Rebleeding in moyamoya disease at different sites from the initial bleeding is usually a result of increased hemodynamic stress and usually occurs after relatively long intervals. In contrast, rebleeding at the initial site generally develops in a fairly short time due to rupture of tiny aneurysms at the periphery of collateral vessels. Herein, we describe a young female patient with moyamoya disease with contralateral rebleeding just four weeks after the first onset. This is quite different from typical rebleeding reports, and raises the concern that rebleeding at different sites can still develop in a relatively short time after the initial attack. Early cerebral angiography is important to confirm the diagnosis. Although the pathogenesis of rebleeding has not been well established, this case report should remind clinicians to be alert to the unusual presentation of moyamoya disease with contralateral hemisphere rebleeding.