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DOI: 10.1055/s-0029-1220775
Zygomycosis in Neonates: An Uncommon but Life-threatening Infection
Publication History
Publication Date:
23 April 2009 (online)
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ABSTRACT
We systematically reviewed all published cases of zygomycosis, an increasingly important infection with high mortality, in neonates. We searched PubMed and individual references for English publications of single cases or case series of neonatal (0 to 1 month) zygomycosis. Cases were included if they fulfilled prespecified criteria. Fifty-nine cases were published through July 2007. Most of the infants (77%) were premature. The most common sites of zygomycosis were gastrointestinal (54%) and cutaneous (36%) diseases. This pattern differs from sinopulmonary and rhinocerebral patterns of older children. Fifty-six percent of cases were diagnosed by histology only and 44% by histology and culture. Rhizopus spp. were isolated from 18/25 (72%) cases. Thirty-seven percent of patients received no antifungal therapy. Thirty-two (54%) neonates underwent surgery with (39%) or without (15%) antifungal agents. Overall mortality was 64%. A higher fraction of neonates treated with amphotericin B and surgery survived than those who received no therapy (70% versus 5%). Zygomycosis is a life-threatening infection in neonates with a distinct pattern of gastrointestinal and cutaneous involvement and high mortality. Combination of amphotericin B and surgery was common management strategy in survivors.
KEYWORDS
Epidemiology - mucormycosis - outcome - Rhizopus spp. - gastrointestinal infection
REFERENCES
- 1 Levy O. Innate immunity of the newborn: basic mechanisms and clinical correlates. Nat Rev Immunol. 2007; 7 379-390
- 2 Marodi L, Johnston Jr R B. Invasive Candida species disease in infants and children: occurrence, risk factors, management, and innate host defense mechanisms. Curr Opin Pediatr. 2007; 19 693-697
- 3 Groll A H, Jaeger G, Allendorf A et al.. Invasive pulmonary aspergillosis in a critically ill neonate: case report and review of invasive aspergillosis during the first 3 months of life. , [see comments] Clin Infect Dis. 1998; 27 437-452
- 4 Ribes J A, Vanover-Sams C L, Baker D J. Zygomycetes in human disease. Clin Microbiol Rev. 2000; 13 236-301
- 5 Kontoyiannis D P, Lionakis M S, Lewis R E et al.. Zygomycosis in a tertiary-care cancer center in the era of Aspergillus-active antifungal therapy: a case-control observational study of 27 recent cases. J Infect Dis. 2005; 191 1350-1360
- 6 Robertson A F, Joshi V V, Ellison D A, Cedars J C. Zygomycosis in neonates. Pediatr Infect Dis J. 1997; 16 812-815
- 7 Diven S C, Angel C A, Hawkins H K, Rowen J L, Shattuck K E. Intestinal zygomycosis due to Absidia corymbifera mimicking necrotizing enterocolitis in a preterm neonate. J Perinatol. 2004; 24 794-796
- 8 Morales-Aguirre J J, Aguero-Echeverria W M, Ornelas-Carsolio M E et al.. Successful treatment of a primary cutaneous zygomycosis caused by Absidia corymbifera in a premature newborn. Pediatr Infect Dis J. 2004; 23 470-472
- 9 Zaoutis T E, Roilides E, Chiou C C et al.. Zygomycosis in children: a systematic review and analysis of reported cases. Pediatr Infect Dis J. 2007; 26 723-727
- 10 Roden M M, Zaoutis T E, Buchanan W L et al.. Epidemiology and outcome of zygomycosis: a review of 929 reported cases. Clin Infect Dis. 2005; 41 634-653
- 11 Mooney J E, Wanger A. Mucormycosis of the gastrointestinal tract in children: report of a case and review of the literature. Pediatr Infect Dis J. 1993; 12 872-876
- 12 Amin S B, Ryan R M, Metlay L A, Watson W J. Absidia corymbifera infections in neonates. Clin Infect Dis. 1998; 26 990-992
- 13 Lewis L L, Hawkins H K, Edwards M S. Disseminated mucormycosis in an infant with methylmalonicaciduria. Pediatr Infect Dis J. 1990; 9 851-854
- 14 Craig N M, Lueder F L, Pensler J M et al.. Disseminated Rhizopus infection in a premature infant. Pediatr Dermatol. 1994; 11 346-350
- 15 Ng P C, Dear P R. Phycomycotic abscesses in a preterm infant. Arch Dis Child. 1989; 64 862-864
- 16 Mitchell S J, Gray J, Morgan M E, Hocking M D, Durbin G M. Nosocomial infection with Rhizopus microsporus in preterm infants: association with wooden tongue depressors. Lancet. 1996; 348 441-443
- 17 Hughes C, Driver S J, Alexander K A. Successful treatment of abdominal wall Rhizopus necrotizing cellulitis in a preterm infant. Pediatr Infect Dis J. 1995; 14 336
- 18 Reimund E, Ramos A. Disseminated neonatal gastrointestinal mucormycosis: a case report and review of the literature. Pediatr Pathol. 1994; 14 385-389
- 19 Linder N, Keller N, Huri C et al.. Primary cutaneous mucormycosis in a premature infant: case report and review of the literature. Am J Perinatol. 1998; 15 35-38
- 20 Woodward A, McTigue C, Hogg G, Watkins A, Tan H. Mucormycosis of the neonatal gut: a “new” disease or a variant of necrotizing enterocolitis?. J Pediatr Surg. 1992; 27 737-740
- 21 Parfrey N A. Improved diagnosis and prognosis of mucormycosis. A clinicopathologic study of 33 cases. Medicine (Baltimore). 1986; 65 113-123
- 22 Shah A, Lagvankar S, Shah A. Cutaneous mucormycosis in children. Indian Pediatr. 2006; 43 167-170
- 23 Kumar V, Aggarwal A, Taneja R et al.. Primary cutaneous mucormycosis in a premature neonate and its management by tumescent skin grafting. Br J Plast Surg. 2005; 58 852-854
- 24 Alexander P, Alladi A, Correa M, D'Cruz A J. Neonatal colonic mucormycosis—a tropical perspective. J Trop Pediatr. 2005; 51 54-59
- 25 Nichol P F, Corliss R F, Rajpal S, Helin M, Lund D P. Perforation of the appendix from intestinal mucormycosis in a neonate. J Pediatr Surg. 2004; 39 1133-1135
- 26 Siu K L, Lee W H. A rare cause of intestinal perforation in an extreme low birth weight infant—gastrointestinal mucormycosis: a case report. J Perinatol. 2004; 24 319-321
- 27 Scheffler E, Miller G G, Classen D A. Zygomycotic infection of the neonatal upper extremity. J Pediatr Surg. 2003; 38 E16-E17
- 28 Buchta V, Kalous P, Otcenasek M, Vanova M. Primary cutaneous Absidia corymbifera infection in a premature newborn. Infection. 2003; 31 57-59
- 29 Oh D, Notrica D. Primary cutaneous mucormycosis in infants and neonates: case report and review of the literature. J Pediatr Surg. 2002; 37 1607-1611
- 30 Sridhar S, Jana A K, Thomas S, Solomon R. Mucormycosis of the neonatal gastrointestinal tract. Indian Pediatr. 2001; 38 294-297
- 31 Nissen M D, Jana A K, Cole M J, Grierson J M, Gilbert G L. Neonatal gastrointestinal mucormycosis mimicking necrotizing enterocolitis. Acta Paediatr. 1999; 88 1290-1293
- 32 Sharma M C, Gill S S, Kashyap S et al.. Gastrointestinal mucormycosis—an uncommon isolated mucormycosis. Indian J Gastroenterol. 1998; 17 131-133
- 33 Kecskes S, Reynolds G, Bennett G. Survival after gastrointestinal mucormycosis in a neonate. J Paediatr Child Health. 1997; 33 356-359
- 34 Rubin S A, Chaljub G, Winer-Muram H T, Flicker S. Pulmonary zygomycosis: a radiographic and clinical spectrum. J Thorac Imaging. 1992; 7 85-90
- 35 Miller R D, Steinkuller P G, Naegele D. Nonfatal maxillocerebral mucormycosis with orbital involvement in a dehydrated infant. Ann Ophthalmol. 1980; 12 1065-1068
- 36 Agarwal K, Sharma M, Singh S, Jain M. Antemortem diagnosis of gastrointestinal mucormycosis in neonates: report of two cases and review of literature. Indian J Pathol Microbiol. 2006; 49 430-432
- 37 Dennis J E, Rhodes K H, Cooney D R, Roberts G D. Nosocomial Rhizopus infection (zygomycosis) in children. J Pediatr. 1980; 96 824-828
- 38 Vadeboncoeur C, Walton J M, Raisen J et al.. Gastrointestinal mucormycosis causing an acute abdomen in the immunocompromised pediatric patient—three cases. J Pediatr Surg. 1994; 29 1248-1249
- 39 Michalak D M, Cooney D R, Rhodes K H, Telander R L, Kleinberg F. Gastrointestinal mucormycoses in infants and children: a cause of gangrenous intestinal cellulitis and perforation. J Pediatr Surg. 1980; 15 320-324
- 40 du Plessis P J, Wentzel L F, Delport S D, van Damme E. Zygomycotic necrotizing cellulitis in a premature infant. Dermatology. 1997; 195 179-181
- 41 Isaacson C, Levin S E. Gastro-intestinal mucormycosis in infancy. S Afr Med J. 1961; 35 581-584
- 42 Varricchio F, Reyes M G, Wilks A. Undiagnosed mucormycosis in infants. Pediatr Infect Dis J. 1989; 8 660
- 43 Grim III P F, Demello D, Keenan W J. Disseminated zygomycosis in a newborn. Pediatr Infect Dis. 1984; 3 61-63
- 44 Hadley G P. Neonatal mucormycosis. P N G Med J. 1981; 24 54-56
- 45 Budhiraja S, Sood N, Singla S, Gupta V. Gastrointestinal mucormycosis in a neonate: survival without antifungal therapy. Indian J Gastroenterol. 2007; 26 198
- 46 Kataria R, Gupta D K, Mitra D K. Colonic mucormycosis in the neonate. Pediatr Surg Int. 1995; 10 271-273
- 47 Pena C E. Deep mycotic infections in Colombia. A clinicopathologic study of 162 cases. Am J Clin Pathol. 1967; 47 505-520
- 48 Levin S E, Isaacson C. Spontaneous perforation of the colon in the newborn infant. Arch Dis Child. 1960; 35 378-382
- 49 Neame P, Rayner D. Mucormycosis. A report on twenty-two cases. Arch Pathol. 1960; 70 261-268
- 50 Gatling R R. Gastric mucormycosis in a newborn infant; a case report. AMA Arch Pathol. 1959; 67 249-255
- 51 Keye Jr J D, Magee W E. Fungal diseases in a general hospital; a study of 88 patients. Am J Clin Pathol. 1956; 26 1235-1253
- 52 Santulli T V, Schullinger J N, Heird W C et al.. Acute necrotizing enterocolitis in infancy: a review of 64 cases. Pediatrics. 1975; 55 376-387
- 53 Starke J R, Mason Jr E O, Kramer W G, Kaplan S L. Pharmacokinetics of amphotericin B in infants and children. J Infect Dis. 1987; 155 766-774
- 54 Kwon D S, Mylonakis E. Posaconazole: a new broad-spectrum antifungal agent. Expert Opin Pharmacother. 2007; 8 1167-1178
- 55 U.S. Food and Drug Administration. Noxafil® product information (posaconazole). Available at: http://www.fda.gov/cder/foi/label/2006/022003lbl.pdf
- 56 de Repentigny L, St-Germain G, Charest H, Kokta V, Vobecky S. Fatal zygomycosis caused by Mucor indicus in a child with an implantable left ventricular assist device. Pediatr Infect Dis J. 2008; 27 365-369
- 57 Kefala-Agoropoulou K, Farmaki E, Tsiouris J, Roilides E, Velegraki A. Cutaneous zygomycosis in an infant with Pearson syndrome. Pediatr Blood Cancer. 2008; 50 939-940
Emmanuel RoilidesM.D. Ph.D.
3rd Department of Pediatrics, Aristotle University School of Medicine, Hippokration Hospital
Konstantinoupoleos 49, GR-546 42 Thessaloniki, Greece
Email: roilides@med.auth.gr