Schistosomiasis is a prevalent parasitic, neglected tropical disease (NTD) affecting over 220 million people globally, especially in sub-Saharan Africa (SSA). The pathology of this disease in terms of intestinal, liver and urinary presentations is well-known, yet its chronic effect on host genitalia is often ignored or overlooked. Male genital schistosomiasis (MGS) is a gender-specific manifestation of urogenital schistosomiasis (UGS), associated with the presence of Schistosoma haematobium eggs and related pathologies in genitalia of men inhabiting or visiting endemic areas in SSA. Despite the first reported by Madden, the epidemiology, diagnostic testing and case management of MGS are not well-described owing to limited research and diminishing focus over several decades.