Objective To survey on the recognition and diagnosis capability of frontline healthcare workers in boy's genital malformation in Shufu, Xinjiang. Thereby to provide data support for the healthcare workers training there. Methods We designed a questionnaire and carried out a survey in Shufu people's hospital, township clinics and village doctors. Based on SPSS 13.0 data analysis, we made comparison on recognition difference between the Han and Uygur healthcare workers. Results 400 questionnaires were handed out, among them 365 were valid which accounted for 91.25%. The survey showed that there were 54.79% survey participators whose recognition correct rate was over 50% in Shufu, Xinjiang (21.92% for man, 32.87% for woman), 50% Han healthcare workers whose recognition correct rate was over 50% and that of 57.4% in Uygur peers.There was no significant difference in statistics(P<0.05). It accounted for 38.36% that participating in training as the access to relevant knowledge of genital malformation,but 61.64% was not. In clinical practice, 86.3% of survey participators screen the possibility of newborn genital malformation. And 27.39% of screened newborn have genital malformation. Conclusion The frontline healthcare workers in Shufu, Xinjiang at large are short of diagnosis knowledge on boy's genital malformation, as a result that they are difficult to recognize and give proper treatment on such cases in practice. The status thereby shows that it is critical to improve the diagnosis and treatment capability of frontline healthcare workers in boy's genital malformation in Xinjiang ethnic group area.