Introduction:  Lucio phenomenon (LP) is characteristic reaction pattern seen in patient of diffuse lepromatous leprosy(DLL). Dual infection of  M. leprae  and  M. lepromatosis  in case of DLL is confirmed from other endemic country but same is yet to be documented from India. Conventionally LP is treated with high dose of systemic glucocorticoid (GC) and anti-leprosy treatment (ALT). Here we are reporting an interesting case of leprosy lymphadenitis as initial presentation in case of LP and DLL due to dual infection with  M. leprae  and  M. lepromatosis  who responded favorably to tofacitinib as adjuvant to ALT and systemic GC therapy.  Case report:  A 24-year-old man presented with complaint of swelling over bilateral inguinal region, pus filled skin lesions with multiple ulcers, fever and joint pain. Post hospitalization investigations showed presence of anemia, leukocytosis and elevated acute and chronic inflammatory markers. Skin and lymph node biopsies were suggestive of LP and Leprosy lymphadenitis. Presence of  M.leprae  and  M. lepromatosis  was confirmed by PCR study of tissue. Despite of anti-leprosy treatment, oral GC and thalidomide therapy patient continue developing new lesions. Post one-month commencement of adjuvant tofacitinib, patient experienced excellent clinical improvement as healing of all existing lesions and cessation of new lesion of LP.  Conclusion:  Our case confirms the presence of dual infection with  M. leprae  and  lepromatosis  in India.  Lymph node involvement as initial presentation of DLL should be considered in endemic area. Tofacitinib can emerge as promising new adjuvant therapy for recalcitrant lepra reaction.

Hypereosinophilia can be primary, including idiopathic hypereosinophilic syndrome (HES) and chronic eosinophilic leukemia, or secondary/reactive to various infective and non-infective stimuli. Chronic oro-genital ulcerations can occur due to various dermatological and non-dermatological disorders, and many times it serves as a useful indicator of an underlying systemic disorder. Hence, a case presenting with chronic oro-genital ulcerations needs a thorough evaluation. We are reporting an interesting case of a middle-aged male who had chronic oro-genital ulcerations as a presenting feature of chronic eosinophilic leukemia with FIP1L1-PDGFRA fusion (FIP1-like 1/platelet-derived growth factor receptor alpha). The patient’s oro-genital ulcerations responded excellently to imatinib.