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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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CASE REPORT
Year : 2011  |  Volume : 29  |  Issue : 6  |  Page : 87-91
 

Mucormycosis associated with juvenile diabetes


1 Department of Pedodontics and Preventive Dentistry, Narayana Dental College, Nellore, Andhra Pradesh, India
2 Department of Oral Pathology, Narayana Dental College, Nellore, Andhra Pradesh, India

Date of Web Publication12-Dec-2011

Correspondence Address:
SVSG Nirmala
Department of Pedodontics and Preventive Dentistry, Narayana Dental College, Nellore, Andhra Pradesh - 524 002
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.90752

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   Abstract 

Mucormycosis is one of the most rapidly progressing and lethal form of fungal infections in humans which usually begins in the nose and paranasal sinuses. The fungus assaults the arteries, leading to thrombosis that subsequently causes necrosis of hard and soft tissues. The purpose of this article is to describe a rare case of maxillary necrosis due to mucormycosis in a 12-year-old diabetic patient and emphasizes on early diagnosis and timely management of this potentially fatal fungal infection.


Keywords: Amphotericin B, juvenile diabetes, maxillary bone necrosis, mucormycosis


How to cite this article:
Nirmala S, Lalitha V, Sivakumar N, Kumar K K, Srikanth M. Mucormycosis associated with juvenile diabetes. J Indian Soc Pedod Prev Dent 2011;29, Suppl S1:87-91

How to cite this URL:
Nirmala S, Lalitha V, Sivakumar N, Kumar K K, Srikanth M. Mucormycosis associated with juvenile diabetes. J Indian Soc Pedod Prev Dent [serial online] 2011 [cited 2020 Jan 27];29, Suppl S1:87-91. Available from: http://www.jisppd.com/text.asp?2011/29/6/87/90752



   Introduction Top


Mucormycosis is an uncommon acute opportunistic infection caused by a saprophytic fungus which belongs to the order Mucorales, family Mucoraceae and class Zygomycetes. [1] It was first described in humans by Paultaufi in 1885, as cited by Marchevsky and colleagues. [2] The genera of Mucorales that are recognized as human pathogens are Rhizopus, Absidia, Rhizomucor and Mucor. However, the Mucorales are present everywhere in the environment and commonly found in bread molds and decaying vegetation. They grow rapidly and constantly discharge spores into the environment. [1],[2],[3] The maxilla rarely undergoes necrosis due to its rich vascularity. [4] Symptoms involving the oral, cranial, and facial tissues account for about 60% of all cases, and rhinocerebral, rhino-orbital forms are most often cited in the literature. [3],[4],[5],[6],[7],[8],[9] Intraorally, the hard palate is usually affected because of its proximity to the infection of the nasal fossa, and the paranasal sinuses, alveolar mucosa, tongue, the buccal mucosa of the lips and cheek may also be affected; isolated intraoral involvement is extremely rare. [10] The purpose of this article is to describe a rare case of maxillary necrosis by mucormycosis in a 12-year-old juvenile diabetic patient and emphasizes on early diagnosis and timely management of this potentially fatal fungal infection.


   Case Report Top


A 12-year-old South Indian boy presented with the complaint of pain and swelling in the left maxillary posterior region since 4 days to the Department of Pedodontics and Preventive Dentistry, Narayana Dental College and Hospital. Pain was reported since 4 months, and it was moderate in nature and aggravated on bending the head and chewing food. There was no history of fever, purulent discharge, paresthesia and foul odor. On general examination, vital signs were within normal limits. Extraoral examination revealed a diffuse swelling on the left side of the face, measuring 4 × 4 inches in size, extending superiorly from the supraorbital margin and inferiorly to the angle of the mouth [Figure 1]. Intraorally, a ragged ulcer was observed on the palate that demonstrated a raised erythematous border and a necrotic bone of about 1 cm in the left maxillary molar region [Figure 2] and presented with permanent dentition with retained maxillary left second primary molar. Radiographic examination using Water's view (paranasal view) showed haziness of left maxillary sinus with erosion of lateral sinus wall [Figure 3]. Biochemical investigations revealed elevated blood sugar levels with fasting blood sugar of 212 mg/dL (normal 90-180 mg/dL) and postprandial blood sugar level of 223 mg/dL (normal 100-180 mg/dL). Surrounding soft tissue along with the hard tissue was excised from the lesion under local anesthesia for histopathologic examination. Histopathology with hemotoxylin and eosin staining showed the presence of large, elongated, non-septate, non-branching fungal hyphae present in the marrow spaces. Grocott's modified silver methenamine special staining technique was used for further confirmation of these non-septate branching hyphae of mucormycosis [Figure 4]a and b. The patient was hospitalized and blood sugar levels were controlled with insulin. The necrotic lesion along with 1 cm of adjacent bone was excised under general anesthesia [Figure 5] and [Figure 6] to avoid further recurrence. The patient was administered amphotericin B 0.8 mg/kg/day intravenously for 2 weeks, which was slowly infused over 4-6 hours, and blood urea and creatinine levels were monitored as the drug can cause renal toxicity. After 2 weeks, the patient was recalled for follow-up and the healing was satisfactory [Figure 7].
Figure 1: Extraoral picture showing facial swelling of the patient on the left side

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Figure 2: Intraoral picture showing necrotic bone of about 1 cm diameter in the left maxillary molar region

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Figure 3: Water's view showing haziness of both maxillary sinuses with erosion in the lateral wall of left maxillary sinus

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Figure 4 a, b: Histopathologic picture showing non-septate fungal hyphae observed under Grocott's modified silver methenamine staining technique

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Figure 5: Intraoral picture showing exposure of raw bony surface and nerve bundle

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Figure 6: Intraoral picture showing mass of tissue with slough after removal

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Figure 7: Postoperative intraoral photograph showing healing

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   Discussion Top


Mucormycosis usually occurs in immunocompromised patients but can infect healthy individuals as well. [5],[6],[7] The predisposing factors are uncontrolled diabetes, particularly in patients having ketoacidosis, malignancies such as lymphomas and leukemias, renal failure, organ transplant, long-term corticosteroid and immunosuppressive therapy, cirrhosis, burns, protein energy malnutrition and AIDS. [4] Tugsel et al. also reported that Type I diabetes mellitus is most commonly associated with 40% of mucormycosis cases overall. [11] Diabetes mellitus is one of the most common endocrinal disorders which alter many physiological processes, presenting as diminished resistance to infections, vascular and micro-vascular changes, and altered tissue response. It has been suggested that the ability of serum of immunocompromised patients to inhibit Rhizopus in vitro is reduced, which makes them suitable hosts to opportunistic fungal infections. [12] This microbe may be cultured from the oral cavity, nasal passages, throat, and stool of healthy patients without clinical signs of infection. [11],[12],[13] This fungal infection usually originates from paranasal sinuses, invades the blood vessels and subsequently spreads through them. Once fungal hyphae enter into blood stream, they can disseminate to other organs such as cerebrum or lungs, which can be fatal to the patient. The mucor hyphae from these thrombi within the blood vessels reduce the vascularity to the tissues and cause necrosis. [5],[6],[7] The peripheral vascular disease of diabetic patients is thought to predispose them to local tissue ischemia and increased susceptibility to numerous infections. [11] Thrombosis of the descending palatine artery, a branch of the internal maxillary artery could be the cause for mucormycotic infection as well as chronic diabetes may result in necrosis of the maxilla. [7]

Usually mucormycosis occurs as pulmonary, gastrointestinal, disseminated or rhinocerebral infection. [11],[13],[14],[15] In the case presented by us, infection was only localized to the maxilla, causing necrosis without any other symptoms. This is one of the most aggressive effects and is potentially fatal in diabetic patients because of impaired host defense mechanism and increased availability of micronutrients such as iron. [11] The general health of this patient was good and he did not develop ketoacidosis that facilitates spread of infection to other organ systems.

In the early stages of the disease, the patients exhibit facial cellulitis, nasal discharge, headache, and lethargy. [15] However, in contrast to other reports, these symptoms were absent in the present case. Among the clinical differential diagnoses, we can consider malignant salivary gland tumor arising from the accessory glands of the palate. [16] Bone necrosis can also occur due to extension of infections such as acute necrotizing ulcerative gingivitis (ANUG) from gingiva to the bone. In the case reported here, the gingiva was normal. There is a close histopathologic resemblance between mucormycosis and aspergillosis. Microscopically, aspergillosis has septate branching hyphae, which can be distinguished from mucormycotic hyphae due to smaller width and prominent acute angulations of branching hyphae. [17]

The diagnosis is based on history, clinical examination, diagnostic radiography and biopsy. Debridement of nonviable necrotic tissue has been the mainstay of treatment over the years and remains so today. With the advent of potent antifungal medications, a combination of surgery and medication has provided better outcomes. In general, aggressive medical support and surgical intervention is required for these critically ill patients. Reversal of the underlying systemic disorder and use of amphotericin B are the most effective management methods. [8]

The principle steps taken for the management of the patient were; initially, diabetes was controlled by insulin therapy and dietary restrictions. Later, surgical removal of the necrotic bone in the maxilla was done, which was accountable as a nidus of infection and prevented the action of systemically administered antifungal drugs. In this patient, entire necrotic bone was removed along with the antral lining, followed by debridement with Povidone Iodine 7.5%. Finally, amphotericin B was administered parenterally as it is the drug of choice in the treatment of mucormycotic infection. [5],[6],[7],[17] Blood urea and creatinine levels were monitored. Postoperatively, the patient was advised an obturator to prevent oro-nasal regurgitation. Mucormycosis was long regarded as a fatal infection with poor prognosis. However, with early medical and surgical management, survival rates are now thought to exceed 80%. [12]

This case epitomizes the kind of treatment planning decisions frequently made by clinicians involved in the care of patients. Management of chronic dental infections in patients should be based on history that correlates examination and investigation findings with outcomes of treatment. However, no controlled studies are available to identify when conservative or more aggressive treatment approaches should be used. As a result, clinicians must have to depend on published case reports, personal experience, and clinical decision.


   Conclusion Top


Early diagnosis of mucormycosis is necessary to avoid the further spread of infection, which may lead to high morbidity and mortality. Hence, health practitioners should be familiar with the signs and symptoms of the disease. It is important to understand and implement the treatment options which can help to manage these patients. Knowledge of potentially devastating complications can help to prevent the unfortunate consequences. Furthermore, children with diabetes should maintain excellent oral hygiene procedures, which will require parental supervision.

 
   References Top

1.Linder N, Keller N, Huri C, Kuint J, Goldshmidt-Reuven A, Barzilai A. Primary cutaneous mucormycosis in a premature infant: Case report and review of the literature. Am J Perinatol 1998;15:35-8.  Back to cited text no. 1
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2.Marchevsky AM, Bottone EJ, Geller SA, Giger DK. The changing spectrum of disease, etiology, and diagnosis of mucormycosis. Hum Pathol 1980;11:457-64.  Back to cited text no. 2
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3.Ribeiro NF, Cousin GC, Wilson GE, Butterworth DM, Woodwards RT. Lethal invasive mucormycosis: Case report and recommendations for treatment. Int J Oral Maxillofac Surg 2001;30:156-9.  Back to cited text no. 3
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4.Pogrel MA, Miller CE. A case of maxillary necrosis. J Oral Maxillofac Surg 2003;61:489-93.  Back to cited text no. 4
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5.Leitner C, Hoffmann J, Zerfowski M, Reinert S. Mucormycosis necrotizing soft tissue lesion of the face. J Oral Maxillofac Surg 2003;61:1354-8.  Back to cited text no. 5
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6.Zapico AD, Suarez AR, Encinas PM, Angulo CM, Pozueglo EC. Mucormycosis of the sphenoidal sinus in an otherwise healthy patient. Case report and literature review. J Laryngol Otol 1996;110:471-3.  Back to cited text no. 6
    
7.Jones AC, Bentsen TY, Fredman PD. Mucormycosis of the oral cavity. Oral Surg Oral Med Oral Pathol 1993;75:455-60.  Back to cited text no. 7
    
8.Salisbury PL 3rd, Caloss R Jr, Cruz JM, Powell BL, Cole R, Kohut RI. Mucormycosis of the mandible after dental extractions in a patient with acute myelogenous leukaemia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:340-4.  Back to cited text no. 8
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9.Shand MJ, Albrecht MR, Burnett FH, Miyake A. Invasive fungal infection of the mid facial and orbital complex due to scedosporium apiospermum and mucormycosis. J Oral Maxillofac Surg 2004;62:231-4.   Back to cited text no. 9
    
10.Tryfan S, Stanopoulous I, Kakavelas E, Nikolaidou A, Kioumis I. Rhinocerebral mucormycosis in a patient with latent diabetic mellitus: A case report. J Oral Maxillofac Surg 2002;60:328-30.  Back to cited text no. 10
    
11.Tugsel Z, Sezer B, Akalin T. Facial swelling and palatal ulceration in a diabetic patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:630-6.  Back to cited text no. 11
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12.Roden MM, Zaoutis TE, Buchanan WL, Epidemiology and outcome of zygomycosis: A review of 929 reported cases. Clin Infect Dis 2005;41:634-53.   Back to cited text no. 12
    
13.Brown OE, Finn R. Mucormycosis of the mandible. J Oral Maxillofac Surg 1986;44:132-6.  Back to cited text no. 13
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14.Napoli JA, Donegan JO. Aspergillosis and necrosis of maxilla: A case report. J Oral Maxillofac Surg 1991;49:532-4.  Back to cited text no. 14
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15.Buhl MR, Joseph TP, Snelling BE, Buhl L. Temporofacial zygomycosis in a pregnant women. Infection 1992;20:230-2.  Back to cited text no. 15
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16.Auluck A. Maxillary necrosis by mucormycosis. A case report and literature review. Med Oral Patol Oral Cir Buccal 2007;12;E360-4.  Back to cited text no. 16
    
17.Van der Westhuijzen AJ, Grotepass FW, Wyma G, Padayachee A. A rapidly fatal palatal ulcer rhinocerebral mucormycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1989;68:32-6.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


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