Background:Cryptococcus, as a classical “opportunistic” fungal pathogen, is capable of disseminating an invasive infection in immunocompromised hosts. The primary sites of infection include the respiratory and central nervous systems, and skeletal infection was rarely reported. In this case, we describe a case of cryptococcal osteomyelitis involving the left side of the acetabulum in a Chinese patient with chronic hepatitis B.Case Presentation:We retrospectively reviewed the case of a female (with chronic hepatitis B) with left acetabulum pain and limited mobility, with fever occurring during the infection who presented to the Fifth Affiliated Hospital of Zunyi Medical University. Upon imaging, we found osteolytic bone destruction in the left acetabulum with inflammatory changes in the surrounding bone and soft tissue, accompanied by abscess formation. Following an 11-month of antifungal therapy, the clinical symptoms improved and the lesion area reduced in size. In addition, there was no sign of recurrence.Conclusion:Cryptococcus infections should be considered in the differential diagnosis of infectious osteolytic bone lesions, particularly when patients with immune insufficiency. Pathological examinations and fungal cultures are essential to provide a differential diagnosis.