Dual Biologic Therapy in an Adolescent With Camurati-Engelmann Disease and Crohn Disease.

Camurati-Engelmann disease (CED) is a rare disorder caused by activating mutations in the TGF-ß1 gene and characterized by hyperostosis of long bones and bone dysplasia. We describe a case of an adolescent with CED and moderate-severe Crohn Disease (CD). Infliximab improved gastrointestinal symptoms but was associated with worsening CED-associated bone pain. Clinical remission was successfully achieved with dual biologic therapy that included vedolizumab and ustekinumab. Possible reasons for this patient's clinical response are advanced and include speculation about the complex role of TGF-ß1 signaling in the etiology of both CED and CD.