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Purpose: Isotretinoin is the most effective anti-acne drug with a long-term remission. However; it contains severe teratogenic effects with serious adverse drug reactions, which limits the use of medication. Methods: To review the use of isotretinoin during pregnancy, we carried out a comprehensive search of literature in Google Scholar, Scopus and PubMed/Medline from their inception until April 2015. Results: Database searching identified 277 records, of which, 38 articles were retrieved according to abstract and title assessment. After full-text review, 17 articles were excluded and finally, a total of 21 studies met the inclusion criteria. Data showed an increased pattern in the use of isotretinoin. In some studies, health care providers were not fully adhered to the risk reduction programs in pregnancy. Exposing to isotretinoin among pregnant women has still occurred due to detrimental adherence to risk reduction programs which resulted in live-born infants with different kinds of abnormalities. Conclusion: Despite the known serious adverse effect of isotretinoin, the use of drug was not based on the guidelines in some cases, which needs more attentions to prevent the severe drug related problems.