Heart Transplantation With Pulmonary Vein Reconstruction in Ewing's Sarcoma in the Right Ventricular Outflow Tract

Cho, Won Chul; Kim, Ho Jin; Jung, Sung‐Ho; Kim, Jae‐Joong; Kim, Dong Kwan

doi:10.1111/jocs.12610

Cited by 4 publications

(7 citation statements)

References 8 publications

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“…Only one patient received radiotherapy along with pre- and post-surgical chemotherapy. 14 Of 11 other cases, 5 of them had just tumor resection, 15–19 3 had tumor resection with both chemotherapy and radiotherapy, 20–22 2 of them had cardiac transplantation, 23,24 and 1 had tumor resection with chemotherapy. 25…”

Section: Resultsmentioning

confidence: 99%

“…Only one patient received radiotherapy along with pre-and post-surgical chemotherapy. 14 Of 11 other cases, 5 of them had just tumor resection, [15][16][17][18][19] 3 had tumor resection with both chemotherapy and radiotherapy, [20][21][22] 2 of them had cardiac transplantation, 23,24 and 1 had tumor resection with chemotherapy. 25 In follow-up of different durations from the beginning to a maximum of 30 months, 53% of patients were alive (n = 9); three had no evidence of disease after 24 months (NED = 24), two had NED = 12, and four of them with NED = 30,86,2 months.…”

Section: Primary Pnetmentioning

confidence: 99%

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Primary and metastatic primitive neuroectodermal tumor of the heart: A systematic review

Hajsadeghi

Iranpour

Amouei

et al. 2022

Asian Cardiovasc Thorac Ann

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Objective Primary and metastatic primitive neuroectodermal tumors of the pericardium are uncommon. Two cases are presented and discussed. Methods The cases of a 17- and 38-year-old male patients with neuroectodermal tumors of the pericardium are presented. In addition, a systematic review was performed according to the Preferred Reporting Items and checklist for Systematic reviews and Meta-Analyses (PRISMA). All selected articles’ quality assessment was done using the Joanna Briggs Institute (JBI) Critical Appraisal Checklist for Case Reports. Results The systematic review yielded 29 patients with primary or metastatic PNET. Two patients underwent cardiac transplantation. It seems that unlike considering total resection in other organs, pericardiectomy in PNET patients with pericardial origin may lead to further invasion of the lesions, and it is not recommended. The average disease-free follow-up was 10 months. The combination of neoadjuvant and adjuvant chemotherapy along with surgery and radiotherapy offered the best disease-free outcomes. Conclusion Neuroectodermal heart tumors are rare, aggressive tumors requiring chemotherapy and radiotherapy in association with tumor resection surgery to have the best disease-free duration.

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Section: Resultsmentioning

confidence: 99%

Section: Primary Pnetmentioning

confidence: 99%

Primary and metastatic primitive neuroectodermal tumor of the heart: A systematic review

Hajsadeghi

Iranpour

Amouei

et al. 2022

Asian Cardiovasc Thorac Ann

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“…Among the primary cardiac sarcomas, Ewing’s sarcoma is quite rare. To our knowledge, only a few reports have described primary cardiac Ewing’s sarcoma [1, 2] and no reports have described primary Ewing’s sarcoma of the RA wall.…”

Section: Discussionmentioning

confidence: 99%

Adult-onset primary Ewing’s sarcoma of the right atrium: a case report

et al. 2019

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BackgroundPrimary cardiac tumors, which are only detected in 0.001–0.03% of autopsies, are rare. Only 25% of primary cardiac tumors are malignant, of which 95% are sarcomas. Ewing’s sarcoma, one of the Ewing’s sarcoma of family tumors, is thought to be derived from neural crest cells. While Ewing’s sarcoma usually presents in the bone of children, Ewing’s sarcoma of cardiac origin is rare, with only a few reports described in the literature. The prognosis is unpredictable because of the scarcity and unestablished treatment. We herein report an extremely rare case of primary cardiac Ewing’s sarcoma in the right atrium of a 64-year-old man.Case presentationThe patient is a 64-year-old Japanese male who was referred to our hospital to treat a floating mass of the right atrium (RA). Although the patient was asymptomatic, we performed an operation to urgently resect the floating mass on the next day of admission due to the risk of pulmonary embolism. The operation was performed under cardiopulmonary bypass and cardiac arrest. We resected the tumor with at least 1.5 cm of the RA wall as a margin. The postoperative pathological diagnosis of the mass was compatible with a primitive neuroectodermal tumor (PNET, a form of Ewing’s sarcoma). The cells were positive for CD56, CD99, and Vimentin and negative for S-100 and Desmin. Although no malignant cells were observed in the margin of the resected RA wall and the sarcoma was completely resected, he was transferred to another hospital to receive adjuvant postoperative chemotherapy to improve the prognosis by preventing subclinical micrometastasis.ConclusionsWe experienced an extremely rare case of primary cardiac Ewing’s sarcoma in the right atrium of a 64-year-old man, which was successfully resected under cardiac arrest. Although the sarcoma was completely resected, postoperative chemotherapy and long-term follow-up are recommended for patients with primary cardiac sarcoma because of the high rates of metastasis and recurrence.

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“…In the literature, there are 2 reported cases in which upfront chemotherapy was administered in adult patients before surgical resection and cardiac transplantation, with long-term good prognosis. 8,11 Previous cooperative studies reported a clinical benefit from high-dose chemotherapy for patients with ES and high-risk factors. 20 The use of radiotherapy as adjuvant therapy needs to be evaluated on a case-to-case basis, as the potential benefits from radiotherapy have to be balanced versus long-term toxicities: when necessary, as in our case, the patient and his family need to be informed about the potential life-threatening morbidities, such as nonreversible restrictive cardiomyopathy and myocardial ischemia.…”

Section: Discussionmentioning

confidence: 99%

“…In 2015, Cho et al 11 described for the first time a heart transplantation with pulmonary vein reconstruction in adult EES of the right ventricular outflow tract, after an upfront chemotherapeutic course.…”

Section: Discussionmentioning

confidence: 99%

Pediatric Extraskeletal Ewing Sarcoma Originating in the Heart: A Case Report and Review of the Literature

Buffoni¹,

Nuri

Pomè

et al. 2019

Journal of Pediatric Hematology/Oncology

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Heart Transplantation With Pulmonary Vein Reconstruction in Ewing's Sarcoma in the Right Ventricular Outflow Tract

Abstract: Ewing's sarcoma is a histological type of malignant mesenchymal tumor and rarely originates from the heart. We report a case of Ewing's sarcoma located in the right ventricular outflow tract that invaded the left pulmonary vein in a 29-year-old female.

Cited by 4 publications

References 8 publications

Primary and metastatic primitive neuroectodermal tumor of the heart: A systematic review

Primary and metastatic primitive neuroectodermal tumor of the heart: A systematic review

Adult-onset primary Ewing’s sarcoma of the right atrium: a case report

Pediatric Extraskeletal Ewing Sarcoma Originating in the Heart: A Case Report and Review of the Literature

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