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Intralymphatic histiocytosis is a rare benign cutaneous condition first reported in 1994 by O’Grady et al.1 The disorder is defined by the accumulation of CD68+ histiocytes within dilated lymphatic vessels podoplanin+vessels).Clinically, intralymphatic histiocytosis presents with isolated asymmetrical plaques, macules or nodules. It has been included within the reactiveangiomatoses and has recently divided into a primary and secondary forms, the latter associated with systemic diseases, notably rheumatoid arthritis (RA).2 Other associated conditions include chronic inflammatory diseases, metal implantation, malignancy, especially breast cancer, and infection. We present the case of a man with intralymphatic histiocytosis occurring in the skin overlying severe degenerative osteoarthritis of the right shoulder and review all published cases.