BACKGROUND: Developmental dysplasia of the hip (DDH) is a congenital musculoskeletal disease that impairs the hip joint and exacerbates hip osteoarthritis. This study aims to investigate the alterations of osteocytic characteristics including apoptosis, lacuna-canalicular network, and perilacunar/canalicular remodeling (PLR) activity in subchondral bone from DDH patients, and potential relationship of these alterations between the cartilage degeneration and DDH progression. METHODS: The femoral head specimens were acquired from 16 patients with hip fractures who received total hip arthroplasty operation, 24 patients with primary hip OA and 25 patients with DDH. The femoral head were scanned by a micro-computed tomography and the volume of interest was used for a micro-finite element analysis. Histological and immunohistochemical staining was used to observe chondrocytes in cartilage and osteocytes in subchondral bone. Terminal deoxynucleotidyl transferase dUTP nick end labeling staining was used to investigate the apoptotic osteocytes in subchondral bone. Ploton silver staining was used to visualize lacunocanalicular network and picrosirius red staining was to visualize collagen fiber orientation in subchondral bone. RESULTS: The DDH group showed the highest apoptosis rate of osteocytes and increased PLR activity among the three groups. The micro-finite-element analysis revealed that DDH group had deteriorative microstructural and biomechanical properties of subchondral bone. The histological and immunohistochemical analyses showed that the cartilage degeneration in DDH group was the most severe. Linear regression analysis revealed a significant correlation between osteocytic activity in subchondral bone and cartilage degeneration in DDH. CONCLUSIONS: Our findings indicate that the abnormal osteocyte activity in subchondral bone might contribute to the deterioration of subchondral bone structure, which accelerates cartilage degeneration and DDH progression. Targeting subchondral bone remodeling could offer a promising therapeutic strategy for DDH.