Neutrophilic panniculitis associated with myelodysplastic syndromes is rare. We report a 59-year-old patient who initially was diagnosed with myelodysplastic syndrome (MDS) and developed a sudden onset of widespread pustulosis and erythematous indurated papules. Examination of skin biopsies of a papule lesion showed dense neutrophilic infiltration limited to the subcutaneous tissue. The pustules and papules disappeared completely after treatment with systemic corticosteroids. To our knowledge, only one patient was identified by MEDLINE search of the English-language literature.