胃滤泡树突状细胞肉瘤：2例报道并文献复习_CHINESE JOURNAL OF BASES AND CLINICS IN GENERAL SURGERY

Authors：

杨珉珉 , 尹丽娟 ,  陈卉娇

四川大学华西医院病理科（成都 610041）;

Corresponding author：

陈卉娇, Email: jj196716@qq.com

Keywords：

滤泡树突状细胞肉瘤; 胃; 临床病理特征; 诊断; 治疗

DOI：

10.7507/1007-9424.202405050

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目的总结胃滤泡树突状细胞肉瘤（follicular dendritic cell sarcoma，FDCS）的临床病理特征。方法回顾性收集四川大学华西医院（简称“我院” ）收治的2例胃FDCS患者的临床病理资料并回顾相关文献进行分析。结果结合我院的2例及文献收集的12例共14例胃FDCS患者的资料发现，胃FDCS好发于女性（男∶女=3∶11）；发病年龄22～67岁、平均49岁、中位53岁；主要临床症状为腹痛（7/14）；胃FDCS最大径2～23 cm、中位10 cm。镜下可见肿瘤细胞多为梭形或卵圆形，呈束状、漩涡状、席纹状、车辐状、弥漫片状或模糊的结节状生长。肿瘤细胞中等大小，边界不清；胞质嗜酸性；胞核椭圆形或梭形，通常呈泡状，核仁明显。部分区域瘤细胞异型性明显，核大呈多边形，双核或多核常见，可观察到瘤巨细胞或合体样细胞存在，核分裂象易见（＞5个/10 HPF）。肿瘤内常见小淋巴细胞浸润，多聚集在血管周围，可围绕血管形成袖套状结构；肿瘤周边淋巴组织可呈透明血管Castleman病样改变。此外，肿瘤内还可见扩张的假血管样腔隙，内含嗜伊红色的蛋白样物，类似于胸腺瘤中的腔隙状结构。部分病例可见明显坏死及黏液变区域。大多数病例与周围组织分界尚清楚，文献报道中2例可见肿瘤浸润性生长。免疫组织化学染色结果：本组资料中胃FDCS细胞多数表达CD21（13/14）、CD23（11/13）、CD35（11/14），部分表达CD68（6/9）、S100（4/9），不表达CD117（0/11）和DOG1（0/8）。胃FDCS应与多种发生于胃的肿瘤相鉴别，正确诊断的关键在于认识和熟悉FDCS的组织学形态特征，在鉴别诊断中纳入滤泡树突状细胞免疫组织化学标志物。治疗多采用手术切除（13/14）。本组胃FDCS中，4例累及网膜或横结肠系膜，3例发生肝转移，5例发生淋巴结转移。有10例患者获得随访资料，随访时间1～31个月，1例因肝转移死亡，1例带瘤生存，余均为无瘤生存。其中我院的病例1术后4个月发现肝多发转移灶，进行特瑞普利单抗免疫治疗和安罗替尼靶向治疗并加用吉西他滨化疗及局部放疗，患者定期随访，目前病情稳定。结论胃FDCS好发于中年女性，临床表现常为腹痛，其组织学特征与其他部位FDCS一致，但因它较罕见，易被误诊为消化道其他肿瘤，存在诊断陷阱。应广泛取材并结合组织学特征以及免疫组织化学染色结果进行鉴别诊断，以提高对胃FDCS诊断的准确性，为患者后续治疗提供支持。

Citation： 杨珉珉, 尹丽娟, 陈卉娇. 胃滤泡树突状细胞肉瘤：2例报道并文献复习. CHINESE JOURNAL OF BASES AND CLINICS IN GENERAL SURGERY, 2024, 31(6): 743-747. doi: 10.7507/1007-9424.202405050 Copy

1.	Han JH, Kim SH, Noh SH, et al. Follicular dendritic cell sarcoma presenting as a submucosal tumor of the stomach. Arch Pathol Lab Med, 2000, 124(11): 1693-1696.
2.	Geerts A, Lagae E, Dhaene K, et al. Metastatic follicular dendritic cell sarcoma of the stomach: a case report and review of the literature. Acta Gastroenterol Belg, 2004, 67(2): 223-227.
3.	涂小予, 盛伟琪, 陆洪芬, 等. 腹腔内淋巴结外滤泡树突细胞肉瘤的临床病理学分析. 中华病理学杂志, 2007, 36(10): 660-665.
4.	王立峰, 王瑞芬, 王正彩, 等. 胃滤泡树突细胞肉瘤一例. 中华病理学杂志, 2008, 37(3): 210-211.
5.	张淑红, 周小鸽, 郑媛媛, 等. 滤泡树突状细胞肉瘤的临床病理观察. 中华肿瘤杂志, 2010, 32(2): 123-127.
6.	Shaw D, Cuison R, Ito H. Follicular dendritic cell sarcoma of the stomach: case report and review of the literature. Curr Oncol, 2014, 21(6): e775-e778. doi: 10.3747/co.21.2091.
7.	Hassan U, Rana IA, Mushtaq S, et al. Follicular dendritic cell sarcoma of gastrointestinal tract: an uncommon lesion, commonly missed. J Gastrointest Cancer, 2019, 50(4): 913-918.
8.	杨高亮, 刘静, 刘繁荣, 等. 发生于胃及腹腔的滤泡树突状细胞肉瘤1例并文献复习. 临床与实验病理学杂志, 2020, 36(5): 566-569.
9.	Wang RF, Han W, Qi L, et al. Extranodal follicular dendritic cell sarcoma: A clinicopathological report of four cases and a literature review. Oncol Lett, 2015, 9(1): 391-398.
10.	Sassi F, Sahraoui G, Charfi L, et al. An incidental discovery of a gastric follicular dendritic cell sarcoma: A rare case report and a literature review. Rare Tumors, 2023, 15: 1-6.
11.	Shacklette AH, Chen S, DeWitt JM, et al. Fine needle aspiration cytology of follicular dendritic cell sarcoma of the stomach masquerading as gastrointestinal stromal tumor: A case report of a unique entity with emphasis on cytomorphology. Diagn Cytopathol, 2023, 51(7): E214-E218. doi: 10.1002/dc.25132.
12.	Zhan T, Xing S, Lu C. Follicular dendritic cell sarcoma arising in the stomach and abdominal cavity: A case report. Medicine (Baltimore), 2023, 102(31): 1-4.
13.	Monda L, Warnke R, Rosai J. A primary lymph node malignancy with features suggestive of dendritic reticulum cell differentiation. A report of 4 cases. Am J Pathol, 1986, 122(3): 562-572.
14.	Chen T, Gopal P. Follicular dendritic cell sarcoma. Arch Pathol Lab Med, 2017, 141(4): 596-599.
15.	Saygin C, Uzunaslan D, Ozguroglu M, et al. Dendritic cell sarcoma: a pooled analysis including 462 cases with presentation of our case series. Crit Rev Oncol Hematol, 2013, 88(2): 253-271.
16.	Chan JKC, Pileri SA. Follicular dendritic cell sarcoma. The WHO classification of digestive system tumors. 5th ed. Lyon: IAPC Press, 2019.
17.	Andersen EF, Paxton CN, O’Malley DP, et al. Genomic analysis of follicular dendritic cell sarcoma by molecular inversion probe array reveals tumor suppressor-driven biology. Mod Pathol, 2017, 30(9): 1321-1334.
18.	Facchetti F, Simbeni M, Lorenzi L. Follicular dendritic cell sarcoma. Pathologica, 2021, 113(5): 316-329.
19.	Ke X, He H, Zhang Q, et al. Epstein-Barr virus-positive inflammatory follicular dendritic cell sarcoma presenting as a solitary colonic mass: two rare cases and a literature review. Histopathology, 2020, 77(5): 832-840.
20.	Bruehl F K, Azzato E, Durkin L, et al. Inflammatory pseudotumor-like follicular/fibroblastic dendritic cell sarcomas of the spleen are EBV-associated and lack other commonly identifiable molecular alterations. Int J Surg Pathol, 2021, 29(4): 443-446.
21.	Go H, Jeon YK, Huh J, et al. Frequent detection of BRAF(V600E) mutations in histiocytic and dendritic cell neoplasms. Histopathology, 2014, 65(2): 261-272.
22.	Shia J, Chen W, Tang LH, et al. Extranodal follicular dendritic cell sarcoma: clinical, pathologic, and histogenetic characteristics of an underrecognized disease entity. Virchows Arch, 2006, 449(2): 148-158.
23.	Youens KE, Waugh MS. Extranodal follicular dendritic cell sarcoma. Arch Pathol Lab Med, 2008, 132(10): 1683-1687.
24.	Chen S, Sun Y, Sun W, et al. Survival analysis in patients with follicular dendritic cell sarcoma: a population-based study. Hematology, 2023, 28(1): 1-8.
25.	Li L, Shi YH, Guo ZJ, et al. Clinicopathological features and prognosis assessment of extranodal follicular dendritic cell sarcoma. World J Gastroenterol, 2010, 16(20): 2504-2519.
26.	Shinagare AB, Ramaiya NH, Jagannathan JP, et al. Primary follicular dendritic cell sarcoma of liver treated with cyclophosphamide, doxorubicin, vincristine, and prednisone regimen and surgery. J Clin Oncol, 2011, 29(35): e849-e851. doi: 10.1200/JCO.2011.37.1906.
27.	Facchetti F, Lorenzi L. Follicular dendritic cells and related sarcoma. Semin Diagn Pathol, 2016, 33(5): 262-276.
28.	Mita MM, Poplin E, Britten CD, et al. Phase Ⅰ/Ⅱa trial of the mammalian target of rapamycin inhibitor ridaforolimus (AP23573; MK-8669) administered orally in patients with refractory or advanced malignancies and sarcoma. Ann Oncol, 2013, 24(4): 1104-1111.
29.	Sun X, Chang K C, Abruzzo L V, et al. Epidermal growth factor receptor expression in follicular dendritic cells: a shared feature of follicular dendritic cell sarcoma and Castleman’s disease. Hum Pathol, 2003, 34(9): 835-840.

1. Han JH, Kim SH, Noh SH, et al. Follicular dendritic cell sarcoma presenting as a submucosal tumor of the stomach. Arch Pathol Lab Med, 2000, 124(11): 1693-1696.
2. Geerts A, Lagae E, Dhaene K, et al. Metastatic follicular dendritic cell sarcoma of the stomach: a case report and review of the literature. Acta Gastroenterol Belg, 2004, 67(2): 223-227.
3. 涂小予, 盛伟琪, 陆洪芬, 等. 腹腔内淋巴结外滤泡树突细胞肉瘤的临床病理学分析. 中华病理学杂志, 2007, 36(10): 660-665.
4. 王立峰, 王瑞芬, 王正彩, 等. 胃滤泡树突细胞肉瘤一例. 中华病理学杂志, 2008, 37(3): 210-211.
5. 张淑红, 周小鸽, 郑媛媛, 等. 滤泡树突状细胞肉瘤的临床病理观察. 中华肿瘤杂志, 2010, 32(2): 123-127.
6. Shaw D, Cuison R, Ito H. Follicular dendritic cell sarcoma of the stomach: case report and review of the literature. Curr Oncol, 2014, 21(6): e775-e778. doi: 10.3747/co.21.2091.
7. Hassan U, Rana IA, Mushtaq S, et al. Follicular dendritic cell sarcoma of gastrointestinal tract: an uncommon lesion, commonly missed. J Gastrointest Cancer, 2019, 50(4): 913-918.
8. 杨高亮, 刘静, 刘繁荣, 等. 发生于胃及腹腔的滤泡树突状细胞肉瘤1例并文献复习. 临床与实验病理学杂志, 2020, 36(5): 566-569.
9. Wang RF, Han W, Qi L, et al. Extranodal follicular dendritic cell sarcoma: A clinicopathological report of four cases and a literature review. Oncol Lett, 2015, 9(1): 391-398.
10. Sassi F, Sahraoui G, Charfi L, et al. An incidental discovery of a gastric follicular dendritic cell sarcoma: A rare case report and a literature review. Rare Tumors, 2023, 15: 1-6.
11. Shacklette AH, Chen S, DeWitt JM, et al. Fine needle aspiration cytology of follicular dendritic cell sarcoma of the stomach masquerading as gastrointestinal stromal tumor: A case report of a unique entity with emphasis on cytomorphology. Diagn Cytopathol, 2023, 51(7): E214-E218. doi: 10.1002/dc.25132.
12. Zhan T, Xing S, Lu C. Follicular dendritic cell sarcoma arising in the stomach and abdominal cavity: A case report. Medicine (Baltimore), 2023, 102(31): 1-4.
13. Monda L, Warnke R, Rosai J. A primary lymph node malignancy with features suggestive of dendritic reticulum cell differentiation. A report of 4 cases. Am J Pathol, 1986, 122(3): 562-572.
14. Chen T, Gopal P. Follicular dendritic cell sarcoma. Arch Pathol Lab Med, 2017, 141(4): 596-599.
15. Saygin C, Uzunaslan D, Ozguroglu M, et al. Dendritic cell sarcoma: a pooled analysis including 462 cases with presentation of our case series. Crit Rev Oncol Hematol, 2013, 88(2): 253-271.
16. Chan JKC, Pileri SA. Follicular dendritic cell sarcoma. The WHO classification of digestive system tumors. 5th ed. Lyon: IAPC Press, 2019.
17. Andersen EF, Paxton CN, O’Malley DP, et al. Genomic analysis of follicular dendritic cell sarcoma by molecular inversion probe array reveals tumor suppressor-driven biology. Mod Pathol, 2017, 30(9): 1321-1334.
18. Facchetti F, Simbeni M, Lorenzi L. Follicular dendritic cell sarcoma. Pathologica, 2021, 113(5): 316-329.
19. Ke X, He H, Zhang Q, et al. Epstein-Barr virus-positive inflammatory follicular dendritic cell sarcoma presenting as a solitary colonic mass: two rare cases and a literature review. Histopathology, 2020, 77(5): 832-840.
20. Bruehl F K, Azzato E, Durkin L, et al. Inflammatory pseudotumor-like follicular/fibroblastic dendritic cell sarcomas of the spleen are EBV-associated and lack other commonly identifiable molecular alterations. Int J Surg Pathol, 2021, 29(4): 443-446.
21. Go H, Jeon YK, Huh J, et al. Frequent detection of BRAF(V600E) mutations in histiocytic and dendritic cell neoplasms. Histopathology, 2014, 65(2): 261-272.
22. Shia J, Chen W, Tang LH, et al. Extranodal follicular dendritic cell sarcoma: clinical, pathologic, and histogenetic characteristics of an underrecognized disease entity. Virchows Arch, 2006, 449(2): 148-158.
23. Youens KE, Waugh MS. Extranodal follicular dendritic cell sarcoma. Arch Pathol Lab Med, 2008, 132(10): 1683-1687.
24. Chen S, Sun Y, Sun W, et al. Survival analysis in patients with follicular dendritic cell sarcoma: a population-based study. Hematology, 2023, 28(1): 1-8.
25. Li L, Shi YH, Guo ZJ, et al. Clinicopathological features and prognosis assessment of extranodal follicular dendritic cell sarcoma. World J Gastroenterol, 2010, 16(20): 2504-2519.
26. Shinagare AB, Ramaiya NH, Jagannathan JP, et al. Primary follicular dendritic cell sarcoma of liver treated with cyclophosphamide, doxorubicin, vincristine, and prednisone regimen and surgery. J Clin Oncol, 2011, 29(35): e849-e851. doi: 10.1200/JCO.2011.37.1906.
27. Facchetti F, Lorenzi L. Follicular dendritic cells and related sarcoma. Semin Diagn Pathol, 2016, 33(5): 262-276.
28. Mita MM, Poplin E, Britten CD, et al. Phase Ⅰ/Ⅱa trial of the mammalian target of rapamycin inhibitor ridaforolimus (AP23573; MK-8669) administered orally in patients with refractory or advanced malignancies and sarcoma. Ann Oncol, 2013, 24(4): 1104-1111.
29. Sun X, Chang K C, Abruzzo L V, et al. Epidermal growth factor receptor expression in follicular dendritic cells: a shared feature of follicular dendritic cell sarcoma and Castleman’s disease. Hum Pathol, 2003, 34(9): 835-840.

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