• Mengyuan Lin, Honghua Wang, Yan Wang, and Shi-Wen Jiang.
• Center of Reproductive Medicine, Women's Hospital of Jiangnan University, Wuxi Jiangsu, China.
• Medicine (Baltimore). 2023 Jun 23; 102 (25): e34023e34023.

RationaleErectile dysfunction (ED) is common in middle-aged and elderly men, affecting more than 100 million males worldwide. Most ED cases can be attributed to organic and/or psychological factors. Here we report an atypical ED case with no clear manifestation fitting the diagnosis for recognized types of ED.Patient ConcernsThe 35-year-old male is unable to have normal erection since puberty, and unable to complete intercourse with his wife. He had no history of trauma, surgery or psychiatric/psychological disease. The patient has a normal male karyotype. There is no significant finding in physical examination, nocturnal penile tumescence test, and ultrasound measurement of penis vascular functions. The serum levels of major hormones are all in normal ranges.DiagnosesAtypical ED, psychogenic ED not excluded; infertility.InterventionsOral phosphodiesterase inhibitors Tadalafil (20 mg, BIW) or Sildenafil (50 mg, BIW) had no effect in this patient. Penile prosthesis implantation helped the patient to acquire normal sexual life, but did solve the ejaculation failure and infertility. Motile sperms were obtained by testicular epididymal sperm aspiration under the guidance of ultrasound, and intracytoplasmic sperm injection was performed with occytes retrieved from his wife.OutcomesThe patient sexual life was significantly improved after penile prosthesis implantation; the patient wife is currently in the first trimester of pregnancy as the result of in vitro fertilization.ConclusionsThe no response to phosphodiesterase type 5 inhibitors (PDE5) treatment may suggest an impediment of PDE5-related pharmacological pathways or the presence of defect/injury in the neural system. This special case raises a question if some patients with persistent ED may have similar manifestations and can be treated with the same procedures.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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