摘要Rationale:Lepromatous leprosy can have many atypical presentations,obscuring early diagnosis.We present a case of lepromatous leprosy,presenting with atypical features,which made a diagnostic dilemma.Patient concerns:A 48-year-old man presented with bilateral lower limb oedema and scaly"ichthyosis like"skin rash in both hands and feet,hepatosplenomegaly and pancytopenia,over a course of three months,without any classical features of leprosy.A skin biopsy revealed an unexpected diagnosis of borderline lepromatous leprosy.Diagnosis:Lepromatous leprosy.Interventions:Multi-drug regimen treatment with rifampicin,dapsone and clofazimine for lepromatous leprosy.Outcomes:The patient made a good clinical recovery.Lessons:In endemic settings,clinicians should be aware of similar atypical manifestations of leprosy to face the global challenge of eradicating this chronic deforming disease.