Fournier Gangrene Caused by Candida albicans in an Infant After Cardiac Surgery
Fournier gangrene is a rare, rapidly progressive, life-threatening condition. We report a 23-day-old boy with pulmonary atresia and ventricular septal defect treated surgically, who developed Fournier gangrene. Emergency surgery was performed with tissue sampling for microbiological examination. Candida albicans was confirmed; caspofungin followed by fluconazole was administered with excellent results.
KeywordsFournier gangrene Candida albicans infection Pediatric cardiac surgery Congenital heart defects Echinocandins Fluconazole
Fournier gangrene (FG) is a rare, rapidly progressive, life-threatening condition with necrotizing fasciitis of the perineum first mentioned by Baurienne in 1764, however, described and named after Jean Alfred Fournier and his report from the year 1883 [1, 2]. The disease mostly affects patients between 50 and 60 years of age and is uncommon in children. The typical flora involves polymicrobial bacteria, most commonly Escherichia coli, Proteus, Staphylococcus, Enterococcus and anaerobes; however, single cases of fungal etiology in adults were reported [2, 3, 4, 5]. The risk factors of FG developing include diabetes, immunosuppression, chronic kidney disease, local trauma, urethral stricture and genitourinary infections . It is essential to diagnose FG as early as possible, because the infection can rapidly spread from local necrosis to adjacent organs, leading to sepsis and death; mortality rates of 7.5–88% are reported [5, 6, 7]. We report a case of a male infant, who developed fungal FG after cardiac surgery.
FG is a rare condition with the incidence of 1.6 cases per 100,000 males adult per year . The disease mostly affects patients between 50 and 60 years of age, and in children FG is not common. However, FG is a fulminant and life-threatening disease, because although proper treatment mortality in these groups of patients remains high, mortality rates of 7.5–88% are reported [2, 5, 6, 7, 8]. A literature review found only <60 pediatric cases of FG worldwide, with 66% of those in infants younger than 3 months .
Commonly, the etiology of FG is polymicrobial and the infection is most often caused by three or more microorganisms: E. coli, Proteus, Staphylococcus, Enterococcus and anaerobes [3, 4, 5]. However, single-case reports describing FG caused by fungi are reported. C. albicans, Candida glabrata and Rhisopus microsporus etiology are described in adults [5, 10, 11]. Our case report is unique because to the best of our knowledge there is no report of solitary C. albicans FG in children.
It is essential to diagnose FG as early as possible, because the infection can rapidly spread from local necrosis to adhering organs, causing sepsis and death. The diagnosis of FG is based on clinical symptoms such as intense pain, pruritus and tenderness in the genitalia, usually associated with edema of the overlying skin. It is worth emphasizing the role of round the clock access to microbiological laboratory with wide spectrum of diagnostics examination. The guidelines for FG treatment recommend hemodynamic stabilization, aggressive wide surgical debridement and empirical broad-spectrum antibiotics therapy. Also hyperbaric oxygen therapy is recognized as an additional form of FG treatment . However, worldwide increase in fungal infections incidence results in suggestion of some authors to add the empirical antifungal treatment, especially in patients at risk for fungal infections. This concerns mainly patients with primary and secondary immunodeficiencies. Fluconazole therapy could be a reasonable therapeutic option; however, due to wide-spread use of azoles a large number of non-albicans Candida species isolates in recent years suggest that echinocandins may be a good choice for empiric therapy, especially in hemodynamic unstable patients [3, 5, 12]. Also combined therapy of different antifungal therapeutics groups may be successful if monotherapy fails; echinocandin and liposomal amphotericin are suggested in this setting .
In our case, secondary immunodeficiency in hypotrophic infant was probably caused by significant blood steal syndrome that occurred postoperatively, resulting in vital organs hypoperfusion. Another risk factor for FG was preoperative traumatic urinary catheterization. However, the first symptoms of FG showed relatively late, on 15th postoperative day. Our patient was colonized with methycillin-resistant S. aureus (MRSA) as well as with C. albicans preoperatively; nevertheless, twice as surprisingly the fungi were an etiological factor for FG. One could explain this phenomenon that the perioperative antibiotic prophylaxis appeared effective against MRSA, while no prophylaxis against fungi was used. In our case, rapid diagnosis based on clinical signs, broad-spectrum empirical antibiotics and aggressive surgical approach with debridement and tissue sampling for microbiological identification of an etiological agent which guided our pharmacotherapy led to final therapeutic success.
In conclusion, the successful treatment of FG depends on early diagnosis and aggressive etiological management. Therapeutic strategy includes hemodynamic stabilization, broad-spectrum antibiotics and wide surgical debridement with maximum excision of all necrotic tissues. General increase in fungal infections and the incidence of fungal colonization worldwide may suggest that empiric antifungal treatment should also be considered in FG.
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Conflict of interest
Dr. Jaworski reports and has become personal fee for lecture from MSD Polska, Dr. Irga-Jaworska reports and has become personal fee for lecture from MSD Polska, Dr. Haponiuk has nothing to disclose, Dr. Naumiuk has nothing to disclose, and Dr. Chojnicki has nothing to disclose.
Informed consent from the patient’s parents for the publication of this case report is provided. Informed consent for the publication of all patient’s pictures from the patient’s parents is provided.
- 9.Pais VM. Fournier gangrene. http://emedicine.medscape.com/article/2028899-overview#a7. Accessed 1 Apr 2016.
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