1887

Abstract

Introduction:

Mucormycosis is a rare fungal infection, with high morbidity and mortality. Palatal ulceration may suggest a number of differential diagnoses, one of which is rhinocerebral/craniofacial mucormycosis and for which it may be the first presenting clinical finding. We report a case of sinus mucormycosis in a patient with chronic myelomonocytic leukaemia‐2 (CMML‐2), now classified in the myelodysplastic/myeloproliferative neoplasms, presenting with dental pain and palatal ulcer.

Case presentation:

A 72‐year‐old female with CMML‐2 presented with pain of the left maxillary molar and a dark‐brown necrotic ulcer with a white irregular border on the hard palate. Invasive fungal infection was included in the differential diagnosis. Computerized tomography disclosed inflammatory lesions in the left nasal, ethmoid and frontal sinuses. Histological examination of the ulcer showed fungal hyphae typical of agents of mucormycosis. was isolated from the culture. Liposomal amphotericin B was introduced, combined with haematological support and maxillectomy. Mucormycosis was controlled, but the patient died of progressive acute myeloid leukaemia and multiple bacteraemias. A literature review of rhinocerebral mucormycosis with palatal involvement disclosed 109 cases; palatal involvement was present among other presenting signs in 34 patients and as the presenting sign leading to diagnosis in nine cases, including the present case. Six of the nine patients (66.6 %) survived the infection, compared with 43 of 101 (42.6 %) with other signs at presentation.

Conclusion:

Palatal ulcer may represent an early sign of sinus mucormycosis. Awareness by healthcare professionals is critical for the prompt diagnosis of this rapidly developing and life‐threatening infection.

Loading

Article metrics loading...

/content/journal/jmmcr/10.1099/jmmcr.0.000014
2015-02-01
2019-09-21
Loading full text...

Full text loading...

/deliver/fulltext/jmmcr/2/1/jmmcr000014.html?itemId=/content/journal/jmmcr/10.1099/jmmcr.0.000014&mimeType=html&fmt=ahah

References

  1. Arce‐Salinas C.A. , Perez‐Silva E. . ( 2010; ). Mucormycosis complications in systemic lupus erythematosus. . Lupus 19:, 985–988.[CrossRef]
    [Google Scholar]
  2. Auluck A. . ( 2007; ). Maxillary necrosis by mucormycosis. A case report and literature review. . Med Oral Patol Oral Cir Bucal 12:, E360–E364.
    [Google Scholar]
  3. Ballester D.G. , Gonzalez‐Garcia R. , Garcia C.M. , Ruiz‐Laza L. , Gil F.M. . ( 2012; ). Mucormycosis of the head and neck: report of five cases with different presentations. . J Cranio‐Maxillo‐Facial Surg 40:, 584–591.[CrossRef]
    [Google Scholar]
  4. Barrak H.A. . ( 2007; ). Hard palate perforation due to mucormycosis: report of four cases. . J Laryngol Otol 121:, 1099–1102.[CrossRef]
    [Google Scholar]
  5. Bitar D. , Van Cauteren D. , Lanternier F. , Dannaoui E. , Che D. , Dromer F. , Desenclos J.C. , Lortholary O. . ( 2009; ). Increasing incidence of zygomycosis (mucormycosis), France, 1997–2006. . Emerg Infect Dis 15:, 1395–1401.[CrossRef]
    [Google Scholar]
  6. Bonifaz A. , Macias B. , Paredes‐Farrera F. , Arias P. , Ponce R.M. , Araiza J. . ( 2008; ). Palatal zygomycosis: experience of 21 cases. . Oral Dis 14:, 569–574.[CrossRef]
    [Google Scholar]
  7. Chamilos G. , Lewis R.E. , Kontoyiannis D.P. . ( 2008; ). Delaying amphotericin B‐based frontline therapy significantly increases mortality among patients with hematologic malignancy who have zygomycosis. . Clin Infect Dis 47:, 503–509.[CrossRef]
    [Google Scholar]
  8. Cornely O.A. , Arikan‐Akdagli S. , Dannaoui E. , Groll A.H. , Lagrou K. , Chakrabarti A. , Lanternier F. , Pagano L. , Skiada A. . other authors ( 2014; ). ESCMID and ECMM joint clinical guidelines for the diagnosis and management of mucormycosis 2013. . Clin Microbiol Infect 20 (Suppl. 3):, 5–26.[CrossRef]
    [Google Scholar]
  9. Doni B.R. , Peerapur B.V. , Thotappa L.H. , Hippargi S.B. . ( 2011; ). Sequence of oral manifestations in rhino‐maxillary mucormycosis. . Indian J Dent Res 22:, 331–335.[CrossRef]
    [Google Scholar]
  10. Gamaletsou M.N. , Sipsas N.V. , Roilides E. , Walsh T.J. . ( 2012; ). Rhino‐orbital‐cerebral mucormycosis. . Curr Infect Dis Rep 14:, 423–434.[CrossRef]
    [Google Scholar]
  11. Garg R. , Gupta V.V. , Ashok L. . ( 2011; ). Rhinomaxillary mucormycosis: a palatal ulcer. . Contemp Clin Dent 2:, 119–123.[CrossRef]
    [Google Scholar]
  12. Garlapati K. , Chavva S. , Vaddeswarupu R.M. , Surampudi J. . ( 2014; ). Fulminant mucormycosis involving paranasal sinuses: a rare case report. . Case Rep Dent 2014:, 465919.
    [Google Scholar]
  13. Ganesan P. , Swaroop C. , Ahuja A. , Thulkar S. , Bakhshi S. . ( 2009; ). Early onset sinonasal mucormycosis during induction therapy of acute lymphoblastiood outcome without surgical intervention. . J Pediatr Hematol Oncol 31:, 152–153.[CrossRef]
    [Google Scholar]
  14. Ibrahim A.S. , Spellberg B. , Edwards J. Jr . ( 2008; ). Iron acquisition: anovel perspective on mucormycosis pathogenesis and treatment. . Curr Opin Infect Dis 21:, 620–625.[CrossRef]
    [Google Scholar]
  15. Islam M.N. , Cohen D.M. , Celestina L.J. , Ojha J. , Claudio R. , Bhattachryya I.B. . ( 2007; ). Rhinocerebral zygomycosis: an increasingly frequent challenge: update and favorable outcomes in two cases. . Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104:, e28–e34.[CrossRef]
    [Google Scholar]
  16. Jayachandran S. , Krithika C. . ( 2006; ). Mucormycosis presenting as palatal perforation. . Phycomycetes 17:, 139–142.
    [Google Scholar]
  17. Karabulut A.B. , Kabakas F. , Berkoz O. , Karakas Z. , Kesim S.N. . ( 2005; ). Hard palate perforation due to invasive aspergillosis in a patient with acute lymphoblastic leukemia. . Int J Pediatr Otorhinolaryngol 69:, 1395–1398.[CrossRef]
    [Google Scholar]
  18. Ketenci I. , Unlu Y. , Kaya H. , Somdas M.A. , Kontas O. , Ozturk M. , Vural A. . ( 1984; ). Rhinocerebral mucormycosis: experience in 14 patients. . J Laryngol Otol 125:, e3.[CrossRef]
    [Google Scholar]
  19. Kim J. , Fortson J.F. , Cook H.E. . ( 2001; ). A fatal outcome from rhinocerebral mucormycosis after dental extractions: a case report. . J Oral Maxillofac Surg 59:, 693–697.[CrossRef]
    [Google Scholar]
  20. Kurosawa M. , Yonezumi M. , Hashino S. , Tanaka J. , Nishio M. , Kaneda M. , Ota S. , Koda K. , Suzuki N. . other authors ( 2012; ). Epidemiology and treatment outcome of invasive fungal infections in patients with hematological malignancies. . Int J Hematol 96:, 748–757.[CrossRef]
    [Google Scholar]
  21. Lanternier F. , Dannaoui E. , Morizot G. , Elie C. , Garcia‐Hermoso D. , Huerre M. , Bitar D. , Dromer F. , Lortholary O. . the French Mycosis Study Group ( 2012; ). A global analysis of mucormycosis in France: the RetroZygo study (2005–2007). . Clin Inf Dis 54:, (Suppl.1) S35–S43.[CrossRef]
    [Google Scholar]
  22. Leithauser M. , Kahl C. , Aepinus C. , Prall F. , Maruschke M. , Riemer H. , Wolff D. , Jost K. , Hilgendorf I. . other authors ( 2009; ). Invasive zygomycosis in patients with graft‐versus‐host disease after allogeneic stem cell transplantation. . Transplant Inf Dis 12:, 251–257.[CrossRef]
    [Google Scholar]
  23. Lewis R.E. , Liao G. , Wang W. , Prince R.A. , Kontoyiannis D.P. . ( 2011; ). Voriconazole pre‐exposure selects for breakthrough mucormycosis in a mixed model of Aspergillus fumigatusRhizopus oryzae pulmonary infection. . Virulence 2:, 348–355.[CrossRef]
    [Google Scholar]
  24. Llata E. , Blossom D.B. , Khoury H.J. , Rao C.Y. , Wannemuehler A. , Noble‐Wang J. , Langston A. , Ribner B.S. , Lyon G.M. . other authors ( 2011; ). A cluster of mucormycosis infections in hematology patients: challenges in investigation and control of invasive mold infections in high‐risk patient populations. . Diagn Microbiol Inf Dis 71:, 72–80.[CrossRef]
    [Google Scholar]
  25. Mane R.S. , Watve J.K. , Mohite A.A. , Patil B.C. . ( 2007; ). Rhinocerebral mucormycosis: a deadly disease on the rise. . Indian J Otolaryngol Head Neck Surg 59:, 112–115.[CrossRef]
    [Google Scholar]
  26. Manrique K. , Martin C. , Sanchez‐Vilar O. , Aragon C. , Rovira A. . ( 2013; ). Rhinomucormycosis and type 1 diabetes mellitus. . Endocrinol Nutr 60:, 149–151.[CrossRef]
    [Google Scholar]
  27. Metzen D. , Bohm H. , Zimmermann M. , Reuther T. , Kubler A.C. , Muller‐Richter U.D.A. . ( 2012; ). Mucormycosis of the head and neck. . J Cranio‐Maxillo‐Facial Surg 40:, e321–e327.[CrossRef]
    [Google Scholar]
  28. Mohindra S. , Gupta R. , Bakshi J. , Gupta S.K. . ( 2007; ). Rhinocerebral mucormycosis: the disease spectrum in 27 patients. . Mycoses 50:, 290–296.[CrossRef]
    [Google Scholar]
  29. Neville B.W. , Damm D.D. , Allen C.M. , Bouquot J.E. . ( 1997; ). Oral & Maxillofacial Pathology. Philadelphia, PA:: W. B. Saunders;.
    [Google Scholar]
  30. Nirmala S.V.S.G. , Lalitha V. , Sivakumar N. , Kumar K.K. , Srikanth M. . ( 2011; ). Mucormycosis associated with juvenile diabetes. . J Indian Soc Pedod Prev Dent 29:, S87–91.[CrossRef]
    [Google Scholar]
  31. Papadogeorgakis N. , Parara E. , Petsinis V. , Vourlakou C. . ( 2010; ). A case of successfully treated rhinocerebral mucormycosis: Dental implications. . Int J Dent 2010:, 273127.[CrossRef]
    [Google Scholar]
  32. Petrikkos G. , Skiada A. , Drogari‐Apiranthitou M. . ( 2014; ). Epidemiology of mucormycosis in Europe. . Clin Microbiol Infect 20:, 67–73.[CrossRef]
    [Google Scholar]
  33. Petrikkos G. , Skiada A. , Lortholary O. , Roilides E. , Walsh T.J. , Kontoyiannis D.P. . ( 2012; ). Epidemiology and clinical manifestations of mucormycosis. . Clin Inf Dis 54:, S23–S34.[CrossRef]
    [Google Scholar]
  34. Prasad K. , Lalitba R.M. , Reddy E.K. , Ranganatb K. , Srinivas D.R. , Singb J. . ( 2012; ). Role of early diagnosis and multimodal treatment in rhinocerebral mucormycosis: experience of 4 cases. . J Oral Maxillofac Surg 70:, 354–362.[CrossRef]
    [Google Scholar]
  35. Roden M.M. , Zaoutis T.E. , Buchanan W.L. , Knudsen T.A. , Sarkisova T.A. , Schaufele R.L. , Sein M. , Sein T. , Chiou C.C. . other authors ( 2005; ). Epidemiology and outcome of zygomycosis: a review of 929 reported cases. . Clin Infect Dis 41:, 634–653.[CrossRef]
    [Google Scholar]
  36. Samanta D.R. , Senapati S.N. , Sharma P.K. , Shruthi B.S. , Paty P.B. , Sarangi G. . ( 2009; ). Hard palate perforation in acute lymphoblastic leukemia due to mucormycosis – a case report. . Indian J Hematol Blood Transf 25:, 36–39.[CrossRef]
    [Google Scholar]
  37. Skiada A. , Lanternier F. , Groll A.H. , Pagano L. , Zimmerli S. , Herbrecht R. , Lortholary O. , Petrikkos G. . ( 2013; ). Diagnosis and treatment of mucormycosis in patients with hematological malignancies: guidelines from the 3rd European Conference on infections in leukemia. . Haematologica 98:, 492–504.[CrossRef]
    [Google Scholar]
  38. Skiada A. , Pagano L. , Groll A. , Zimmerli S. , Dupont B. , Lagrou K. , Lass‐Florl C. , Bouza E. , Klimko N. . other authors ( 2011; ). Zygomycosis in Europe: analysis of 230 cases accrued by the registry of the European Confederation of Medical Mycology (ECMM) Working Group on Zygomycosis between 2005 and 2007. . Clin Microbiol Infect 17:, 1859–1867.[CrossRef]
    [Google Scholar]
  39. Spellberg B. , Edwards J. Jr , Ibrahim A. . ( 2005; ). Novel perspectives on mucormycosis: pathophysiology, presentation, and management. . Clin Microbiol Rev 18:, 556–569.[CrossRef]
    [Google Scholar]
  40. Sun H.Y. , Singh N. . ( 2011; ). Mucormycosis: its contemporary face and management strategies. . Lancet Infect Dis 11:, 301–311.[CrossRef]
    [Google Scholar]
  41. Toma A. , Fenaux P. , Dreyfus F. , Cordonnier C. . ( 2012; ). Infections in myelodysplastic syndromes. . Haematologica 97:, 1459–1470.[CrossRef]
    [Google Scholar]
  42. Vardiman J.W. , Harris N.L. , Brunning R.D. . ( 2002; ). The World Health Organization (WHO) classification of the myeloid neoplasms. . Blood 100:, 2292–2302.[CrossRef]
    [Google Scholar]
  43. Vardiman J.W. , Thiele J. , Arber D.A. , Brunning R.D. , Borowitz M.J. , Porwit A. , Harris N.L. , Le Beau M.M. , Hellström‐Lindberg E. . other authors ( 2009; ). The 2008 revision of the World Health Organization (WHO) classification of myeloid neoplasms and acute leukemia: rationale and important changes. . Blood 114:, 937–951.[CrossRef]
    [Google Scholar]
  44. Verma A. , Singh V. , Jindal N. , Yadav S. . ( 2013; ). Necrosis of maxilla, nasal, and frontal bone secondary to extensive rhino‐cerebral mucormycosis. . Nat J Maxillofac Surg 4:, 249–251.[CrossRef]
    [Google Scholar]
  45. Viterbo S. , Fasolis M. , Garzino‐Demo P. , Griffa A. , Boffano P. , Iaquinta C. , Tanteri G. , Modica R. . ( 2011; ). Management and outcomes of three cases of rhinocerebral mucormycosis. . Oral Surg Oral Med Oral Pathol Oral Radiol Endod 112:, e69–e74.[CrossRef]
    [Google Scholar]
  46. Yohai R.A. , Bullock J.D. , Aziz A.A. , Markert R.J. . ( 1994; ). Survival factors in rhino‐orbital‐cerebral mucormycosis. . Surv Ophthalmol 39:, 3–22.[CrossRef]
    [Google Scholar]
http://instance.metastore.ingenta.com/content/journal/jmmcr/10.1099/jmmcr.0.000014
Loading
/content/journal/jmmcr/10.1099/jmmcr.0.000014
Loading

Data & Media loading...

Most Cited This Month

This is a required field
Please enter a valid email address
Approval was a Success
Invalid data
An Error Occurred
Approval was partially successful, following selected items could not be processed due to error