REVIEW Pediatric Dermatology Vol. 31 No. 3 267–270, 2014

Cutaneous Fungal Infection in a Neonatal Intensive Care Unit Patient: A Case Report and Literature Review Vesta Kucinskiene, M.D., Ph.D., Auguste Sutkute, M.D., and Skaidra Valiukeviciene, M.D., Ph.D. Department of Skin and Venereal Diseases, Lithuanian University of Health Sciences, Kaunas, Lithuania

Abstract: Fungal skin infections are not uncommon in healthy, premature or immunocompromised newborns. Healthy neonates usually develop fungal skin infections caused by dermatophytes, Candida and Malassezia species, whereas immunocompromised neonates are more susceptible to skin infections with opportunistic pathogens (Aspergilus, Zygomycetes). Therefore neonatal fungal skin infections can range from generally benign superficial lesions to potentially fatal, deep, necrotic forms with dissemination. We present the case of a premature neonate twin with cutaneous fungal infection in a neonatal intensive care unit. Because there were doubts concerning the correspondence of the clinical features with the cultured species in the newborn, a literature review was performed searching for similar clinical cases.

CASE REPORT Female monozygotic twins (0.590 and 0.584 kg) born by vaginal delivery to a 35-year-old mother at the 24th week of gestation after premature rupture of the membranes were hospitalized in a neonatal intensive care unit (NICU). The twins were placed into one neonatal incubator and vital signs were monitored (Fig. 1). Mechanical ventilation, tube feeding, and antibacterial and oxygen therapy were applied. Fourteen days later the second of the twins developed a widespread skin eruption, whereas the

first twin was not affected. Physical examination revealed a symmetrical, annular, erythematosquamous eruption with a scaling edge on the trunk and the upper legs (Fig. 2). Clinically the lesions resembled tinea corporis. Potassium hydroxide (KOH) examination of skin scrapings was positive for hyphae and Syncephalastrum spp. (Zygomycetes class) was cultured after 2 weeks. Clotrimazole cream 1% was applied twice a day to the affected areas of skin along with intravenous fluconazole 4 mg/kg for 2 weeks. After 4 weeks of treatment, the skin lesions cleared, but after several days, they

Address correspondence to Vesta Kucinskiene, M.D., Ph.D., Department of Skin and Venereal Diseases, Lithuanian University of Health Sciences, Eiveniu 2, LT-50009 Kaunas, Lithuania, or e-mail: DOI: 10.1111/pde.12323

© 2014 Wiley Periodicals, Inc.


268 Pediatric Dermatology Vol. 31 No. 3 May/June 2014

Figure 1. The premature neonate twins in the neonatal intensive care unit.

Figure 3. After successful treatment.


Figure 2. A symmetrical, annular, erythematosquamous eruption with scaling edges on the trunk and upper legs.

recurred on the trunk, and 1% clotrimazole cream without systemic antifungal therapy was repeated for 2 weeks. No further skin lesions developed (Fig. 3). To achieve successful antifungal therapy, a search for a possible source of the fungal infection was required. Clinical examination of the parents did not reveal any skin eruption. Screening of the medical staff by questioning was also negative. A more detailed medical history revealed a maternal fungal skin infection during the second trimester of pregnancy; KOH examination had been positive for hyphae, but the culture test was not performed. The mother had been treated with 1% clotrimazole cream twice a day on the affected skin for 2 weeks. The family pet had also had a cutaneous fungal infection that a veterinary doctor cured. Contact between the mother and child was discontinued to prevent possible infection.

Zygomycosis is a term used to describe diseases caused by the members of the class Zygomycetes, which contains the Mucorales and the Entomophthorales orders. Mucorales usually causes human zygomycosis and is commonly linked to Rhizopus, although an association with other organisms, including Syncephalastrum, is also possible (1–4). Syncephalastrum is a saprophytic organism that can be isolated from environmental sources worldwide, although it is found mainly in the soil and dung in tropical and subtropical regions (5). It is a potent human pathogen and can also be a laboratory contaminant. Zygomycetes are associated with various underlying immunosuppressive conditions, and an increasing frequency of fungal skin infections has been reported in premature, low-birthweight, or otherwise immunocompromised neonates (1,4, 6–8). Various predisposing factors increase the risk of neonatal fungal infection in the NICU, including mechanical ventilation, artificial nutrition, antibacterial therapy, catheters, dressings, and iatrogenic skin trauma (6,9–12). Clinically zygomycosis may present as rhino-orbito-cerebral, pulmonary, cutaneous, gastrointestinal, or miscellaneous forms (13), with the major routes of transmission being inhalation, ingestion, and cutaneous exposure (5). Cutaneous forms may be gradually progressive or fulminant and aggressive, leading to necrosis and dissemination (13). In our case, the lesions clinically did not match cutaneous zygomycosis reported in the literature. The twin developed superficial skin lesions without systemic dissemination, which is specific to human zygomycotic infection invading the inner organs, most commonly the lungs. Treatment with combined

Sutkute et al: Cutaneous Fungal Infection in a Neonatal Patient 269

TABLE 1. Review of Published Clinical Cases with Syncephalastrum Patient characteristics

Immune state and predisposing factors

Kamalam et al (15)

Adult man

Schlebusch et al (16)

23-year-old man

Pavlovic (17)

45-year-old man

Baradkar et al (18)

45-year-old man

Chronic hepatitis B infection and cirrhosis of liver

Amatya et al (19)

30-year-old woman

Kumar (20)

39-year-old man

Immunocompetent, nonpregnant; trauma 6 months before presentation (bamboo stick accidentally pricked foot while working in fields) Immunocompetent


Clinical manifestation



Diabetic ketosis

Skin infection of the thumb


Immunocompetent; trauma (falling and being impaled on a steel reinforcing rod) Immunocompetent; nail injury 7 months before consultation

Large abdominal wound

Partial surgical debridement and lipid-associated amphotericin B Surgical extirpation of nail plate and nystatin ointment twice a day for 2 weeks for exposed toenail bed Partial surgical debridement and liposomal amphotericin B ND

Died from complications of diabetes mellitus without associated systemic mycosis Successfully treated

Discolored, thickened right great toenail

Nasal pain; swelling, watering, and congestion of the left eye; bilateral bleeding from nose Multiple discharging lesions over the dorsum of the left foot

Left flank pain, intermittent high-grade fever, passage of whitish flakes in urine

Endoscopic removal of fungus and antifungal medication

Remained disease free for 4 years

Successfully treated Lost to follow-up

Doing well at 1-, 3-, and 6-month follow-up evaluations

ND, no data.

intravenous fluconazole and topical clotrimazole was successful in our case. The combination of intravenous amphotericin B and surgical debridement is the usual treatment of cutaneous zygomycosis, and azoles have no role in the management of zygomycosis (2,7,13). Inappropriate or delayed treatment in neonatal mucormycosis is associated with high mortality, so prompt recognition and appropriate treatment of cutaneous fungal disease in neonates is essential (6). Well-documented cases associated with Syncephalastrum infection are scarce. We reviewed the literature for human diseases caused by Syncephalastrum and only six proven cases in adults were found (Table 1) (15–20). In our case, Syncephalastrum was cultured in the laboratory from the skin lesions, although we consider this fungus to be a contaminant because clinical findings and treatment of Syncephalastrum infection did not point to the invasive disease mentioned in the previously published cases. In our case, the infection was clinically superficial and classical antifungal therapy was sufficient and successful. Also, the twin may have

been infected with dermatophytes, and an interruption of contact with the mother helped to stop the spread of the infection.

REFERENCES 1. Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13:236– 301. 2. Skiada A, Petrikkos G. Cutaneous zygomycosis. Clin Microbiol Infect 2009;15(Suppl 5):41–45. 3. Zaoutis TE, Roilides E, Chiou CC et al. Zygomycosis in children: a systematic review and analysis of reported cases. Pediatr Infect Dis J 2007;26:723–727. 4. Dehority W, Willert J, Pong A. Zygomycetes infections in pediatric hematology oncology patients: a case series and review of the literature. J Pediatr Hematol Oncol 2009;31:911–919. 5. Chayakulkeeree M, Ghannoum MA, Perfect JR. Zygomycosis: the re-emerging fungal infection. Eur J Clin Microbiol Infect Dis 2006;25:215–229. 6. Smolinski KN, Shah SS, Honig PJ et al. Neonatal cutaneous fungal infections. Curr Opin Pediatr 2005;17:486–493.

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7. Roilides E, Zaoutis TE, Walsh TJ. Invasive zygomycosis in neonates and children. Clin Microbiol Infect 2009;15(Suppl 5):50–54. 8. Kaufman D. Fungal infection in the very low birthweight infant. Curr Opin Infect Dis 2004;17:253–259. 9. El-Masry FAY, Neal TJ, Subhedar NV. Risk factors for invasive fungal infection in neonates. Acta Paediatr 2002;91:198–202. 10. Robertson AF, Joshi VV, Ellison DA et al. Zygomycosis in neonates. Pediatr Infect Dis J 1997;16: 812–815. 11. Maheshwari A, Stromquist CI, Pereda L et al. Mixed infection with unusual fungi and staphylococcal species in two extremely premature neonates. J Perinatol 2004;24:324–326. 12. Mulholland A, Casey T, Cartwright D. Microsporum canis in a neonatal intensive care unit patient. Australas J Dermatol 2008;49:25–26. 13. Chakrabarti A. Cutaneous zygomycosis: major concerns. Indian J Med Res 2010;131:739–741. 14. Inoue S, Odaka A, Hashimoto D et al. Rare case of disseminated neonatal zygomycosis mimicking

15. 16.

17. 18.

19. 20.

necrotizing enterocolitis with necrotizing fasciitis. J Pediatr Surg 2011;46:E29–E32. Kamalam A, Thambiah AS. Cutaneous infection by Syncephalastrum. Sabouradia 1980;18:19–20. Schlebusch S, Looke DF. Intraabdominal zygomycosis caused by Syncephalastrum racemosum infection successfully treated with partial surgical debridement and high dose amphotericin B lipid complex. J Clin Microbiol 2005;43:5825–5827. Pavlovic MD, Bulajic N. Great toenail onychomycosis caused by Syncephalastrum racemosum. Dermatol Online J 2006;12:7. Baradkar VP, Mathur M, Panda M et al. Sino-orbital infection by Syncephalastrum racemosum in chronic hepatorenal disease. J Oral Maxillofac Pathol 2008; 12:45–47. Amatya R, Khanal B, Rijal A. Syncephalastrum species producing mycetoma-like lesions. Indian J Dermatol Venereol Leprol 2010;76:284–286. Kumar S. Isolated renal zygomycosis caused by Syncephalastrum species in an immunocompetent host: a case report. Uro Today Int J 2010; 3:1–3.

Cutaneous fungal infection in a neonatal intensive care unit patient: a case report and literature review.

Fungal skin infections are not uncommon in healthy, premature or immunocompromised newborns. Healthy neonates usually develop fungal skin infections c...
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