Effective amelioration of the Lucio phenomenon with adjuvant tofacitinib therapy in a patient with dual infection of Mycobacterium leprae and Mycobacterium lepromatosis: a case report from India

Abstract

Introduction. The Lucio phenomenon (LP) is a characteristic reaction pattern seen in patients with diffuse lepromatous leprosy (DLL). Dual infection with Mycobacterium leprae and Mycobacterium lepromatosis in DLL has been confirmed from other endemic countries but not previously documented from India. Conventionally, LP is treated with a high dose of systemic glucocorticoid (GC) and anti-leprosy treatment (ALT). Here we report a case of leprosy lymphadenitis at initial presentation in a patient with LP and DLL due to dual infection with M. leprae and M. lepromatosis who responded favourably to tofacitinib as adjuvant to ALT and systemic GC therapy. Case report. A 20- to 30-year-old man presented with swelling over the bilateral inguinal region, pus-filled skin lesions with multiple ulcers, fever and joint pain. Post-hospitalization investigations showed the presence of anaemia, leukocytosis, and elevated acute and chronic inflammatory markers. Skin and lymph node biopsies were suggestive of LP and leprosy lymphadenitis. The presence of M. leprae and M. lepromatosis was confirmed by PCR followed by DNA sequencing of PCR amplicons from tissue. Despite anti-leprosy treatment, oral GC and thalidomide therapy, the patient continued to develop new lesions. One month after the commencement of adjuvant tofacitinib, the patient showed excellent clinical improvement with healing of all existing lesions and cessation of new LP lesions. Conclusion. Our case confirms the presence of dual infection with M. leprae and lepromatosis in India. Lymph node involvement as an initial presentation of DLL should be considered in endemic areas. Tofacitinib may be a promising new adjuvant therapy for recalcitrant lepra reactions.