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Abstract

Intra-lymphatic histiocytosis (ILH) is an uncommon dermal condition characterized by the accumulation of histiocytes within dilated lymphatic vessels, traditionally associated with chronic inflammatory diseases such as rheumatoid arthritis. Recent reports have also linked ILH to orthopedic procedures involving joint replacements and metal implants, presumably due to chronic inflammation and lymphatic stasis induced by surgical trauma or implant presence. We describe a case of an 86-year-old male with a remote history of a right shoulder rotator cuff repair with anchor screw placement who developed septic arthritis and was treated with multiple incision and drainage procedures, followed by arthroscopic debridement and synovectomy. The patient developed a complex cystic skin lesion on the right shoulder, which on histopathological examination revealed dilated cystic spaces lined by endothelial cells and filled with numerous histiocytes, including multinucleated forms, along with mild endothelial hyperplasia and areas of hyalinized and myxoid stroma. Special stains were negative for fungal and mycobacterial organisms, and there was no evidence of malignancy or foreign material. The clinical and histologic findings, together with the patient’s orthopedic and infectious history, supported a diagnosis of ILH likely secondary to chronic inflammation and lymphatic obstruction related to prior surgical interventions. This case underscores the importance of considering ILH in the differential diagnosis of skin lesions arising in patients with complex joint infections and surgical histories. Awareness of this entity is crucial to prevent misdiagnosis and to inform appropriate management.