To the Editors:
Invasive candidal infections are increasing in incidence and have become the third most common cause of late-onset infection among premature infants. Candida endocarditis has been found in 5.5% to 15.2% of cases of fungal sepsis and can be associated with higher mortality than fungemia alone.1–3 Candida species other than C. albicans have been rarely isolated.1 We report a premature infant, successfully treated for a large mycotic vegetation in the right atrium (RA) secondary to C. dubliniensis sepsis refractory to antimycotic treatment. This is a second report of C. dubliniensis as a cause of neonatal endocarditis.4
A 1400-g female was delivered at 33 weeks gestation with gastroschisis and apple-peel intestinal atresia accompanied with a short gut and atretic microcolon originating from the splenic flexure. Prophylactic antibiotic treatment and total parenteral nutrition were started through a catheter placed at the RA/inferior vena cava junction. Initially, necrotic parts of intestine were removed, and enterostomy with temporary abdominal closure was performed. She subsequently developed ileus with an enterocutaneous fistula, and additional intestinal resection was done. The proximal 30 cm of the intestines were preserved only.
At the age of 10 days, she deteriorated with apnea, bradycardia, hypotension, lethargy, and fever. Complete blood count revealed thrombocytopenia (T 5 × 109/L), leukopenia with a left shift (L 5.9 × 109/L, immature/mature neutrophil ratio 0.56), and elevated C-reactive protein (216 mg/L). Blood cultures grew C. dubliniensis susceptible to amphotericin B and fluconazole. The catheter was withdrawn, and treatment with liposomal amphotericin B (5 mg/kg/d) was started because of impaired renal function (BUN 131 mg/dl and creatinine 1.7 mg/dl). Despite therapy, the infant remained thrombocytopenic and blood cultures continued to grow C. dubliniensis. Echocardiogram revealed a gigantic floating polypoid thrombus in the RA measuring 20 × 10 mm (Fig. 1), originating from the inferolateral RA wall next to the inferior vena cava orifice intermittently reaching the right ventricular cavity (see Video, Supplemental Digital Content 1, https://links.lww.com/INF/A474, which represents echocardiogram in subcostal 4 chamber view). Candidal involvement of other organs was excluded. Therapy was modified to liposomal amphotericin B (20 mg/kg/d) with fluconazole (12 mg/kg/d).
Because antifungal therapy after 9-week duration failed to obtain sterilization and regression of the thrombus, the infant was referred for surgery. Her body weight was 2300 g, at that time. After successful thrombectomy, blood cultures become negative. Diagnosis was verified by the clinical pathologist and microbiology tests. Antimycotic therapy was discontinued 8 weeks after negative cultures were achieved. Two more operations were required to reestablish intestinal continuity. On 18 months follow-up, she has no neurodevelopmental abnormalities, she is gaining weight on full enteral feeding, and there has been no recurrence of Candida infection.
The small number of cases reported so far precludes a definitive recommendation about the optimal treatment concerning infected intracardiac thrombi. Current options include antifungal chemotherapy, thrombolysis, and surgical intervention.5 We believe that the excellent outcome in our case validates the strategy that infected atrial thrombus refractory to medical treatment can be safely removed by cardiotomy even in preterm infants and that surgery could be potentially lifesaving in such cases.
Ruža Grizelj, MD
Jurica Vuković, MD, PhD
Dalibor Šarić, MD
Department of Pediatrics
University of Zagreb
Tomislav Luetić, MD, PhD
Department of Pediatric Surgery
University of Zagreb
1. Kaufman D, Fairchild D. Clinical microbiology of bacterial and fungal sepsis in very-low-birth-weight infants. Clin Microbiol Rev
2. Chapman RL, Faix RG. Persistently positive cultures and outcome in invasive neonatal candidiasis. Pediatr Infect Dis J
3. Noyola DE, Fernandez EH, Moylett H, et al. Ophtalmologic, visceral, and cardiac involvement in neonates with candidemia. Clin Infect Dis
4. Kaufman D, Boyle R, Hazen KC, et al. Fluconazole prophylaxis against fungal colonization and infection in preterm infants. N Engl J Med
5. Rimensberger PC, Humbert JR, Beghetti M. Management of preterm infants with intracardiac thrombi. Pediatr Drugs