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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 315-318

Rhinomaxillary mucormycosis in an immunocompetent individual: Importance of early diagnosis


Department of Oral Medicine and Radiology, Saraswati Dental College and Hospital, Lucknow, Uttar Pradesh, India

Date of Submission29-Aug-2014
Date of Acceptance12-Nov-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Sunira Chandra
Reader, Department of Oral Medicine and Radiology, Saraswati Dental College and Hospital, Faizabad Road, 233 Tiwari Ganj, Lucknow - 227 105, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.145017

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   Abstract 

Rhinomaxillary mucormycosis presents with a characteristic destruction and necrosis most commonly affecting immunocompromised patients, especially diabetics. The present reported case was extremely unusual that the characteristic clinical feature of ulceration or necrosis was absent and the patient was apparently healthy (immunocompetent) with mere findings of generalized periodontal pockets, mobility of maxillary teeth, and a diffuse mild swelling in the anterior region of the hard palate on initial (clinical and radiographic) examination. This emphasized the need to include this fatal opportunistic infection in the differential diagnosis of maxillary swellings in otherwise healthy patients showing minimal clinical signs which could camouflage its presence.

Keywords: Immunocompetent, maxilla, mucormycosis


How to cite this article:
Chandra S, Reddy Enja SP, Nigam N, Nethan S. Rhinomaxillary mucormycosis in an immunocompetent individual: Importance of early diagnosis . J Indian Acad Oral Med Radiol 2014;26:315-8

How to cite this URL:
Chandra S, Reddy Enja SP, Nigam N, Nethan S. Rhinomaxillary mucormycosis in an immunocompetent individual: Importance of early diagnosis . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:315-8. Available from: https://www.jiaomr.in/text.asp?2014/26/3/315/145017


   Introduction Top


Mucormycosis is a fatal opportunistic infection caused by a saprophytic fungus belonging to the Phycomycete group. Six clinical variants are known, of which the rhinomaxillary form is the most common. [1] This article reports a case of rhinomaxillary mucormycosis with an atypical clinical presentation. Initial examination (clinical and radiographic) demonstrated mere findings of generalized periodontal pockets, mobility of maxillary teeth, and a diffuse mild swelling in the anterior region of the hard palate. No clinical signs of ulceration or necrosis were appreciated. It was clinically diagnosed as aggressive periodontitis and was histopathologically confirmed as rhinomaxillary mucormycosis.


   Case Report Top


A 28-year-old male Indian farmer reported with the chief complaint of generalized mobility of the maxillary posterior teeth since 1 month. He was apparently healthy, and his medical and dental histories were non-contributory. There was no associated history of any habit and he was not under any medication. No extraoral abnormalities and no lymphadenopathy were appreciated. The intraoral examination demonstrated generalized periodontal pockets with generalized grade I mobility and grade II mobility of 25 and 26. Severe gingival recession was also observed in relation to 26, with bleeding on probing [Figure 1]. Diffuse mild swelling in the anterior hard palatal region was observed [Figure 2]. On palpation, the swelling was hard in consistency and non-tender. The swelling appeared to be a normal anatomic variation. His oral hygiene was fair with generalized mild stains and calculus.
Figure 1: Intraoral photograph

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Figure 2: Mild diffuse swelling in the anterior region of the hard palate

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An orthopantomograph (OPG) revealed generalized bone loss (horizontal and vertical) which was more in the left maxillary anterior teeth region to 26 [Figure 3]. A provisional clinical diagnosis of aggressive periodontitis was made. Overall appraisal of the patient was done. Oral prophylaxis was advised along with systemic antibiotics (amoxicillin with tinidazole, 500 mg, tid, for 5 days) and nutritional therapy (multi-vitamins). Second phase of therapy was planned and the patient was recalled after a week. Patient did not respond to the first phase of treatment and his symptoms were not relieved. He also complained of pain with increased mobility.
Figure 3: OPG revealing generalized bone loss (horizontal and vertical) which was more in the left maxillary anterior teeth region to 26

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To rule out any nasomaxillary pathology and to investigate the mild swelling in the anterior hard palate, a computed tomography (CT) scan was advised, which revealed diffuse areas of destruction of hard palate, bilaterally with complete obstruction of the left maxillary sinus and partial obstruction of the right maxillary sinus. Obliteration of the right nasal cavity with deviation of the nasal septum toward the left side was appreciated [Figure 4]. Based on the CT findings, a biopsy was performed. The histopathological examination revealed necrosed bony trabeculae devoid of osteoblasts and osteocytes, surrounded by granulomatous inflammation containing numerous irregular, non-septate, broad fungal hyphae within the central fibrinous debris suggestive of rhinomaxillary mucormycosis [Figure 5]. The fungus was further demonstrated on the Gomori's silver methenamine stain confirming the presence of mucorales [Figure 6].

The patient underwent a wide surgical debridement of the involved dead and infected tissue. Intravenous treatment was administered under the supervision of an ENT specialist with metronidazole (500 mg, 4 times daily), liposomal amphotericin-B (1 mg/kg daily), and ceftriaxone (1 g, 2 times daily) for 2 months. The patient is under follow-up since 6 months with no signs of recurrence.
Figure 4: CT scans showing bilateral partial areas of destruction (hard palate) with complete obstruction of left maxillary sinus and partial obstruction of right maxillary sinus, and obliteration of the right nasal cavity with deviation of the nasal septum toward the left side

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Figure 5: Photomicrograph showing necrosed bony trabeculae surrounded by granulomatous inflammation containing numerous irregular, non-septate, broad fungal hyphae (H and E stain, 20×)

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Figure 6: Photomicrograph showing mucorales (Gomori's silver methenamine stain, 100×)

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   Discussion Top


Mucormycosis is an uncommon but fatal opportunistic infection caused by a saprophytic fungus belonging to the Phycomycete group that occurs in soil or as a mold on decaying food. Some synonyms used for this condition are zygomycosis and phycomycosis. [2] Paltauf (1885) described the first case of mucormycosis in human beings. He coined the term mycosis mucorina, which subsequently was renamed as mucormycosis. [1] It is the third most common invasive fungal infection, following aspergillosis and candidiasis. It accounts for 8.3-13.0% of all fungal infections and can be commonly cultured from the nose, throat, mouth, and feces. [3] Mucormycosis is found worldwide. It is virtually unheard of in otherwise healthy individuals. [2] No specific racial or geographic distribution was seen; neither any specific gender or age predilection was noticed. [4]

Five major clinical forms are described in literature depending on the portal of entry and the predisposing risk factors of the patient. These are: Rhinocerebral/rhinomaxillary and pulmonary, being the most common forms, followed by the gastrointestinal, cutaneous, and disseminated types. [5] Rhinocerebral zygomycosis is usually caused by Rhizopus oryzae or Rhizopus arrhizus, and typically occurs in the nasal cavity or paranasal sinuses. Nose is the most common portal of entry. These fungi erode and invade small blood vessels, leading to thrombosis, ischemia, and tissue necrosis. Involvement of sinuses or spread into the oral cavity (hard palate) is common. Pain, nasal discharge, and foul smell with black necrotic oral ulceration are the common findings. [6],[7],[8] From the palate and nasal mucosa, the infection may spread via the angular, lacrimal, and ethmoidal vessels, even extending into the retro-orbital region. It can further cause cranial nerve (II, III, IV, and VI) functional impairment. Hematogenous spread to the cavernous sinus and cavernous sinus thrombosis also have been reported. [9],[10]

It is most commonly seen in immunocompromised individuals, especially diabetics. However, this disease has also gained incidence in immunocompetent individuals. Literature analysis (1978-2009) carried out by Mignogna et al. [7] revealed that mucormycosis infection can even affect immunocompetent or otherwise healthy individuals. In the present case, mucormycosis affected an immunocompetent host, which was an unusual finding. This finding is also consistent with the other reported cases by Zapico et al. [11] and Sridhara et al. [12]

Intraorally, it presents as a palatal ulcer with raised erythematous borders. Surface of the ulcer appears black and necrotic with areas of denudation. [4],[8] It can also present with a palatal swelling, rather than the ulcer, and is associated with adjacent bony/alveolar denudation. [6] In the present case, merely a mild diffuse swelling in the anterior hard palate region was observed without any ulceration, with generalized periodontal pockets and mobility, which is another unusual clinical presentation. Other associated signs and symptoms are nasal obstruction, bloody nasal discharge, facial pain and headache, visual disturbances, and facial paralysis. In case of cranial vault involvement, blindness, lethargy, seizures, or even death may occur. [1]

Plain radiograph of sinuses and orbits may demonstrate sinus mucosal thickening, sinus opacification without fluid levels, and spotty destruction of the bony walls of the paranasal sinuses. CT scan or Magnetic Resonance Imaging (MRI) is helpful to delineate the extent of the disease. [3] In the present case, the OPG revealed only generalized bone loss (horizontal and vertical) because of which an initial diagnosis of aggressive periodontitis was made. It was the CT scan which revealed the extent of the underlying destruction.

Histopathologically, mucormycosis is characterized by numerous large non-septate hyphae (budding, non-dichotomous branching) approximately at 90° angle with extensive tissue necrosis. Angioinvasion by the fungi and neutrophilic infiltrate in the necrotic tissue can also be appreciated. [4],[6],[8],[9] Definitive diagnosis of mucormycosis is only made histopathologically. The fungus can further be demonstrated by use of a special stain, such as Grocott-Gomori methenamine silver stain, periodic acid Schiff, or calcofluor stain. [2] The Gomori's silver methenamine stain was used to demonstrate the fungal hyphae in the present case.

Successful treatment of mucormycosis requires early diagnosis. There are two primary aspects of the management of the rhinomaxillary mucormycosis for an immunocompetent host: Systemic antifungal therapy in the form of amphotericin-B or the more effective liposomal amphotericin-B and the surgical debridement of the dead, infracted, or infected tissue. The primary aspects of the management of the rhinomaxillary mucormycosis are reversal of the underlying risk factors (immunosuppression), prompt administration of antifungal therapy, and surgical debridement of the dead, infracted, or infected tissue. Primary antifungal therapy can be administered in the form of conventional amphotericin-B or lipid formulations of amphotericin (1 mg/kg/day) or in combination with echinocandin, posaconazole, or deferasirox (salvage therapy). [2],[6],[10]


   Conclusion Top


  • The incidence of development of mucormycosis, even in immunocompetent individuals, is on the rise.
  • Characteristic clinical feature of ulceration or necrosis can sometimes be absent in the early stages of the infection.
  • Advanced radiological techniques such as CT are helpful in its early detection.
  • The infection can only be diagnosed histopathologically.
  • Management of the rhinomaxillary mucormycosis for an immunocompetent host includes systemic antifungal therapy in the form of amphotericin-B or the more effective liposomal amphotericin-B and the surgical debridement of dead, infracted, or infected tissue.
  • Early diagnosis is of paramount importance which can prevent fatal potential systemic complications.


 
   References Top

1.Garg R, Gupta VV, Ashok L. Rhinomaxillary mucormycosis: A palatal ulcer. Contemp Clin Dent 2011;2:119-23.  Back to cited text no. 1
[PUBMED]  Medknow Journal  
2.Scully C, de Almeida OP. Orofacial manifestations of the systemic mycoses. J Oral Pathol Med 1992;21:289-94.  Back to cited text no. 2
    
3.Prabhu RM, Patel R. Mucormycosis and entomophthoramycosis: A review of the clinical manifestations, diagnosis and treatment. Clin Microbiol Infect 2004;10(Suppl 1):31-47.  Back to cited text no. 3
    
4.Pandey A, George J, Rao NN. Mucormycosis causing extensive destruction of maxilla. Indian J Dent Res 2004;15:74-7.  Back to cited text no. 4
[PUBMED]    
5.Hauman CH, Raubenheimer EJ. Oro-facial mucormycosis. Oral Surg Oral Med Oral Pathol 1989;68:624-7.  Back to cited text no. 5
    
6.Goel S, Palaskar S, Shetty VP, Bhushan A. Rhinomaxillary mucormycosis with cerebral extension. J Oral Maxillofac Pathol 2009;13:14-7.  Back to cited text no. 6
[PUBMED]  Medknow Journal  
7.Mignogna MD, Fortuna G, Leuci S, Adamo D, Ruoppo E, Siano M, et al. Mucormycosis in immunocompetent patients: A case-series of patients with maxillary sinus involvement and a critical review of the literature. Int J Infect Dis 2011;15:e533-40.  Back to cited text no. 7
    
8.Jayachandran S, Krithika C. Mucormycosis presenting as palatal perforation. Indian J Dent Res 2006;17:139-42.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13:236-301.  Back to cited text no. 9
    
10.Sun HY, Singh N. Mucormycosis: Its contemporary face and management strategies. Lancet Infect Dis 2011;11:301-11.  Back to cited text no. 10
    
11.Del Valle Zapico A, Rubio Suárez A, Mellado Encinas P, Morales Angulo C, Cabrera Pozuelo E. Mucormycosis of the sphenoid sinus in an otherwise healthy patient. Case report and literature review. J Laryngol Otol 1996;110:471-3.  Back to cited text no. 11
    
12.Sridhara SR, Paragache G, Panda NK, Chakrabarti A. Mucormycosis in immunocompetent individuals: An increasing trend. J Otolaryngol 2005;34:402-6.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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