• Dagmar Adamkova Krakorova, Jana Halamkova, Stepan Tucek, Ondrej Bilek, Jan Kristek, Tomas Kazda, Iva Staniczkova Zambo, Regina Demlova, and Igor Kiss.
• Department of Cancer Comprehensive Care Masaryk Memorial Cancer Institute, Zluty kopec 7.
• Medicine (Baltimore). 2021 Mar 19; 100 (11): e25074e25074.

RationaleThe Ewing sarcoma family of malignant tumors is a group of tumors characterized by morphologically similar round-cell neoplasms and by the presence of a common chromosomal translocation; Ewing sarcoma family of tumors typically occur in children and young adults between 4 to 15 years of age. The primary tumor usually originates in the bone, extraskeletal localization is rare.Patient ConcernWe present a case report concerning a 32-year-old male patient with a primary involvement of the penis.DiagnosisThe histopathology from the first penile biopsy showed a small-cell neuroendocrine carcinoma; however, that result was based on a sample obtained at a different facility than the Sarcoma Center, where the investigating pathologist did not have the adequate expertise. The patient then underwent a radical penectomy and a second reading of the histology was demanded after a radical penile amputation when Ewing sarcoma with R1 resection was confirmed.InterventionsThe patient was referred to the national Sarcoma Center, where - using a multidisciplinary approach - the treatment was started with curative intent. However, it was preceded by a non-standard initiation of the therapy due to the poor primary diagnosis.OutcomesThe non-standard therapy at the onset of the disease caused a poor prognosis of an otherwise curable diagnosis. Despite all that, the patient survived for a relatively long time.LessonsThe treatment of sarcomas with atypical localizations should be conducted by an experienced multidisciplinary team in a center with experience in sarcoma treatment.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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