| Summary: | Alveolar rhabdomyosarcoma (RMS) is one of the most common pediatric soft-tissue neoplasms. Breast involvement either as primary tumor or metastasis is extremely rare. Herein, we report a case of primary limb alveolar RMS with breast metastases in a young premenarcheal girl that relapsed only to the metastatic breast site after achieving complete response. Accordingly, we believe that investigations of the mammary glands should be part of the routine diagnostic workup in adolescent females with RMS. Local therapeutic measures to control breast disease, including surgery or radiotherapy has to be considered for better prognosis. Newer radiation modalities aiming at reducing side effects should be developed.
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