Angela Djunaedi, Robertus Arian Datusanantyo, Tjahyo Suryanto
Perineal groove (PG) is a rare congenital anomaly of the perineum, often asymptomatic and expected to undergo spontaneous epithelialization. Due to its rarity, PG is frequently misdiagnosed, leading to inappropriate counseling, unnecessary treatment, and overlooked complications. The infant, delivered through cesarean section at 38–39 weeks due to maternal preeclampsia, presented with a red, moist mucosal tract extending from the posterior fourchette to the anterior anal verge, with no associated anogenital anomalies. Congenital hydrocephalus was confirmed by antenatal imaging and postnatal computed tomography scan, with no familial history of malformations. While PG and hydrocephalus are embryologically unrelated, shared potential risk factors in this case include maternal hypertension, possible folic acid deficiency, secondhand smoke exposure during pregnancy, and maternal betel nut chewing. The PG was managed conservatively and showed spontaneous epithelialization over 2 years. This case highlights the importance of recognizing PG to avoid mismanagement and suggests that maternal health and environmental exposures may contribute to its development. Surgical intervention should be reserved for associated anomalies or complications. © 2026 Turkish Journal of Plastic Surgery.
Department of Surgery, Prof. Dr. W. Z. Johannes General Hospital, Kupang, Indonesia; Department of Plastic Reconstructive and Aesthetic Surgery, Faculty of Medicine, Universitas Brawijaya, Malang, Indonesia; Department of Pediatrics, Prof. Dr. W. Z. Johannes General Hospital, Kupang, Indonesia; Department of Pediatrics, Faculty of Medicine and Veterinary Medicine, Universitas Nusa Cendana, Kupang, Indonesia
