Background: Congenital candidiasis is rare, with less than 100 cases reported and an estimated incidence of 0.1% admitted to the neonatal intensive care units (NICUs). It may manifest as cutaneous or systemic disease, often presenting with polymorphic skin lesions within the first six days of life.This case describes a preterm twin infant with congenital candidiasis caused by Candida albicans.Case Presentation Summary: A female preterm twin infant was delivered at 31 weeks’ gestation via cesarean section for suspected chorioamnionitis. The infants’ birth weight was 1690 g, length 41 cm, and Apgar scores were 8 and 9 at the 1st and 5th minute, respectively. Maternal history was significant for treated vaginal candidiasis in pregnancy. Following delivery room stabilization, the neonate was transferred to the NICU supported by nasal positive airway pressure (nCPAP). Initial antibiotics were given for suspected early-onset sepsis. At six hours of life, the infant developed erythematous, macular skin lesions on the back.Due to a maternal history of candidiasis and suspected sepsis followed by a skin rash, intravenous fluconazole was started at 12 mg/kg/day. Lumbar puncture was not performed due to the involvement of skin on the back. By 24 hours, the erythematous rash, now macular and papular, spread over the neck, trunk, and upper extremities as a burn-like dermatitis.Candida albicans was confirmed by the skin swab and blood culture. By day six, the dose of fluconazole was halved to 6 mg/kg/day because of significant rash regression. Antifungal therapy was continued for 14 days in total. The clinical course was favorable with unremarkable follow-up inflammatory markers, sterile blood cultures, and successful respiratory management. Echocardiography was normal, as well as kidney and liver function. Regarding the second male twin, his laboratory findings were normal on several occasions, and he did not experience any skin manifestations. However, due to congenital candidiasis in the female twin sister, prophylactic fluconazole of 3 mg/kg/day was administered for 14 days. His blood culture was sterile.Learning Points Discussion: Preterm infants, due to their immature skin and immune system, present a higher risk for candidemia, which may mimic an early-onset bacterial sepsis and manifest with symptoms ranging from mild cutaneous signs to severe respiratory distress, liver dysfunction, and multiorgan failure.Prompt initiation of systemic antifungal therapy, such as fluconazole or amphotericin B for at least 14 days, is essential, especially for those exhibiting burn-like dermatitis associated with invasive infection. Our patient had several risk factors, including low birth weight, a central venous catheter, antibiotic therapy, and positive skin swab and blood culture. Delay in systemic treatment, use of only oral or topical therapy, or treating for <10 days in preterm infants has been associated with poor outcomes. Optimal management strategies in term neonates remain a topic of ongoing debate.

