imaging,2005,5(1):73-77.
[20] Abbott RM,Levy AD,Aguilera NS,et al.From the archives of the AFIP:primary vascular neoplasms of the spleen:radiologicpathologic correlation[J].Radiographics,2004,24(4):1137-1163.
[21] Singh P,Mathur SK,Singh S,et al.Cystic lymphangioma of the spleen-a case report.Indian J Pathol Microbiol,2005,48(2):202-203.
[22] Delacruz V,Jorda M,Gomez_Fernandez C,et al.Fine_needle aspiration diagnosis of angiosarcoma of the spleen:A case report and review of the literature[J].Arch Pathol Lab Med,2005,129(8):1054-1056.
[2] ?nder S,Kosemehmetoglu K,Himmetoglu ?.Firat P and Uner A:Sclerosing angiomatoid nodular transformation (SANT) of spleen:A case report describing cytology,histology,immunoprofile and differential diagnosis[J].Cytopathology,2012,23(2):129-132.
[3] Thipphavong S,Duigenan S,Schindera ST,Gee MS and Philips S:Nonneoplastic,benign,and malignant splenic diseases:Cross_sectional imaging findings and rare disease entities[J].AJR Am J Roentgenol,2014,203(2):315-322.
[4] Bamboat ZM,Masiakos PT.Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain[J].J Pediatr Surg,2010,45(7):E13-E16.
[5] Falk GA,Nooli NP,Morris-Stiff G,et al.Sclerosing Angiomatoid Nodular Transformation (SANT) of the spleen:case report and review of the literature[J].Int J Surg Case Rep,2012,3(10):492-500.
[6] Wang HL,Li KW,Wang J.Sclerosing angiomatoid nodular transformation of the spleen:report of five cases and review of literature[J].Chin Med J (Engl),2012,125(13):2386-2389.
[7] Diebold J,Le Tourneau A,Marmey B,et al:Is sclerosing angiomatoid nodular transformation (SANT) of the splenic red pulp identical to inflammatory pseu-dotumour?Report of 16 cases[J].Histopathology,2008,53(3):299-310.
[8] Lewis RB,Lattin GE Jr,Nandedkar M and Aguilera NS:Sclerosing angiomatoid nodular transformation of the spleen:CT and MRI features with pathologic correlation[J].AJR Am J Roentgenol,2013,200(4):W353-360.
[9] Karaosmanoglu DA,Karcaaltincaba M,Akata D.CT and MRI findings of sclerosing angiomatoid nodular transformation of the spleen:Spoke wheel pattern[J].Korean J Radiol 9(Suppl),2008,9(增刊):S52-S55.
[10] Raman SP,Singhi A,Horton KM,et al.Sclerosing angiomatoid nodular transformation of the spleen (SANT):Multimodality imaging appearance of five cases with radiology_pathology correlation[J].Abdom Imaging,2013,38(4):827-834.
[11] Agrawal M,Uppin SG,Bh S,et al.Sclerosing angiomatoid nodular transformation of the spleen:A new entity or a new name?[J].Turk Patoloji Derg,Apr 9 2014 (Epub ahead of print).
[12] Tian Bao Wang,Bao Guang Hu,Da Wei Liu,et al.Sclerosing angiomatoid nodular transformation of the spleen:A case report and literature review[J].ONCOLOGY LETTERS,2016,12(2):928-932.
[13] Awamleh AA,Perez-Ordonez B.Sclerosing angiomatoid nodular transformation of the spleen[J].Arch Pathol Lab Med,2007,131(6):974-978.
[14] Kim HH,Hur YH,Koh YS,et al.Sclerosing angiomatoid nodular transfor-mation of the spleen related to IgG4_associated disease:Report of a case[J].Surg Today,2013,43(8):930-936.
[15] Di Blasi A,Boscaino A.De Dominicis G and MarinoMarsilia G.Splenic hamartoma[J].Pathologica,2005,93(3):124-129,
[16] Lee H,Maeda K.Hamartoma of the spleen[J].Arch Pathol Lab Med,2009,133(1):147-151.
[17] Emir S,Sozen S,Yazar MF,et al.Arslan Solmaz O and Ozkan Z:Littoralcell angioma of the spleen[J].Arch Iran Med,2013,16(3):189-191.
[18] Lee SH.Fine-needle aspiration cytology of splenic hamartoma[J].Diagn Cytopathol,2003,28(2):82-85.
[19] Giovagnoni A,Giorgi C,Goteri G.Tumours of the spleen[J].Cancer Imaging,2005,5(1):73-77.
[20] Abbott RM,Levy AD,Aguilera NS,et al.From the archives of the AFIP:primary vascular neoplasms of the spleen:radiologicpathologic correlation[J].Radiographics,2004,24(4):1137-1163.
[21] Singh P,Mathur SK,Singh S,et al.Cystic lymphangioma of the spleen-a case report.Indian J Pathol Microbiol,2005,48(2):202-203.
[22] Delacruz V,Jorda M,Gomez_Fernandez C,et al.Fine_needle aspiration diagnosis of angiosarcoma of the spleen:A case report and review of the literature[J].Arch Pathol Lab Med,2005,129(8):1054-1056.
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