We report an unusual case of unilateral limb pseudo hypertrophy in a 21-year-old lady who developed progressive enlargement of the right calf followed by thigh in association with chronic leg pain. Magnetic resonance imaging (MRI) of the affected limb confirmed enlargement of various muscles. Electromyography revealed neurogenic features consistent with S1 radiculopathy. MRI of the lumbosacral spine showed tethered cord with a lipoma infiltrating multiple sacral roots. Our case illustrates that muscular pseudo hypertrophy may follow chronic denervation as a consequence of spinal neural compressive disease. The various mechanisms postulated for this distinct condition are outlined.