Introduction 
Intramedullary osteosclerosis (IMOS) is a sclerosing dysplasia associated with increased bone formation in the medullary cavity. The term intramedullary osteosclerosis was introduced by Abdul-Karim and al in 1988 to describe a rare pathology associated with new bone formation localized mainly in the diaphysis of the tibia in adult female patients [1]. It is a rare disease, without specific radiological signs except for the osteosclerotic lesion and it is not associated with a family history, infection, trauma or systemic disease [2]. Although the diagnosis of IMOS is confirmed after excluding other sclerotic lesions, it is not well recognized due to its rarity and absence of specific signs. Consequently, these situations can lead to a delay in the diagnosis. Here, we report the clinical and radiological characteristics of a patient diagnosed with IMOS.