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Abstract:  We hereby report a 2-year-old boy who presented with a peculiar combination of hyperpigmented and hypopigmented skin lesions along the lines of Blaschko, sharply demarcated at the midline, both on the anterior and posterior trunk. Although combinations of hyperpigmented and hypopigmented skin lesions distributed along the lines of Blaschko have been described, the combination of skin lesion comprised hypomelanosis of Ito and whorled hypermelanosis distributed in the extremely unusual manner described in this patient has not been reported previously.