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Beh乊et's disease (BD) is a multisystem disorder with a wide variety of clinical presentation, but myelodysplastic syndrome (MDS) is rarely associated with this disease. Among 18 cases of BD-associated MDS previously reported, only 10 cases showed intestinal involvement. Furthermore, there is no case report of MDS combined with intestinal BD. Herein, we report a patient of intestinal BD who had MDS with chromosomal trisomy 8.