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European Journal of Gynaecological Oncology  2020, Vol. 41 Issue (3): 326-331    DOI: 10.31083/j.ejgo.2020.03.5082
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Development of lymphangiosarcoma after pelvic radiation therapy for uterine cervical cancer: A case report and literature review
Hikari Unno1, Seiji Mabuchi3, Isao Arai2, Tomoko Wakasa4, Seiichi Yamamasu1()
1Department of Obstetrics and Gynecology, Hannan Chuo Hospital, 3-3-28, Minamishinmachi, Matsubara, Osaka 580-0023, Japan
2Department of Surgery, Hannan Chuo Hospital, 3-3-28, Minamishinmachi, Matsubara, Osaka 580-0023, Japan
3Department of Obstetrics and Gynecology, Osaka University Graduate School of Medicine, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan
4Department of Diagnostic Pathology, Kindai University Nara Hospital, 1248-1 Otodacho, Ikoma, Nara 630-0293, Japan
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Abstract  

Angiosarcoma accounts for 1-2% of all soft tissue sarcomas. Common sites of occurrence include the skin, breast, soft tissue, and liver. Angiosarcoma of the gastrointestinal (GI) tract is very rare. We herein present a case of a 41-year-old woman with lymphangiosarcoma of the small intestine occurring 7 years after pelvic radiotherapy for uterine cervical cancer.

Key words:  Lymphangiosarcoma      small intestine      cervical cancer      radiotherapy     
Submitted:  01 December 2018      Accepted:  14 February 2019      Published:  15 June 2020     
*Corresponding Author(s):  Seiichi Yamamasu     E-mail:  smabuchi@gyne.med.osaka-u.ac.jp

Cite this article: 

Hikari Unno, Seiji Mabuchi, Isao Arai, Tomoko Wakasa, Seiichi Yamamasu. Development of lymphangiosarcoma after pelvic radiation therapy for uterine cervical cancer: A case report and literature review. European Journal of Gynaecological Oncology, 2020, 41(3): 326-331.

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https://ejgo.imrpress.com/EN/10.31083/j.ejgo.2020.03.5082     OR     https://ejgo.imrpress.com/EN/Y2020/V41/I3/326

Figure 1.  - Resected small intestine showing perforation and tumor nodules.

Figure 2.  - Histopathological analyses of the removed small intestine. (i) Enlarged vessels observed in the submucosal tissue (Hematoxylin and eosin staining; 20× magnification). (ii) Small lumens consisted of dense atypical spindle cells around the enlarged vessels Eosinophilic fluid and erythrocytes observed in the enlarged vessels (Hematoxylin and eosin staining; 200× magnification).

Figure 3.  - Immunohistochemical analyses of the tumor. (i) Immunoreactivity for cytokeratin AE1/AE3 (200× magnification). (ii) Immunoreactivity for D2-40 (200× magnification). (iii) Immunoreactivity for CD31 (200× magnification). (iv) Immunoreactivity for vimentin (200× magnification)

Table 1  - Reported cases of post-radiation angiosarcoma arising in the small intestine.
Authors Year (Reference) Age/Sex Immunoreactivity AS/LAS Previous radiotherapy
Location Survival Treatment Irradiated tumor Radiation dose Time from RT to AS/LAS
Chen et al. 1979 [23] 66/Female NA AS: Small intestine (terminal ileum) 14 months Surgery Ovarian cancer 60 Gy 8 years
Nanus et al. 1987 [24] 42/Female NA AS: Small intestine (terminal ileum) 36 months Surgery Ovarian dysgerminoma 48 Gy 16 years
Wolov et al. 1991 [25] 80/Female Factor VIII-related antigen AS: Small and large intestines 23 days Surgery Cervical cancer 55 Gy 20 years
Wolov et al. 1991 [25] 69/Female Factor VIII-related antigen AS: Small and large intestines 3 weeks Surgery Endometrial cancer 50 Gy 7 years
Berry et al. 1991 [26] 51/Male Ulex Europaeus Agglutinin 1 Vimentin AS: Small intestine (terminal ileum) 5 months Surgery Adriamycin +dacarbazine Hodgkin’s lymphoma NA 3 years
Su et al. 1991 [27] 48/Female Factor VIII-related antigen Ulex Europaeus Agglutinin 1 AS: Small intestine (terminal ileum) 23 days Surgery Cervical cancer NA 3.2 years
Hwang et al. 1993 [28] 60/Female Ulex Europaeus Agglutinin 1 AS: Small intestine 2 months Surgery Cervical cancer 96.5 Gy 8 years
Hansen et al. 1996 [29] 76/Female Factor VIII-related antigen Vimentin AS: Small intestine 5 months Surgery Endometrial cancer 45.1 Gy 7 years
Suzuki et al. 1999 [15] 61/Female Factor VIII-related antigen Ulex Europaeus Agglutinin 1 AS: Small intestine (terminal ileum) 12 months Surgery Cisplatin Cervical cancer NA 20 years
Aitola et al. 1999 [30] 50/Female CD31 CD34 Factor VIII-related antigen AS: Small intestine (terminal ileum) 21 months Surgery Doxorubicin Endometrial cancer 55.6 Gy 14 years
Aitola et al. 1999 [30] 78/Female Factor VIII-related antigen CD31 CD34 Ulex Europaeus Agglutinin 1 AS: Small intestine (jejunum) 24 months Surgery Endometrial cancer 55.5 Gy 10 years
Selk et al. 2004 [31] 57/Male NA AS: Small intestine 4 months Surgery Chondrosarcoma of the pelvis NA 8 years
Khalil et al. 2005 [32] 68/Male CD31 CD34 Vimentin AS: Small intestine (Ileocecal) 6 months Surgery Occupational radiation exposure NA 30 years
Policarpio-Nicolas et al. 2006 [17] 51/Female CD31 CD34 Factor VIII-related antigen AS: Small intestine (terminal ileum) 10 months Surgery Cervical cancer 50 Gy 9 years
Navarro-Chagoya et al. 2015 [33] 45/Male CD31 Cytokeratin AE1/AE3 Factor VIII-related antigen AS: Small intestine (ileum and jejunum) NA Surgery Unknown pelvic tumor NA 10 years
Present case 41/Female CD31 CD34 Vimentin D2-40 LAS:Small intestine (terminal ileum) 7 months Surgery Nivolumab Cervical cancer 50 Gy 7 years
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