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A 36-year-old female presented with two episodes of hemoptysis induced by exertion and acute dyspnea. She was diagnosed as a case of hypertrophic obstructive cardiomyopathy (HOCM) with systolic anterior motion (SAM)-induced severe mitral regurgitation (MR). She underwent extended septal myectomy with mitral valve replacement under cardiopulmonary bypass and recovered successfully. This is a unique and rare combination of HOCM with hemoptysis.Cardiac rhabdomyoma is the most common primary heart tumor in childhood. This tumor, which is fre