Diffuse sclerosing osteomyelitis (DSO) in mandible is a disease with unusual and undefined clinicoradiographic manifestations. Several medicines have been administrated for therapy, but the efficacy in bone remolding remains to be systematically evaluated.

A 37-year-old male and a 28-year-old female were diagnosed DSO after revealing diffuse sclerosis and osteolytic changes in the mandible, and treated by Janus-activated kinase signal and IL-6 inhibitor, respectively. Their symptoms were well controlled and the inflammatory indicators were decreased. During the mandible remolding, the osteolytic destruction in one patient migrated from the left mandible to the right, while DSO lesion migrated from the left mandibular body to the ramus and condyle in the other.

DSO presented as migratory lesions in mandible after biological inhibitors therapy, while the periosteal reaction wasn't conformed to the osteolytic focus. These findings suggest that periostitis may be the direct cause of DSO rather than the infectious triggers.