Abstract Limited progress has been made over the past three decades in improving survival for patients with osteosarcoma after standard therapy. ALMB0168 is a first-in-class therapeutic antibody agonist targeting Cx43 hemichannels and has demonstrated to suppress the growth and migration of osteosarcoma in preclinical studies. This article presents a case of a patient with bilateral femoral osteosarcoma who was treated with ALMB0168. The patient, who had metastases in both contralateral limb and lung with ≥3 prior lines chemotherapy, showed immediate lesion shrinkage upon treatment and subsequently achieved durable partial response with a long duration of the response. The patient had a progression-free survival of 23 months to date, and the treatment was still ongoing. This case highlights that ALMB0168 may represent a promising treatment option for patients with metastatic or unresectable osteosarcoma after standard chemotherapy.
Huang et al. (2026) studied this question.