Journal of Pathology and Translational Medicine

Original Article

A clinicopathologic and immunohistochemical study of primary and secondary breast angiosarcoma: Evi Abada, Hyejeong Jang, Seongho Kim, Rouba Ali-Fehmi, Sudeshna Bandyopadhyay; J Pathol Transl Med. 2022;56(6):342-353. Published online October 27, 2022; DOI: https://doi.org/10.4132/jptm.2022.08.31

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Background
We aimed to study the clinicopathologic and immunohistochemical (IHC) (CD117, c-Myc, and p53) characteristics, and overall survival of primary and secondary breast angiosarcoma (BAS).
Methods
This was a retrospective study of BAS cases diagnosed between 1997 and 2020 at our institution. Hematoxylin and eosin-stained slides were reviewed for tumor morphology, margin status, and lymph node metastasis. CD117, p53, D2-40, CD31, and c-Myc IHC stains were performed on 11 viable tissue blocks. Additional clinical information was obtained from the electronic medical records.
Results
Seventeen patients with BAS were identified. Of these, five (29%) were primary and 12 (71%) were secondary BAS, respectively. The median age at diagnosis for primary BAS was 36 years. The median age at diagnosis for secondary BAS was 67 years. The median time to secondary BAS development following radiotherapy was 6.5 years (range, 2 to 12 years). There was no significant difference between primary and secondary BAS in several histopathologic parameters examined, including histologic grade, necrosis, mitotic count, lymph node metastasis, and positive tumor margins. There was also no difference in CD117, p53, D2-40, CD31, and c-Myc expression by IHC between primary and secondary BAS. During a median followup of 21 months, primary BAS had two (40%) reported deaths and secondary BAS had three (25%) reported deaths. However, this difference in survival between both groups was not statistically significant (hazard ratio, 0.51; 95% confidence interval, 0.09 to 3.28; p = .450).
Conclusions
BAS is a rare and aggressive disease. No histologic, IHC (CD117, c-Myc, and p53), or survival differences were identified between primary and secondary BAS in this study.

Citations

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Etiology, pathogenesis, and management of angiosarcoma associated with implants and foreign body: Clinical cases and research updates
Ramy Samargandi
Medicine.2024; 103(18): e37932. CrossRef
Ovarian angiosarcoma: A systematic review of literature and survival analysis
Shafi Rehman, Arya Harikrishna, Amisha Silwal, B.R. Sumie, Safdar Mohamed, Nisha Kolhe, Meghana Maddi, Linh Huynh, Jesus Gutierrez, Yoshita Rao Annepu, Ameer Mustafa Farrukh
Annals of Diagnostic Pathology.2024; 73: 152331. CrossRef
Neoadjuvant chemotherapy for radiation associated angiosarcoma (RAAS) of the breast: A retrospective single center study
Stijn J.C. van der Burg, Sophie J.M. Reijers, Anke Kuijpers, Lotte Heimans, Astrid N. Scholten, Rick L.M. Haas, Hester van Boven, Willemijn M. Kolff, Marie-Jeanne T.F.D. Vrancken Peeters, Martijn Kerst, Beatrijs A. Seinstra, Neeltje Steeghs, Winette T.A.
The Breast.2024; 78: 103825. CrossRef
Primary ovarian angiosarcoma: Two case reports and review of literature
Ying Zhou, Yi-Wen Sun, Xiao-Yang Liu, Dan-Hua Shen
World Journal of Clinical Cases.2023; 11(21): 5122. CrossRef

Case Study

A Case of Metastatic Angiosarcoma Diagnosed by Liquid-Based Preparation: Peculiar Cytoplasmic Changes: Min Jung Jung, Young Ok Kim; Korean J Pathol. 2014;48(3):241-247. Published online June 26, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.3.241

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Abstract PDF

Angiosarcoma with predominantly epithelioid features is a rare soft tissue neoplasm and the interpretation of its cytopathologic findings may be difficult. We report a case of metastatic angiosarcoma with predominantly epithelioid features diagnosed by liquid-based cytology. The cytopathologic findings in this case differed from those of the conventional preparation and we found a clean background, no hyperchromatic nuclei and several cytoplasmic changes, including intracytoplasmic vacuoles with peculiar shapes, juxtanuclear condensation and perinuclear clearing. Identification of these changes using liquid-based cytology supplemented with immunochemistry may be helpful in reaching a correct cytopathologic diagnosis.

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Cytological Features of a Metastatic Angiosarcoma in the Lymph Node Diagnosed via Liquid-Based Cytology
Jie-Yang Jhuang, Chih-Yi Liu, Min-Hui Tseng, Shih-Sung Chuang
Diagnostics.2023; 13(12): 2124. CrossRef
Radiation-associated Angiosarcoma Presenting as Massive Pleural Effusion
Hirokazu Ogino, Makoto Tobiume, Kozo Kagawa, Hiroshi Kawano, Satoshi Sakaguchi, Atsuro Saijo, Daisuke Matsumoto, Hiromitsu Takizawa, Yuriko Morikawa, Yoshimi Bando, Hisatsugu Goto, Hiroshi Nokihara, Yasuhiko Nishioka
Internal Medicine.2022; 61(9): 1393. CrossRef
Delayed diagnosis of angiosarcoma of the spleen: clinically presenting as recurrent haemoperitoneum following embolisation
Verena Kornmann, Philip van Rijn, Dries Mulder, Koen Reijnders
BMJ Case Reports.2015; : bcr2014208956. CrossRef
A Case of Angiosarcoma of the Scalp with Invasion to the Pleural Effusion
Yusuke Amano, Yukari Obana, Yoko Nakanishi, Ryusuke Tsujimura, Kayomi Wakamatsu, Fumiko Uemura, Yoshihisa Katsura, Masahiko Sugitani, Norimichi Nemoto
Journal of Nihon University Medical Association.2015; 74(3): 113. CrossRef

Case Reports

Fine Needle Aspiration Cytologic Findings of Angiosarcoma: Report of Two Cases.: Jin Xian Ji, Young Chae Chu, Lucia Kim, Suk Jin Choi, In Suh Park, Jee Young Han, Joon Mee Kim, Kyu Ho Kim, Ju Young Song; Korean J Pathol. 2011;45(2):217-222.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.2.217

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Abstract PDF

Angiosarcoma is a rare malignant vascular neoplasm which can arise in any part of the body. Specific recognition of this neoplasm in cytological specimens is difficult in the absence of an ancillary method. Herein, we present the cytologic findings of two cases of angiosarcomas diagnosed on fine needle aspiration cytology. One case is a recurred angiosarcoma in the left chest wall and the other case is a lymphedema-associated angiosarcoma in the left lower leg. The cytologic findings of both cases are similar. Cytologic features that identified this neoplasm as an angiosarcoma included arborizing microtissue fragments, irregular anastomosing vascular spaces lined by atypical cells, microacini, intracytoplasmic lumen, and intracellular red blood cells, marked cell discohesiveness, spindle to ovoid, irregular, hyperchromatic nuclei, and elongated cytoplasmic processes with indistinct borders. This report emphasizes that when aspiration smears show vasoformative features in a bloody background, angiosarcoma should be included in the differential diagnosis.

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Four newly reported ophichthid leptocephali species revealed by mitochondrial 12S rDNA, with implications of their occurrence in Korea
Hwan Sung Ji, Hae Won Lee, Byung Kyu Hong, Jin Koo Kim
Animal Cells and Systems.2012; 16(5): 415. CrossRef

A Case of Ovarian Microinvasive Mucinous Carcinoma and Co-existent Angiosarcoma.: Jin Hyung Heo, Yoon Hee Lee, Gwang Il Kim, Tae Heon Kim, Haeyoun Kang, Hee Jung An, Bo Sung Yoon, Seok Ju Seong, Hyun Park, Ji Young Kim; Korean J Pathol. 2011;45(1):96-100.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.1.96

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Abstract PDF

Primary ovarian angiosarcoma is very rare with only 27 cases reported so far in the medical literature. We report here on a rare case of ovarian microinvasive mucinous carcinoma that was coexistent with angiosarcoma in a 54-year-old woman. The tumor was a 26x19x10 cm-sized multilocular cystic mass with a 4x3 cm-sized solid hematoma-like nodule in the center. Microscopically, it was composed mostly of mucinous tumor of various grades from borderline to microinvasive carcinoma. The hematoma-like area turned out to be an angiosarcoma, composed of pleomorphic cells that formed slit-like spaces, spindle cells that formed short fascicles and anastomosing vascular channels with atypical endothelial cells. All these cells were positive for CD31, CD34 and factor VIII-related antigen. The patient developed peritoneal and pleural metastases, which were angiosarcoma and mucinous carcinoma, respectively. We believe this case is only the fourth example of an ovarian collision tumor of angiosarcoma and surface epithelial tumor.

Citations

Citations to this article as recorded by

Ovarian angiosarcoma: A systematic review of literature and survival analysis
Shafi Rehman, Arya Harikrishna, Amisha Silwal, B.R. Sumie, Safdar Mohamed, Nisha Kolhe, Meghana Maddi, Linh Huynh, Jesus Gutierrez, Yoshita Rao Annepu, Ameer Mustafa Farrukh
Annals of Diagnostic Pathology.2024; 73: 152331. CrossRef
Tumor to Tumor Metastasis: A Case Report of Metastatic Angiosarcoma to an Ovarian Brenner Tumor and Review of the Literature
Bilge Dundar, Audai Alrwashdeh, Laila Dahmoush
International Journal of Gynecological Pathology.2023; 42(2): 176. CrossRef
Collision Tumors in Ovary: Case Series and Literature Review
Borges A, Loddo A, Martins A, Peiretti M, Fanni D, Djokovic D
Journal of Surgical Oncology.2019; : 1. CrossRef
Angiosarcoma Arising in Ovarian Mucinous Tumor: A Challenge in Intraoperative Frozen Section Diagnosis
Surapan Khunamornpong, Jongkolnee Settakorn, Kornkanok Sukpan, Tip Pongsuvareeyakul, Sumalee Siriaunkgul
Case Reports in Pathology.2016; 2016: 1. CrossRef
Impact of body burden of pesticide residues on the reproductive tract of buffalo
KARANPREET KAUR, SARVPREET SINGH GHUMAN, OPINDER SINGH, JASBIR SINGH BEDI, JATINDER PAUL SINGH GILL
The Indian Journal of Animal Sciences.2016;[Epub] CrossRef

The Cytology of Metastatic Angiosarcoma in Pleural Fluid : A Case Report.: Na Rae Kim, Dong Hae Chung, Hyun Yee Cho; Korean J Pathol. 2009;43(3):285-259.; DOI: https://doi.org/10.4132/KoreanJPathol.2009.43.3.285

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Abstract PDF

A 74-year-old woman presented with an abrupt onset of dyspnea that she had experienced for a week. She had been suffering from cutaneous nodules in the scalp for a year. Thoracentesis of the pleural fluid was performed. The Papanicolaou-stained smears, Thin prep and cell block preparations revealed clusters of oval-shaped cells concentrically layered about amorphous acellular cores, i.e., there was microacinar lumen formation as well as singly scattered atypical cells. The cells occasionally demonstrated intracytoplasmic vacuoles and hemosiderin deposits. Those cells stained for CD31 and they were negative for pancytokeratin. Punch biopsy from the scalp nodules revealed angiosarcoma. There are currently few reported cases of angiosarcoma in an exfoliative pleural effusion. Angiosarcoma has diverse, heterogeneous cytologic features. Making the cytologic diagnosis of metastatic angiosarcoma in pleural fluid is a challenge for pathologists. Knowledge of the clinical history is of great help for diagnosing this tumor when it appears in rare sites. Immunopanels with CD31, pancytokeratin and TTF-1 are helpful for making the differential diagnosis. The pathologists should look for clues suggesting the presence of vascular differentiation in the exfoliative cytologic materials when a diagnosis of angiosarcoma is suspected.

Citations

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Metastatic pleural angiosarcoma: A diagnostic pitfall might be overcome by morphologic clues and clinical correlation
Ok Ran Shin, Uiju Cho, Eundeok Chang, Kyung Jin Seo
Diagnostic Cytopathology.2015; 43(8): 669. CrossRef
Pleural fluid cytology in metastatic uterine angiosarcoma—A case report—
Shiho KURODA, Shioto SUZUKI, Akira KURITA, Mari MURAKI, Fumihiko TANIOKA
The Journal of the Japanese Society of Clinical Cytology.2014; 53(6): 498. CrossRef
Intranasal delivery of biologics to the central nervous system
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Fine Needle Aspiration Cytologic Findings of Angiosarcoma - Report of Two Cases -
Jin Xian Ji, Young Chae Chu, Lucia Kim, Suk Jin Choi, In Suh Park, Jee Young Han, Joon Mee Kim, Kyu Ho Kim, Ju Young Song
The Korean Journal of Pathology.2011; 45(2): 217. CrossRef

Original Article

Intra-abdominal Angiosarcomatosis Induced by Irradiation.: Hee Jin Chang, Youn Joo Kim, Sung Eun Yang, Sung Sook Paeng, Jung Il Suh; Korean J Pathol. 1997;31(3):269-274.

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Abstract PDF: Therapeutic irradiation can induce angiosarcoma. Radiation-induced angiosarcoma constitutes 20% of all angiosarcomas. Although its common site of origin is the skin and subcutaneous tissue, it rarely arises in small or large bowels with a presentation as multifocal abdominal angiosarcomatosis. We report a case of intra-abdominal angiosarcomatosis involving the jejunum, ileum, transverse colon, mesentery and right ovary in a 63-year-old female. It developed 10 years after therapeutic irradiation for squamous cell carcinoma of uterine cervix. She developed panperitonitis due to intestinal perforation. She died from sepsis 3 days after segmental resection of the small bowel and right oophorectomy. We reviewed the previously reported cases and describe the clinicopathologic features of this tumor.

Case Reports

Malignant Glomus Tumor Arising in Benign Glomus Tumor.: Seung Sam Paik, Eun Sun Kim, Young Chun Moon, Chan Kum Park, Moon Hyang Park, Jung Dal Lee; Korean J Pathol. 1997;31(3):280-283.

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Abstract PDF: The glomus tumor is a distinct neoplasm composed of modified perivascular smooth-muscle cells identical to those described in the glomus body. Malignant glomus tumor, also referred to as glomangiosarcoma, is exceedingly rare. It has been subdivided into locally infiltrative glomus tumor, glomangiosarcoma arising in a benign glomus tumor, and glomangiosarcoma arising in de novo. A few cases of malignant glomus tumor arising in a benign glomus tumor have been reported. A 55-year-old man underwent resection of a solitary nodule in the right shoulder area. The tumor was biphasic with a typical benign glomus tumor at the periphery and the central sarcomatous area composed of oval to elongated pleomorphic cells with a single prominent nucleolus and occasional mitotic figures. Both tumor components showed intense staining for vimentin, smooth muscle actin, and muscle specific actin. Herein, we report a case of a malignant glomus tumor arising in a benign glomus tumor with an immunohistochemical study and a review of literatures.

Angiosarcoma of the Uterus: A case report.: Young Kyung Bae, Joon Hyuk Choi, Mi Jin Kim; Korean J Pathol. 1999;33(7):521-524.

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Abstract PDF: Primary angiosarcoma of the female reproductive system is extremely rare, and its management is not well understood. We experienced a very rare case of angiosarcoma arising in the uterus of a 37-year-old woman who presented abdominal distension. At laparotomy, the uterus was observed to be enlarged and deformed by multilobulated hemorrhagic mass. Tumor deposits were present on both ovaries and the omentum. The operation consisted of total abdominal hysterectomy with salpingo-oophorectomy, and omentectomy. Microscopic examination showed that the tumor consisted mostly of solid sheets of pleomorphic and spindle cells forming irregular, communicating cleft-like spaces. Well-formed vascular channels lined by atypical endothelial cells were seen in differentiated areas. Some vessels were partially lined by normal endothelial cells and partially by neoplastic endothelial cells. Papillary architecture composed of epithelioid tumor cells was noted in a focal area. The tumor cells were immunoreactive for factor VIII-related antigen, Ulex europaeus agglutinin-1, CD31 and CD34. Weibel-Palade bodies were not seen in the tumor cells. She received adjuvant chemotherapy, but died 13 months after the diagnosis.

Emphysematous Cystic Pulmonary Metastasis of Angiosarcoma.: Tae Heon Kim, Ji Sun Song, Soon Hee Jung, Ki Jun Sung, Eun Gi Kim; Korean J Pathol. 2001;35(6):544-546.

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Abstract: Pulmonary metastatic angiosarcoma usually reveals multiple nodular lesions associated with parenchymal hemorrhage. It is presented, in rare cases, as multiple emphysematous cystic lesions, complicated by pneumothorax. We experienced a case of pulmonary metastastasis from angiosarcoma of the scalp in a 58-year-old male showing multiple thin-walled emphysematous cystic lesions. The tumor cells spread along the subpleural and interlobular septa, focally surrounding the cystic lesions. This case suggests that a rare metastatic pattern seen in angiosarcoma of the scalp should be considered as a differential diagnosis of bullous emphysematous lesion.

Fine Needle Aspiration Cytology of Angiosarcoma of the Rib: A Case Report.: Hee Jung Kim, Mee Yon Cho, Soon Hee Jung, Kwang Gil Lee; Korean J Cytopathol. 1996;7(2):207-212.

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Abstract PDF: Angiosarcoma of the bone is rare with an incidence of 0.13%. It may be solitary or multiple. Its cytologic findings by FNA have rarely been reported. We report a case of angiosarcoma of the rib in a 66 year-old man. FNA revealed single or clusters of round to oval shaped cells in a hemorrhagic and myxoid background. The large central nuclei had irregular nuclear membrane, chromatin clumping and prominent nucleoli. The cytoplasm was scanty with an eosinophilic distinct cytoplasmic border. Erythrophagocytosis by malignant cells was also found. Histopathologic examination confirmed the diagnosis of angiosarcoma revealing irregular and com- plex anastomosing vascular channels lined by malignant round tumor cells, protruding into the lumen. Immunohistochemical staining revealed diffuse strong positive reaction to factor VIII-related antigen and CD31.

Cytologic Features of an Angiosarcoma in Pleural Fluid: A Case Report .: Young Chae Chu, In Seo Park, Yoon Ju Kim, Hye Seung Han, Jee Young Han; Korean J Cytopathol. 1999;10(1):61-66.

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Abstract PDF: Angiosarcomas are rare tumors, seen most commonly in the skin and soft tissue of head and neck region. But it has been described in numerous body sites including thyroid, breast, liver, spleen, bone, etc.. Their biological behaviors depend on the microscopic grade, site of origin, and multifocality. We report the unique cytological features of an angiosarcoma in pleural fluid. A 61-year-old woman presented with a 6 month history of dyspnea on exertion and chest pain. Chest computerized tomography(CT) revealed multiple subpleural small nodules in the right lung and widespread air space consolidation and pleural effusion in the left lung. CT of liver revealed multiple small low attenuated lesion. The smears obtained from pleural fluid showed hypocellularity with a hemorrhagic background. The tumor cells were highly pleomorphic oval or spindle in shape and presented singly, in loose groups, in knitted syncytial aggregates, and in acinar pattern. Their nuclei had vesicular chromatin with delineated, thick nuclear membranes and occasionally a large eosinophilic, prominent nucleolus. The cytoplasm was plump, thin or projected in spindly fashion. Almost all tumor cells showed variable sized intracytoplasmic vacuoles and their nuclei were sometimes crescentic by a huge vacuole. Occasional binucleated tumor cells and mitotic figures were present. Cellular debris and streaky materials were identified. Needle biopsy specimen from the pleura revealed anastomosing slit-like spaces lined by pleomorphic tumor cells. The tumor cells showed a strong reactivity for CD31 and vimentin and focal weak reactivity for factor VIII-related antigen.

Composite Epithelioid Hemangioendothelioma in Pleural Effusion Mimicking Metastatic Adenocarcinoma: Cytologic and Immunocytochemical Findings.: Ki Seok Jang, Hong Xiu Han, Moon Hyang Park; Korean J Cytopathol. 2003;14(1):36-41.

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Abstract PDF: Epithelioid hemangioendothelioma is a rare vascular tumor of borderline malignancy which is characterized by the presence of "epithelioid" or "histiocytoid" endothelial cells. Superficial and deep tumors have been recognized in the extremities, head, neck, chest, and mediastinum of adult patients. It may also occur as a primary tumor of liver, bone, and other visceral organs. Few effusion cytologic findings of epithelioid hemangioendothelioma have been reported. We report a case of composite epithelioid hemangioendothelioma with focal epithelioid angiosarcomatous areas of the iliac bone and adjacent soft tissue in a 38-year-old female, which, during its metastatic course, was presented as a pleural effusion. The effusion was cellular with epithelioid cells presenting both singly and in clusters. The tumor cells were round to ovoid showing cytoplasmic vacuolization, variability in cell size, and prominent nucleoli. The effusion smears and cell block sections revealed strong positive staining for CD31 and vimentin, weak positive for CD34 and Factor VIII-related antigen, and negative for cytokeratin, CEA, and calretinin. The cytologic findings in this case were similar to that of metastatic adenocarcinoma or malignant mesothelioma. Therefore, immunocytochemical staining in smear and cell block is a helpful tool to differentiate malignant "epithelioid" cells in effusion.

Multifocal Epithelioid Angiosarcoma of the Stomach: A Case Report.: Kyeongmee Park, Sungjig Lim, Jungyeon Kim, Young Jin Yuh, Sung Rok Kim, Hong Yong Kim; Korean J Pathol. 2003;37(6):435-437.

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Abstract PDF: Angiosarcoma is a very rare neoplasm, and even more so in the gastrointestinal tract where its distinction from adenocarcinoma may be extremely difficult. We report a case of multifocal epithelioid angiosarcoma of the stomach in a 65-year-old woman. Histologically, the tumor foci were composed of haphazard, anastomosing channels lined by malignant endothelial cells with epithelioid features. Those neoplastic cells stained positive for CD31, CD34, and factor VIII-related antigen.

Primary Cardiac Angiosarcoma: A case report and rewiew of literture.: Mee Yon Cho, Soon Hee Jung, Woo Ick Yang, Kyung Hoon Choe, Chong Kook Lee; Korean J Pathol. 1993;27(4):397-401.

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Abstract PDF: We report a case of primary cardiac angiosarcoma in a 35 year-old woman. She presented with dyspnea, facial edema and neck vein distension during 1 month. The oval round large mass(8x4 cm) filling the right atrium infiltrated into the entire thickness of the lateral wall and extended to the vena cava and ventricle. The interatrial septum and pericardium seemed to be intact. Despite the surgical excision and adjuvant chemo-and radiotherapy, the patient died 8 months later due to tumor recurrence. The histologic findings of tumor varied from benign looking capillary proliferation mimicking granulation tissue to sarcoma composed of spindle cells. But anastomosing vascular channels lined by anaplastic polygonal cells and intracytoplasmic lumen containing red blood cells were characteristic findings. These tumor cells showed positive reaction to endothelial cell antigen and factor VIII-related antigen. The intercellular junctional complex and vascular channels formed by cytoplasmic process of tumor cells were identified by the ultrastructural study.

Primary Angiosarcoma of the Spleen associated with Kasabach-Merritt Syndrome: Report of a case.: Mee Soo Chang, Yun Kyung Kang, Yong Il Kim, Kun Wook Lee; Korean J Pathol. 1992;26(4):389-393.

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Abstract PDF: A 46-year-old woman presented with multiple purpuric skin rashes together with splenomegaly and thrombocytopenia. The resected spleen weighed 720 gm, and contained multiple, discrete, dark red, bulging nodules throughout the entire splenic parenchyma. Microscopically, the nodules revealed various range of endothelial cell proliferation which included cavernous vasculature, anastomosing vascular channels, papillary intravascular growth and solid pattern. Intrasinusoidal sequestration of platelets along with extramedullary hematopoiesis was present in the neoplastic vascular spacws. Neither exposure to thorotrast nor vinyl chloride was nonfirmed. Ultrastructurally, the tumor cells demonstrated a row of pinocytotic vesicles, occasional intercellular junctional structures, and the paucity/absence of filamentous structures or Weibel-Palade bodies supports origin of sinus lining type cells. After splenectomy, the thrombocyte count returned to normal. We conclude this is the first case of primary splenic angiosarcoma with Kasabach-Merritt syndrome in Korean literature.

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