PurposeDisseminated nontuberculous mycobacterial (DNTM) infection can involve multiple organs, including the lungs, skin and soft tissues and lymph nodes. However, NTM infection leading to osteolysis has been rarely reported. Here, we analyzed the clinical features, osteolytic mechanisms, treatment and prognosis of patients with DNTM disease with osteolytic lesions.Patients and MethodsThis retrospective study was conducted between January 1, 2011, and December 31, 2020, at the First Affiliated Hospital of Guangxi Medical University and the Fourth People’s Hospital of Nanning City. Patients who had culture and/or histopathological proof of DNTM disease with osteolytic lesions were included.ResultsTen HIV-negative patients with DNTM disease with osteolytic lesions were enrolled. Five of these patients had underlying diseases. Seven and three of the patients were positive and negative for anti-interferon-γ autoantibodies (AIGAs), respectively. The AIGA positivity rate was 70% (7/10). Ostealgia and anemia were the most common symptoms, followed by fever, emaciation, cough, expectoration, anorexia, subcutaneous abscesses and lymphadenopathy. Leukocyte and neutrophil counts were increased. The most common sites were the vertebrae, sternum, clavicle and ribs, although the femur, ilium, humerus, and scapula were also involved. Radiography and computed tomography (CT) showed moth-eaten or irregular destruction of bone, bone defects, pathological fracture, periosteal proliferation and surrounding abscesses. Emission CT (ECT) bone scans showed significantly increased uptake in many skeletal regions. Positron emission tomography(PET)/CT showed metabolic activity in multiple bones. All patients received anti-nontuberculous therapy, and five underwent surgery. Two died during treatment.ConclusionDNTM infection of bone and leading to osteolysis usually occurs in patients with AIGA-positive antibodies. DNTM disease with osteolysis is characterized by increased leukocytes and neutrophil counts, focal suppurative granulomas, and multiple areas with moth-eaten or irregular destruction of bone with increased radioactive concentrations. Early diagnosis and timely, effective combination anti-NTM therapy can improve the prognosis.