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1. The findings are described in a male infant, one day old, with congenital bowing of both tibiae amid the left femur, associated with syndactylism, hydrocephalus, and anomalies of the urinary tract.2. Pathologically, the left femur and both tibiae showed narrowing of the diaphysis, anterolateral bowing, marked cortical sclerosis at the site of angulation, and periosteal thickening.3. Cystic areas of rarefaction seen on the roentgenograms were the result of disruption of the normal trabecular pattern of bone rather than proliferation of connective tissue.4. No evidence of fibrous dysplasia was encountered.5. Neurofibromata, café au lait spots, or cutaneous nodules were not found.