| Summary: | Introduction Spina bifida is a congenital anomaly caused by a neural tube closure defect that may result in sexual dysfunction. Sexual dysfunction and infertility are prevalent in spina bifida patients but have been scarcely reported. Case presentation We report the case of a 36‐year‐old man with spina bifida and mild‐moderate erectile dysfunction. He could experience erection and ejaculation. He got married at the age of 28 years, but his wife was unable to conceive for 2 years thereafter. Semen analyses revealed that semen volume, sperm density, and sperm motility rate were below normal levels. It was concluded that natural conception would be difficult, and assisted reproductive strategies were planned. After 4 years, his wife conceived through intracytoplasmic sperm injection and gave birth to a healthy baby. Conclusion Fertility treatment, including intracytoplasmic sperm injection, is a useful therapeutic method for male patients with spina bifida who desire to father a child.
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