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CASE REPORT
Year : 2014  |  Volume : 42  |  Issue : 3  |  Page : 112-114

Mucormycosis of the hard palate: a rare case report


Department of Pathology, Dr. D. Y. Patil Medical College and Hospital, Pimpri, Pune, India

Date of Submission26-Mar-2014
Date of Acceptance06-Aug-2014
Date of Web Publication29-Oct-2014

Correspondence Address:
Atul Jain
MD, Department of Pathology, Dr. D. Y. Patil Medical College and Hospital, Pimpri, Pune
India
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DOI: 10.4103/1110-1415.143566

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  Abstract 

Mucormycosis is a rare but aggressive opportunistic fungal infection. Mucormycosis refers to several different diseases; the causative agents of mucormycosis are the filamentous fungi of the Mucoraceae family, of the order Mucorales. Here, we report a case of mucormycosis in the hard palate of a 46-year-old woman without any predisposing factors. For early diagnosis of mucormycosis, histopathological examination is essential. In the present case, the fungus was identified by hematoxylin and eosin staining and confirmed by PAS and Grocott's silver methenamine special staining. As mucormycosis occurs infrequently, it may pose a diagnostic and therapeutic dilemma to those who are not familiar with its clinical presentation. The patient was successfully treated with a combination of surgical debridement and amphotericin-B. Through this case report, we would like to emphasize that mucormycosis of the hard palate occurs even in immunocompetent patients.

Keywords: Grocott′s silver methenamine, hard palate, mucormycosis, PAS


How to cite this article:
Jain A, Jain R, Banyameen IM, Shetty T. Mucormycosis of the hard palate: a rare case report. Tanta Med J 2014;42:112-4

How to cite this URL:
Jain A, Jain R, Banyameen IM, Shetty T. Mucormycosis of the hard palate: a rare case report. Tanta Med J [serial online] 2014 [cited 2020 Nov 29];42:112-4. Available from: http://www.tdj.eg.net/text.asp?2014/42/3/112/143566


  Introduction Top


Mucormycosis is a rare but aggressive opportunistic fungal infection. The genera most commonly responsible are Mucor or Rhizopus spp. [1]. Mucormycosis is an invasive fungal infection that was first described by Paulltauf A. in 1885. On the basis of the clinical presentation and particular site of involvement, six manifestations of the disease can be described: rhinocerebral, pulmonary, cutaneous, gastrointestinal, disseminated, and localized infections not otherwise belonging to the previous categories [2]. The causative agents of mucormycosis are the filamentous fungi of the Mucoraceae family, of the order Mucorales, subphylum Mucormycotina [3]. Infection usually results from inhalation of spores through the nose or mouth [4]. The fungus invades the blood vessels and subsequently spreads through them. Once fungal hyphae enter the blood stream, they can disseminate to other organs such as the cerebrum or lungs, which can prove fatal [5].

Rhinocerebral mucormycosis is most common type and its extension to the orbit or brain is quite usual; mucormycosis of the palate is a rare and late occurrence [6]. Here, we present a case of hard palate mucomycosis in a 46-year-old woman.


  Case report Top


A 46-year-old woman presented at the Department of Oral Medicine with a chief complain of swelling in the right side of her face for 10 days, associated with pain in the right side of her cheek and a blackish discharge from her nose and mouth. She had a history of fever for 10 days. No history of loss of vision or diabetes mellitus was recorded. On physical examination, the patient was found to be moderately built, well nourished, and febrile, with swelling over the right side of her cheek. Extraoral findings revealed bilateral submandibular lymphadenopathy and a blackish discharge from the nose and mouth of the patient. Anterior rhinoscopy showed erosion of a part of the septum, a blackish discharge from the right-side middle meatus, and blackish discoloration over the hard palate (around 2 × 1 cm). The ear was normal on examination. On the basis of history and clinical examination, a provisional diagnosis of a tuberculous ulcer or invasive fungal sinusitis was made. Endoscopic surgical debridement under general anesthesia was performed, and the sample was sent for histopathological examination. Thereafter, the patient was administered amphotericin-B and showed a better outcome. We excised a single grayish-black soft-tissue sample measuring 2 × 1 cm.

Histopathological examination by hematoxylin and eosin staining showed infected necrotic tissue with neutrophilic infiltration, interspersed with multiple colonies of thick nonseptate fungal hyphae of mucormycosis. PAS and Grocott's silver methenamine staining showed broad, thick-walled, infrequently nonseptate hyphae, with nonparallel sides and randomly spaced branches, indicative of mucormycosis [Figure 1],[Figure 2],[Figure 3],[Figure 4] and [Figure 5].
Figure 1:

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Figure 2:

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Figure 3:

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Figure 4:

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Fgiure 5:

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  Discussion Top


Mucormycosis is an infection that is caused by several species belonging to the orders Mucorales and Entomopthorales. The infectious agents most commonly involved are Rhizopus, Mucor, and Absidia spp.; Rhizomucor, Cunnighamella, and Mortierell spp. are the less commonly involved species [7]. Infection arises through inhalation of spores and contamination of traumatized tissue, or through ingestion and direct inoculation [8]. Mucormycosis is an opportunistic, rapidly progressing, and lethal form of fungal infection in humans [9],[10]. The fungus is nonpathogenic in healthy individuals and is present in normal cultures of nasal, throat, and oral cavity samples, representing opportunistic pathogenicity rather than true pathogenicity [11]. Mucormycosis has no geographic distribution, or specific racial, sex, or age predilection [12]. Our patient is a 46-year-old woman. Mucormycosis has been reported to occur in otherwise healthy individuals with various predisposing medical conditions, such as uncontrolled diabetes, blood dyscrasias, and protein calorie malnutrition, as well as in those using corticosteroids, immunosuppressive agents, etc. [5],[9]. In our case, the patient presented with fever, pain over the right side of her cheek, and a blackish discharge from her nose and mouth. Our patient also had features of tissue necrosis. Mucormyocosis is characterized by angioinvasion, thrombosis, infarction, and necrosis of the involved tissue [9]. The fungus invades arteries and causes damage secondary to thrombosis and ischemia [11].

Extension from the sinuses to the mouth caused the painful, black necrotic ulceration of the hard palate [9]. Our patient presented with erosion of a part of the septum, blackish discharge from the right-side middle meatus, and blackish discoloration of the hard palate. Mucormycosis is easily diagnosed using tissue sections. Involved tissues show focal areas of infection, necrosis, hemorrhage, and the presence of numerous large fungal hyphae, which are nonseptate, with a ribbon-like appearance and angle branching ranging from 450 to 900 [9],[12]. Our patient presented with similar findings. Successful treatment of mucormycosis involves aggressive surgical debridement of necrotic tissue, systemic antifungal therapy, and control of any underlying disease process [11]. Recently, intravenous liposomal amphotericin, intravenous lipid complex, and hyperbaric oxygen therapy have also been used [9]. There is a close histopathological resemblance between mucormycosis and aspergillosis; however, microscopically, aspergillosis has septate, branching hyphae, which can be distinguished from mucormycotic hyphae through its smaller width and the prominent acute angulations of the branching hyphae [14].


  Conclusion Top


Mucormycosis is a rapidly progressing fungal infection among humans, which can prove fatal; it can lead to the formation of palatal fistula in immunoincompetent patients [15]. Control and prevention of opportunistic fungal infections in patients suffering from debilitating diseases is very important. The major advantage in this case was early diagnosis and early management, as the disease is rapidly progressive in nature.


  Acknowledgements Top


Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Khan AR, Khan MU, Ullah S, et al. Rhinocerebral mucormycosis. J Coll Physicians Surg Pak 2002; 12:639-641.  Back to cited text no. 1
    
2.
Spellberg B, Edwards JJr, Ibrahim A. Novel perspectives on mucormycosis: pathophysiology, presentation, and management. Clin Microbiol Rev 2005; 18:556-569.  Back to cited text no. 2
    
3.
Hibbett Ds, Binder M, Bischoff Jf, Blackwell M, Cannon Pf, Eriksson Oe, et al. A higher-level phylogenetic classification of the fungi. Mycol Res 2007; 111:509-547.  Back to cited text no. 3
    
4.
Tugsel Z, Sezer B, Akalin T, et al. Facial swelling and palatal ulceration in a diabetic patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004; 98:630-636.  Back to cited text no. 4
    
5.
Auluck A. Maxillary necrosis by mucormycosis. a case report and literature review. Med Oral Patol Oral Cir Bucal 2007; 12:E360-E364.  Back to cited text no. 5
[PUBMED]    
6.
Kyrmizakin DF, Doxas PG, Hajiioannou SK, et al. Palatal ulcer due to mucormycosis. J Laryngol Otol 2002; 116:146-147.  Back to cited text no. 6
    
7.
Smith AG,Busstamante CI Gilmor GD. Zygomycosis (absidiomycosis) in an AIDS patient. Mycopathologia 1989; 105:7-10.  Back to cited text no. 7
    
8.
Sugar AM. Mucormycosis. Clin Infect Dis 1992; 1:s126-s129.  Back to cited text no. 8
    
9.
Jayachandran S, Kritika C. Mucormycosis presenting as palatal perforation. Indian J Dent Res 2006; 17:139-142.  Back to cited text no. 9
[PUBMED]  Medknow Journal  
10.
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.  Back to cited text no. 10
    
11.
Greenberg MS. Buket′s oral medicine diagnosis and treatment. 10th ed. Canada: B.C. Decker; 2003. 78-79.  Back to cited text no. 11
    
12.
Pandey A, George J, Rao NN. Mucormycosis causing extensive destruction of mailla. Ind J Dent Res 2006; 15:74-77.  Back to cited text no. 12
    
13.
Jones AC, Bentsen TY, Freedman PD, et al. Mucormycosis of the oral cavity. Oral Surg Oral Med Oral Pathol Oral Radiol 1993; 75:455-460.  Back to cited text no. 13
    
14.
Manjunatha BS, Das N, Rakesh V, Sutariya RV, Ahmed T. Mucormycosis of the hard palate masquerading as carcinoma. Clin Pract 2012; 2:28.  Back to cited text no. 14
    
15.
Batool S-N, Nickakhlagh S. Palate fistula due to sinonasal mucormycosis, a case report in diabetic patient. Jundishapur J Microbiol 2013; 6: p. 79. Special edition.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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