A Unique Case of Late Recurrent Langerhans Cell Histiocytosis and Postpartum Rheumatoid Arthritis Offers Insight into Possible Disease Triggers - Abstract
We report a unique case of relapsed Langerhans cell histiocytosis (LCH) occurring 29 years after initial diagnosis. Recurrence was temporally related to discontinuation of methotrexate, originally used as a treatment for the patient’s poorly responsive rheumatoid arthritis, which also developed within a year postpartum. There are rare late relapses of LCH described in the literature and these have been largely limited to the orbit or bony structures. In contrast, our case showed widespread multisystem disease with lymph node, lung, and bone involvement at initial diagnosis and relapsed with only nodal disease and focal bone involvement. The association with new onset postpartum rheumatoid arthritis is also novel and offers possible insight into pathogenesis. We discuss the association between Langerhans cell histiocytosis, rheumatoid arthritis and pregnancy. We also review late recurrent LCH and potential treatment strategies for this unique population.