Abstract
Horseshoe kidney (HSK) is not an uncommon genitourinary anomaly, with incidence of about 1 in 400. Males are more commonly affected than females. HSK with a single ureter is extremely rare. We report the case of infant boy with HSK with a single ureter, presenting as case of posterior urethral valves (PUV) and obstructive megaureter. Radiological investigations showed PUV, which was managed via resection using urethrotome, and obstructive megaureter, which was managed temporarily by distal cutaneous ureterostomy and then by definitive ureteral reimplantation surgery. To our knowledge, no single case with a similar presentation has been reported in the literature.