Aim Scarcity of the interstitial cells of Cajal (ICC) is related to motility disorders. In the study, we aimed to evaluate the number and density of ICCs in the fetal rat esophagus in the adriamycin - esophageal atresia (EA) model. Material and Method: Rat fetuses were divided into three groups as a control, adriamycin group without EA and adriamycin group with EA. Four doses of adriamycin, 2 mg/kg each, were injected intraperitoneally to the adriamycin group rats between on 6 and 9 days of gestation. The presence of ICCs in the esophagus of the rat fetuses was determined by using an immunohistochemistry technique (c-kit, CD117). The average numbers of ICCs were calculated with microscopic evaluation by using a visual scoring system (range1 to 3). Results: Seven fetuses were included in each group. The ICCs score 3 distributions of fetuses were 5 (72%) fetuses in the control group, 3 (43%) fetuses in the adriamycin group without EA, 1 (14%) fetus in the adriamycin group with EA. It have been found that there was a marked reduction of ICCs distribution in the adriamycin group with EA compared to control group (p < 0.05). There was no significant difference the ICCs distribution between the control group and the adriamycin group without EA (p > 0.05). Discussion: ICCs density was significantly decreased in the rat fetuses with EA compared to the fetuses without EA. These findings support the idea that ICCs density may be congenitally abnormal in EA. This may be led to dismotility seen in the operated esophagus due to EA.