Methods: A 49 year old male was admitted with fever, night sweats and weight loss. He was started on adalimumab 6 weeks prior for severe HS. On admission he was noted to have generalized lymphadenopathy and labs showed leukocytosis and hypercalcemia. CT revealed new bulky retroperitoneal and inguinal adenopathy, as well as splenomegaly. An occipital lymph node biopsy was non diagnostic with extensive infarction and minimal viable tissue. Peripheral blood flow cytometry showed an aberrant T cell population comprising 36% of gated cells and he was found to have positive HTLV-1 serology. Despite treatment with systemic and intra-thecal chemotherapy and Zidovudine, he died 5 months later.
Results: HTLV-1 has a global prevalence of 10-20 million and is highly endemic in Southwest Japan, sub-Saharan Africa, South America and the Caribbean. Major modes of transmission are perinatal, breast feeding, sexual intercourse and blood transfusion. HTLV-1 is associated with ATLL, HTLV-1 associated myelopathy (HAM), HTLV associated uveitis (HU) and an infectious dermatitis. The pathogenesis of ATLL is incompletely understood, however, triggers may include Tax, a viral protein implicated in transformation of infected cells, smoking, high levels of HTLV-1 antibody and TNF-α genetic polymorphisms. There has been only one prior case report linking adalimumab and ATLL in the setting of HTLV-1. A recent case report describes a patient with RA and HTLV-1 and worsening HAM and HU symptoms with Tocilizumab.
Conclusion: Given the poor prognosis of ATLL, known risk of ATLL with HTLV-1 infection and link between HTLV-1 and TNF-α, it may be beneficial to screen patients in highly endemic areas for underlying HTLV-1 infection, prior to TNF-α antagonist treatment.
D. Ahuja, None
J. Horvath, None