J Korean Assoc Oral Maxillofac Surg.  2016 Apr;42(2):105-110. 10.5125/jkaoms.2016.42.2.105.

Oral mucormycosis in an 18-month-old child: a rare case report with a literature review

• Kalaskar R
• Kalaskar A
• Ganvir S

• KMID: 2351733
• DOI: http://doi.org/10.5125/jkaoms.2016.42.2.105

Abstract

Oral mucormycosis is a fungal infection observed mainly in elderly immunocompromised patients. In rare instances, the disease occurs in healthy individuals and those patients that are below preschool age. Although this condition mainly involves the maxilla, it may also manifest in any part of the oral cavity based on the source of infection. Mucormycosis of the maxilla spreads rapidly, leading to necrosis of the palatal bone and palatal perforation. Such patients are usually rehabilitated using bone grafting or free flap surgeries. However, when surgeries are delayed, palatal prosthesis is an interim treatment modality that can prevent nasal regurgitation and aspiration of food or fluids. Palatal prostheses also help with mastication, speech, and swallowing. The present case describes a rare case of oral mucormycosis in an 18-month-old male involving the maxilla that was managed by palatal prosthesis.

Keyword

Immunocompromised; Maxilla; Mucormycosis; Oronasal fistula; Palatal obturator

MeSH Terms

Aged
Bone Transplantation
Child*
Deglutition
Free Tissue Flaps
Humans
Immunocompromised Host
Infant*
Male
Mastication
Maxilla
Mouth
Mucormycosis*
Necrosis
Palatal Obturators
Prostheses and Implants

Figure

• 

  Fig. 1 Intraoral photograph shows exposed necrotic bone.

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  Fig. 2 Occlusal radiograph of the maxilla shows diffuse radiolucency in the mid-palatine region.

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  Fig. 3 Histological image of mucormycosois (Gram staining, ×40).

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  Fig. 4 Extra oral photograph of the fabricated palatal prosthesis.

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  Fig. 5 Diagrammatic representation of the palatal prosthesis.

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  Fig. 6 Palatal prosthesis in place.

Reference

1. Rosenberg SW, Lepley JB. Mucormycosis in leukemia. Oral Surg Oral Med Oral Pathol. 1982; 54:26–32. PMID: 6956824.

2. Abramson E, Wilson D, Arky RA. Rhinocerebral phycomycosis in association with diabetic ketoacidosis. Report of two cases and a review of clinical and experimental experience with amphotericin B therapy. Ann Intern Med. 1967; 66:735–742. PMID: 4960691.

3. Paltauf A. Mycosis mucorina. Virchows Arch Pathol Anat. 1885; 102:543–564.

4. Lehrer RI, Howard DH, Sypherd PS, Edwards JE, Segal GP, Winston DJ. Mucormycosis. Ann Intern Med. 1980; 93:93–108. PMID: 6772062.

5. Taylor CG, Alexander RE, Green WH, Kramer HS Jr. Mucormycosis (phycomycosis) involving the maxilla. Report of a case with survival. Oral Surg Oral Med Oral Pathol. 1969; 27:806–822. PMID: 5254571.

6. Jones AC, Bentsen TY, Freedman PD. Mucormycosis of the oral cavity. Oral Surg Oral Med Oral Pathol. 1993; 75:455–460. PMID: 8464609.

7. Weprin BE, Hall WA, Goodman J, Adams GL. Long-term survival in rhinocerebral mucormycosis. Case report. J Neurosurg. 1998; 88:570–575. PMID: 9488314.

8. Bonifaz A, Macias B, Paredes-Farrera F, Arias P, Ponce RM, Araiza J. Palatal zygomycosis: experience of 21 cases. Oral Dis. 2008; 14:569–574. PMID: 18248590.

9. Deboni MC, Pozzani VR, Lisboa T, Hiraki K, Viplich R, Naclério-Homem MG. Mucormycosis in an immunocompetent patient: follow-up of 1 year after treatment. Acta Otolaryngol. 2006; 126:993–996. PMID: 16864500.

10. Aras MH, Kara MI, Erkiliç S, Ay S. Mandibular mucormycosis in immunocompromised patients: report of 2 cases and review of the literature. J Oral Maxillofac Surg. 2012; 70:1362–1368. PMID: 21820787.

11. Samanta DR, Senapati SN, Sharma PK, Shruthi BS, Paty PB, Sarangi G. Hard palate perforation in acute lymphoblastic leukemia due to mucormycosis: a case report. Indian J Hematol Blood Transfus. 2009; 25:36–39. PMID: 23100971.

12. McNulty JS. Rhinocerebral mucormycosis: predisposing factors. Laryngoscope. 1982; 92:1140–1143. PMID: 7132514.

13. Eisenberg L, Wood T, Boles R. Mucormycosis. Laryngoscope. 1977; 87:347–356. PMID: 839930.

14. Leitner C, Hoffmann J, Zerfowski M, Reinert S. Mucormycosis: necrotizing soft tissue lesion of the face. J Oral Maxillofac Surg. 2003; 61:1354–1358. PMID: 14613095.

15. Pogrel MA, Miller CE. A case of maxillary necrosis. J Oral Maxillofac Surg. 2003; 61:489–493. PMID: 12684969.

16. Huang JS, Kok SH, Lee JJ, Hsu WY, Chiang CP, Kuo YS. Extensive maxillary sequestration resulting from mucormycosis. Br J Oral Maxillofac Surg. 2005; 43:532–534. PMID: 16024140.

17. Fogarty C, Regennitter F, Viozzi CF. Invasive fungal infection of the maxilla following dental extractions in a patient with chronic obstructive pulmonary disease. J Can Dent Assoc. 2006; 72:149–152. PMID: 16545177.

18. Kumar JA, Babu P, Prabu K, Kumar P. Mucormycosis in maxilla: Rehabilitation of facial defects using interim removable prostheses: a clinical case report. J Pharm Bioallied Sci. 2013; 5(Suppl 2):S163–S165. PMID: 23956598.

19. Bakathir AA. Mucormycosis of the jaw after dental extractions: two case reports. Sultan Qaboos Univ Med J. 2006; 6:77–82. PMID: 21748139.

20. Zlotolow IM. Dental oncology and maxillofacial prosthtetics. In : Shah JP, editor. Cancer of head and neck: atlas of clinical oncology. Hamilton: BC Decker Inc.;2001. p. 373–376.

21. Tanaka H. Speech patterns of edentulous patients and morphology of the palate in relation to phonetics. J Prosthet Dent. 1973; 29:16–28. PMID: 4508620.

22. Shah RJ, Katyayan MK, Katyayan PA, Chauhan V. Prosthetic rehabilitation of acquired maxillary defects secondary to mucormycosis: clinical cases. J Contemp Dent Pract. 2014; 15:242–249. PMID: 25095851.

23. Gowda ME, Mohan MS, Verma K, Roy ID. Implant rehabilitation of partial maxillectomy edentulous patient. Contemp Clin Dent. 2013; 4:393–396. PMID: 24124314.

24. Genden EM, Okay D, Stepp MT, Rezaee RP, Mojica JS, Buchbinder D, et al. Comparison of functional and quality-of-life outcomes in patients with and without palatomaxillary reconstruction: a preliminary report. Arch Otolaryngol Head Neck Surg. 2003; 129:775–780. PMID: 12874081.

25. Owotade FJ, Ugboko VI, Kolude B. Herpes zoster infection of the maxilla: case report. J Oral Maxillofac Surg. 1999; 57:1249–1251. PMID: 10513873.

26. Hall HD, Jacobs JS, O'Malley JP. Necrosis of maxilla in patient with herpes zoster. Report of a case. Oral Surg Oral Med Oral Pathol. 1974; 37:657–662. PMID: 4524373.

27. Barrett AP, Katelaris CH, Morris JG, Schifter M. Zoster sine herpete of the trigeminal nerve. Oral Surg Oral Med Oral Pathol. 1993; 75:173–175. PMID: 8381216.

28. Weatherall DJ, Ledingham JGG, Warrell DA. Oxford teatbook of medicine. 3rd ed. Oxford: Oxford University Press;1996. p. 197–221.

29. Mintz SM, Anavi Y. Maxillary osteomyelitis and spontaneous tooth exfoliation after herpes zoster. Oral Surg Oral Med Oral Pathol. 1992; 73:664–666. PMID: 1437032.

30. Cooper JC. Tooth exfoliation and osteonecrosis of the jaw following herpes zoster. Br Dent J. 1977; 143:297–300. PMID: 270353.

31. Nandakumar H, Shankaramba KB. Massive sequestration of the upper jaw: a case report. Br J Oral Maxillofac Surg. 1990; 28:55–56. PMID: 2182111.

32. Garg R, Schalch P, Pepper JP, Nguyen QA. Osteomyelitis of the hard palate secondary to actinomycosis: a case report. Ear Nose Throat J. 2011; 90:E11–E12. PMID: 21412725.

33. Finley AM, Beeson MS. Actinomycosis osteomylelitis of the mandible. Am J Emerg Med. 2010; 28:118.e1–118.e4. PMID: 20006231.

34. Manjunatha BS, Das N, Sutariya RV, Ahmed T. Mucormycosis of the hard palate masquerading as carcinoma. Clin Pract. 2012; 2:e28. PMID: 24765427.

35. Verma A, Singh V, Jindal N, Yadav S. Necrosis of maxilla, nasal, and frontal bone secondary to extensive rhino-cerebral mucormycosis. Natl J Maxillofac Surg. 2013; 4:249–251. PMID: 24665188.