Pathologic myopia is a subgroup of high myopia with degenerative changes in the posterior segment of the eye. Pathologic myopia appearring in early childhood often has a genetic basis. Pathologic myopia may present alone or accompanied with inherited ocular or systemic diseases. Pathologic myopia as well as high myopia has been studied as either Mendelian trait or complex trait in recent years. A great number of progress have been made on genetic study of pathologic myopia, especially in the identification of genes responsible for it when mutated. In this chapter, progress on genetic study of pathologic myopia, especially causative genes, are briefly described. Application of the genetic information in the clinic as well as future expectation is also briefly discussed.