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Concomitant angiosarcoma and lymphoproliferative disorder in solid organ transplant recipients.

Baer LN, Savage DG, Hibshoosh HH, Kalinsky K - Clin Sarcoma Res (2014)

Bottom Line: The malignancies most commonly encountered are non-melanoma skin cancers, carcinomas of lung or breast and posttransplant lymphoproliferative disorders.Angiosarcoma, an uncommon vascular mesenchymal neoplasm, is rare in the posttransplant setting.Notably, in both patients, the diagnosis of angiosarcoma was preceded by diagnosis of a lymphoproliferative disorder with monoclonal immunoglobulin heavy chain rearrangement.

View Article: PubMed Central - PubMed

Affiliation: Division of Hematology and Oncology, Stony Brook Medicine, New York, NY USA.

ABSTRACT
An increased risk of posttransplant malignancy has been consistently reported following various solid organ transplants. The malignancies most commonly encountered are non-melanoma skin cancers, carcinomas of lung or breast and posttransplant lymphoproliferative disorders. Angiosarcoma, an uncommon vascular mesenchymal neoplasm, is rare in the posttransplant setting. This report describes two patients who developed high-grade angiosarcoma following a solid organ transplant. Notably, in both patients, the diagnosis of angiosarcoma was preceded by diagnosis of a lymphoproliferative disorder with monoclonal immunoglobulin heavy chain rearrangement.

No MeSH data available.


Related in: MedlinePlus

Histopathology of patients with angiosarcoma in the setting of solid organ transplant. A) CM angiosarcoma 200x: Needle core biopsy of the liver showing inter-anastomosing channels with papillary formation lined by atypical endothelial cells typical of angiosarcoma (200X). B) CM angiosarcoma 400X CD31: Immunohistochemical stain against CD31(endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this angiosarcoma (400X). C) KM angiosarcoma 200X: A high grade angiosarcoma composed of cellular spindle malignancy growing in fascicles with multiple mitotic figures with scattered red blood cells but without evidence of vascular formation (200X). D) KM CD31 400X: Immunohistochemical stain against CD31 (endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this high-grade angiosarcoma (400X).
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Fig1: Histopathology of patients with angiosarcoma in the setting of solid organ transplant. A) CM angiosarcoma 200x: Needle core biopsy of the liver showing inter-anastomosing channels with papillary formation lined by atypical endothelial cells typical of angiosarcoma (200X). B) CM angiosarcoma 400X CD31: Immunohistochemical stain against CD31(endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this angiosarcoma (400X). C) KM angiosarcoma 200X: A high grade angiosarcoma composed of cellular spindle malignancy growing in fascicles with multiple mitotic figures with scattered red blood cells but without evidence of vascular formation (200X). D) KM CD31 400X: Immunohistochemical stain against CD31 (endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this high-grade angiosarcoma (400X).

Mentions: 62-year-old man with alcoholic cirrhosis initially treated with chemoembolization for hepatocellular carcinoma. He underwent orthotopic liver transplant in January 2006. Following transplant, he was maintained on the immunosuppressant tacrolimus. A routine surveillance MRI performed two years later (April 2008) revealed a 3.5 cm left adrenal mass. Systemic imaging, including PET/CT, reported an adrenal mass, splenomegaly (18 cm) and FDG-avid mesenteric and para-aortic lymphadenopathy. Biopsy of the left adrenal mass showed a post-transplant lymphoproliferative disorder (PTLD), consistent with an Epstein-Barr virus (EBV) negative, monomorphic, diffuse large B-cell lymphoma. Spinal fluid and bone marrow were both unremarkable. EBV DNA was undetectable by PCR. The tacrolimus dose was decreased by half, and the patient was started on CHOP chemotherapy (cyclophosphamide, doxorubicin, vincristine and prednisone) with rituximab. Interim evaluation with a PET/CT showed disease progression and treatment was switched to the ICE regimen (ifosfamide, carboplatin and etoposide) with rituximab. Autologous stem cells were harvested and stored after his ICE chemotherapy. Upon completion of chemotherapy, PET/CT demonstrated disease remission and bone marrow biopsy remained negative for lymphoma. Five months later, the patient developed a firm swelling of his upper lip. A biopsy revealed monophasic diffuse large-B-cell lymphoma with clonal heavy chain rearrangement. A PET/CT showed FDG avidity in the upper lip and sub-mandibular lymph node. Due to concern for significant local morbidity, he was treated with dexamethasone and a palliative course of radiation. In view of limited improvement, the radiation was discontinued after only four fractions. He then received 2 additional cycles of ICE with rituximab. In April 2009, the patient was admitted for autologous stem cell transplant, following etoposide, carboplatin and thiotepa conditioning. He tolerated the autologous transplant well, but a subsequent PET/CT showed continued FDG avidity in the upper lip. Local radiation therapy was resumed. Following stem cell transplant, he was maintained on rituximab every three months, the last of which was given in November 2010.In February 2011, he presented with dyspnea on minimal exertion, abdominal dissension, malignant ascites, and disseminated intravascular coagulation. Imaging revealed a cirrhotic liver, portal hypertension, and a new 23 cm hepatic mass with hemoperitoneum. Biopsy of the liver mass revealed highly atypical cells with hyperchromasia and markedly pleomorphic nuclei with prominent nucleoli and markedly increased mitotic activity including atypical mitotic figures (Figure 1A). Immunohistochemical assessment revealed tumor cells to be positive for CD31 (Figure 1B) and focally positive for CD34, suggestive of high-grade angiosarcoma. The cells were negative for hepatocellular specific antigen and Human Herpes virus 8 (HHV-8). EBV DNA was undetectable by PCR. His hospital stay was further complicated by respiratory failure and renal failure requiring initiation of dialysis. The patient passed away prior to initiation of systemic therapy.Figure 1


Concomitant angiosarcoma and lymphoproliferative disorder in solid organ transplant recipients.

Baer LN, Savage DG, Hibshoosh HH, Kalinsky K - Clin Sarcoma Res (2014)

Histopathology of patients with angiosarcoma in the setting of solid organ transplant. A) CM angiosarcoma 200x: Needle core biopsy of the liver showing inter-anastomosing channels with papillary formation lined by atypical endothelial cells typical of angiosarcoma (200X). B) CM angiosarcoma 400X CD31: Immunohistochemical stain against CD31(endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this angiosarcoma (400X). C) KM angiosarcoma 200X: A high grade angiosarcoma composed of cellular spindle malignancy growing in fascicles with multiple mitotic figures with scattered red blood cells but without evidence of vascular formation (200X). D) KM CD31 400X: Immunohistochemical stain against CD31 (endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this high-grade angiosarcoma (400X).
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4233048&req=5

Fig1: Histopathology of patients with angiosarcoma in the setting of solid organ transplant. A) CM angiosarcoma 200x: Needle core biopsy of the liver showing inter-anastomosing channels with papillary formation lined by atypical endothelial cells typical of angiosarcoma (200X). B) CM angiosarcoma 400X CD31: Immunohistochemical stain against CD31(endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this angiosarcoma (400X). C) KM angiosarcoma 200X: A high grade angiosarcoma composed of cellular spindle malignancy growing in fascicles with multiple mitotic figures with scattered red blood cells but without evidence of vascular formation (200X). D) KM CD31 400X: Immunohistochemical stain against CD31 (endothelial marker) demonstrates with brown staining the neoplastic endothelial cells of this high-grade angiosarcoma (400X).
Mentions: 62-year-old man with alcoholic cirrhosis initially treated with chemoembolization for hepatocellular carcinoma. He underwent orthotopic liver transplant in January 2006. Following transplant, he was maintained on the immunosuppressant tacrolimus. A routine surveillance MRI performed two years later (April 2008) revealed a 3.5 cm left adrenal mass. Systemic imaging, including PET/CT, reported an adrenal mass, splenomegaly (18 cm) and FDG-avid mesenteric and para-aortic lymphadenopathy. Biopsy of the left adrenal mass showed a post-transplant lymphoproliferative disorder (PTLD), consistent with an Epstein-Barr virus (EBV) negative, monomorphic, diffuse large B-cell lymphoma. Spinal fluid and bone marrow were both unremarkable. EBV DNA was undetectable by PCR. The tacrolimus dose was decreased by half, and the patient was started on CHOP chemotherapy (cyclophosphamide, doxorubicin, vincristine and prednisone) with rituximab. Interim evaluation with a PET/CT showed disease progression and treatment was switched to the ICE regimen (ifosfamide, carboplatin and etoposide) with rituximab. Autologous stem cells were harvested and stored after his ICE chemotherapy. Upon completion of chemotherapy, PET/CT demonstrated disease remission and bone marrow biopsy remained negative for lymphoma. Five months later, the patient developed a firm swelling of his upper lip. A biopsy revealed monophasic diffuse large-B-cell lymphoma with clonal heavy chain rearrangement. A PET/CT showed FDG avidity in the upper lip and sub-mandibular lymph node. Due to concern for significant local morbidity, he was treated with dexamethasone and a palliative course of radiation. In view of limited improvement, the radiation was discontinued after only four fractions. He then received 2 additional cycles of ICE with rituximab. In April 2009, the patient was admitted for autologous stem cell transplant, following etoposide, carboplatin and thiotepa conditioning. He tolerated the autologous transplant well, but a subsequent PET/CT showed continued FDG avidity in the upper lip. Local radiation therapy was resumed. Following stem cell transplant, he was maintained on rituximab every three months, the last of which was given in November 2010.In February 2011, he presented with dyspnea on minimal exertion, abdominal dissension, malignant ascites, and disseminated intravascular coagulation. Imaging revealed a cirrhotic liver, portal hypertension, and a new 23 cm hepatic mass with hemoperitoneum. Biopsy of the liver mass revealed highly atypical cells with hyperchromasia and markedly pleomorphic nuclei with prominent nucleoli and markedly increased mitotic activity including atypical mitotic figures (Figure 1A). Immunohistochemical assessment revealed tumor cells to be positive for CD31 (Figure 1B) and focally positive for CD34, suggestive of high-grade angiosarcoma. The cells were negative for hepatocellular specific antigen and Human Herpes virus 8 (HHV-8). EBV DNA was undetectable by PCR. His hospital stay was further complicated by respiratory failure and renal failure requiring initiation of dialysis. The patient passed away prior to initiation of systemic therapy.Figure 1

Bottom Line: The malignancies most commonly encountered are non-melanoma skin cancers, carcinomas of lung or breast and posttransplant lymphoproliferative disorders.Angiosarcoma, an uncommon vascular mesenchymal neoplasm, is rare in the posttransplant setting.Notably, in both patients, the diagnosis of angiosarcoma was preceded by diagnosis of a lymphoproliferative disorder with monoclonal immunoglobulin heavy chain rearrangement.

View Article: PubMed Central - PubMed

Affiliation: Division of Hematology and Oncology, Stony Brook Medicine, New York, NY USA.

ABSTRACT
An increased risk of posttransplant malignancy has been consistently reported following various solid organ transplants. The malignancies most commonly encountered are non-melanoma skin cancers, carcinomas of lung or breast and posttransplant lymphoproliferative disorders. Angiosarcoma, an uncommon vascular mesenchymal neoplasm, is rare in the posttransplant setting. This report describes two patients who developed high-grade angiosarcoma following a solid organ transplant. Notably, in both patients, the diagnosis of angiosarcoma was preceded by diagnosis of a lymphoproliferative disorder with monoclonal immunoglobulin heavy chain rearrangement.

No MeSH data available.


Related in: MedlinePlus