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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 4  |  Issue : 3  |  Page : 160-164

Rhino-maxillary osteomyelitis due to mucormycosis in an immunocompromised geriatric patient: A case report with review of treatment options


1 Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, India
2 Department of Prosthodontics and Crown and Bridge, Purvanchal Institute of Dental Sciences, Gorakhpur, Uttar Pradesh, India
3 Department of Orthodontics and Dentofacial Orthopaedics, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, India

Date of Web Publication16-Jul-2015

Correspondence Address:
Akhilesh Kumar Singh
Department of Oral and Maxillofacial Surgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-344X.160890

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  Abstract 

Mucormycosis is a type of opportunistic fungal infection which most commonly involves structures in the maxillofacial region such as paranasal air sinuses, maxilla, orbit and in advanced stages it may affect the brain. The most common route of its entry is through inhalation. Common predisposing condition is diabetes mellitus leading to immunocompromised state. We present a case of 63-year-old female patient with widespread maxillary osteomyelitis following teeth extraction later diagnosed as mucormycosis.

Keywords: Maxillary infection, mucormycosis, osteomyelitis, paranasal sinuses


How to cite this article:
Kumar N, Singh AK, Pandey S, Singh S. Rhino-maxillary osteomyelitis due to mucormycosis in an immunocompromised geriatric patient: A case report with review of treatment options. Int J Health Allied Sci 2015;4:160-4

How to cite this URL:
Kumar N, Singh AK, Pandey S, Singh S. Rhino-maxillary osteomyelitis due to mucormycosis in an immunocompromised geriatric patient: A case report with review of treatment options. Int J Health Allied Sci [serial online] 2015 [cited 2019 Sep 21];4:160-4. Available from: http://www.ijhas.in/text.asp?2015/4/3/160/160890


  Introduction Top


Mucormycosis is a rare condition of maxillofacial region caused by fungi belonging to the Mucorales order and the Mucoraceae family. [1] This fungus invades the arteries, forms thrombi within the blood vessels that reduce blood supply and cause necrosis of hard and soft tissues. It is a rapidly progressive, fatal infection mainly occurs in immunocompromised conditions such as diabetes mellitus, long-term steroid therapy, neutropenia, hematological malignancies (lymphoma and leukemia), organ transplantation, malnutrition, burns and immunosuppressive therapy. [2] Rhino-orbital-cerebral disease may present as rhinosinusitis, sinusitis, rhino-orbital, or rhinocerebral disease. Rhinocerebral mucormycosis is the most common type affecting paranasal air sinuses, maxilla, orbit and may extend to the brain. Clinical features include orbito-maxillary cellulitis, necrosis or eschar in the nasal cavity or on the palate with off and on pus discharge, nasal congestion, halitosis, usually asymptomatic but few cases reported with dull pain. Opthalmoplegia and loss of vision signifies disease progression. Intracranial complications include epidural and subdural abscesses and cavernous or sagittal sinus thrombosis. It is associated with very high mortality rate. [3]

Most often the clinical presentation is found to be a delay in healing of the socket following tooth extraction. Although the mortality rate is very high but an early clinical diagnosis followed by a prompt treatment may surely increase the chances of survival.

We present a case of 63-year-old diabetic female with a widespread rhino-maxillary osteomyelitis following dental extraction, which was managed by an aggressive surgical debridement and supportive medical therapy.


  Case report Top


A 63-year-old female patient reported to the Department of Oral and Maxillofacial Surgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi with a chief complaint of pain in upper jaw for the last 3 months. The patient also had a complaint of nasal congestion with mild headache. There was no history of fever, purulent discharge and paresthesia. The patient gave a history of extraction of maxillary anterior teeth 6 months back. Following dental extraction, a delay in healing of the socket was reported by the patient and had a persistent pain and discomfort which was affecting her physical and psychological status as well. There was no history of any systemic illness. On intraoral examination, we found a completely edentulous maxillary arch with necrotic bone of about 5-6 cm size in anterior region [Figure 1]. Computed tomographic scan showed bone destruction in anterior maxillary wall with obliteration of left maxillary sinus [Figure 2].
Figure 1: Preoperative photograph showing necrotic bone

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Figure 2: Computed tomographic scan (coronal view) showing bone destruction of maxillary anterior alveolar region with obliteration of left maxillary antrum

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All the biochemical investigations were within normal limit except random blood sugar (346 mg/dl). She was referred to Endocrinology Department for further investigations. There we found fasting blood sugar - 136 mg/dl and postprandial blood sugar - 226 mg/dl. Glycosylated hemoglobin value was 8.3. Diagnosis of type-II diabetes mellitus was made. She was advised insulin therapy for control of blood sugar. A biopsy was subsequently advised. Under local anesthesia, necrotic bone along with the adjacent soft tissue was excised and sent for a histopathological examination. Hematoxylin and eosin stained specimen showed multiple nonseptate mucomycotic hyphae thereby confirming its diagnosis as mucormycosis [Figure 3].
Figure 4: Removal of necrosed palatal bone

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The patient was planned for a surgical debridement under general anesthesia. On surgical exposure the whole of the necrotic bone and lining from left maxillary sinus was removed [Figure 4] and [Figure 5]. Primary closure was subsequently done [Figure 6]. To prevent the chance of hematoma, an arch bar was attached to a palatal splint, which was further fixed with circum-zygomatic suspension [Figure 7] and [Figure 8]. To our delight postoperative recovery was found to be absolutely uneventful. The patient was administered amphotericin-B 1 mg/kg/day in 5% Dextrose solution slowly infused for 2 weeks under close monitoring of renal and liver functions. After 2 weeks a satisfactory wound healing was observed. No recurrence was found [Figure 9].
Figure 5: Primary closure of the defect

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Figure 6: Modified palatal splint made up of self-cure acrylic with attached arch bar

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Figure 7: Stabilization of palatal splint with circum-zygomatic suspension

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Figure 8: Photomicrograph with H and E stained specimen revealed multiple nonseptate mucomycotic hyphae (×10)

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Figure 9: After 6 months, photograph showing satisfactory healing

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Figure 3: Exposure of the involved site via reflection of buccal and palatal mucoperiosteum

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  Discussion Top


Historically, Paltauf was credited with the first histologic description of generalized mucormycosis in a 52-year-old patient in 1885. Mucormycosis (phycomycosis, zygomycosis) is an opportunistic fulminant fungal infection, which mainly affects immunocompromised patients. It is caused by usually nonseptate fungi belonging to the class Zygomycota (now replaced by Glomeromycota). [4] The infection is acquired through the inhalation of spores. The three genera responsible for most cases include Rhizopus, Absidia, and Rhizomucor. Rhizopus is the predominant pathogen accounting for 90% cases of rhinocerebral mucormycosis. [5]

On the basis of clinical presentation and affected structure, it can be divided into six categories: Rhinocerebral, pulmonary, gastrointestinal, cutaneous, disseminated and miscellaneous. Out of all rhinocerebral type (44-49%) was the most common, followed by cutaneous (10-15.5%), pulmonary (10-11%), disseminated (6-11%) and gastrointestinal (2-11%). [6] The rhino-maxillary form of the disease, a subdivision of the rhinocerebral form, begins with the inhalation of the fungus by a susceptible individual. The infection begins in the nose and paranasal sinuses then spread to orbital and intracranial structures either by direct invasion or through the blood vessels. The fungus invades the arteries leading to thrombosis that subsequently causes necrosis of hard and soft tissues. [7]

The phagocytes of the healthy host are able to kill the spores of Mucorales by generating oxidative metabolites and defensins (cationic peptides). However, in case of uncontrolled diabetes ketoacidotic cases, the iron binding capacity of transferring is reduced leading to increased levels of iron in blood, which in turn is utilized by mucor for proliferation. [8] Such patients have decreased granulocyte phagocytic ability with altered polymorphonuclear leukocyte response. [9]

Mortality rate is higher than 50% with an incidence ranging from 62.5% in rhinocerebral form to 100% in disseminated form. [10],[11] Its high morbidity and mortality rates are related to its capacity for rapidly vascular invasions, with subsequent tissue necrosis and infarction. [12] The overall survival rate patients with mucormycosis ranges approximately from 50% to 85% with a higher survival rate of rhinocerebral mucormycosis than pulmonary or disseminated form. If the disease has not extended beyond the maxillary sinus, the prognosis is much better. [10] Surgical debridement reduces mortality rates from 55% to 27%, whereas medical treatment alone has a mortality rate varying from 39% to 70%. [13]

Aggressive medical treatment with conventional antifungals and nonconventional therapeutics are corner stone for successful treatment. [14] Polyenes like amphotericin-deoxycholates and lipid complex are primary therapeutic agents for mucormycosis. The dosage varies from 0.5 to 1.5 mg/kg body weight once daily for not <4 weeks. There should be close monitoring of serum electrolytes, as polyenes are known to cause potassium imbalance. [8],[15] Data from comparison of lipid complex amphotericin-B and deoxycholate amphotericin-B showed improved survival rates less side effects with lipid complex amphotericin-B. [14] Angioinvasion, thrombosis and tissue necrosis of Mucor leads to poor penetration of antifungal agents at the site of infection and compromises the efficacy of the same. [16]

Nonconventional therapeutic agents like anti-hypoglycemics, iron chelating agents, statins, granulocyte transfusions, cytokines, and hyperbaric oxygen have increased the survival rates to 94%. [14] Deferasirox (15-20 mg/kg daily), an approved oral iron chelator, has fungicidal effects and interacts synergistically with liposomal amphotericin-B against Glomeromycota. [17] In addition to lipid-lowering attributes, statins (lovastatin) have fungicidal activity against Glomeromycota and act synergistically with other antifungal agents, such as voriconazole. [18] Granulocyte transfusions specially in patients with neutropenia improves the treatment outcomes against mucormycosis. Nevertheless, in combination with granulocyte-macrophage colony-stimulating factor (GM-CSF) and interferon γ, the antifungal activities of neutrophils are notably increased. [19] Cytokines play a critical part in activation and recruitment of granulocyte and mononuclear phagocytes. Moreover, combined GM-CSF and liposomal amphotericin-B have notably prolonged survival and reduced fungal burden of the brain tissue in a murine model of systemic mucormycosis compared with liposomal amphotericin-B alone. [20] Hyperbaric oxygen has a direct antifungal effect in vitro through increased production of oxygen-based free radicals. This approach also yields several indirect antimicrobial effects and contributes to tissue healing. [21]

Prevention always remains a gold standard. Maintenance of immune balance especially in immunocompromised patients plays an important role in preventing this fulminant infection. Dental extraction in diabetic patients should be done cautiously under close observation and regular follow-up. Cornely et al. recommended prophylactic antifungal posoconazole in immunocompromised patients such as neutropenia. [22] Although the study done by Trifilio et al. found increase in number of mucormycosis in patients with regular antifungal prophylaxis which could be due to the fact that most of the prophylactic antifungals do not cover mucormycosis due to development of resistance. [23] Hence antifungal prophylaxis before surgical management remains controversial.


  Conclusion Top


Nonhealing tooth socket or bare bone of maxilla in a diabetic patient should be considered highly suspicious of mucormycosis. With similarities in clinical presentation of mucormycosis and osteomyelitis, the early diagnosis of former becomes difficult. Moreover the fungi are sensitive to culture growth. Hence, mucormycosis being a life threatening condition not only demands an early clinical diagnosis, but also an efficient and aggressive surgical management for its control. Amphotericin-B forms a failsafe medical therapy which is beneficial for the attainment of better results and prevents the disease progression as well.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Viterbo S, Fasolis M, Garzino-Demo P, Griffa A, Boffano P, Iaquinta C, et al. Management and outcomes of three cases of rhinocerebral mucormycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e69-74.  Back to cited text no. 1
    
2.
Jagdish C, Reetika S, Pallavi S, Jayanthi S, Divya P, Balasubramanya A. Mucormycosis of the paranasal sinus with gas forming maxillary osteomyelitis - A case report. Internet J Microbiol 2012. p. 10.  Back to cited text no. 2
    
3.
Kontoyiannis DP, Lewis RE. Agents of mucormycosis and entomophthoramycosis. In: Mandell GL, Bennett JE, Dolin R, editors. Principles and Practice of Infectious Diseases. 7 th ed. Philadelphia, PA: Churchill Livingstone/Elsevier; 2009.  Back to cited text no. 3
    
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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;111:509-47.  Back to cited text no. 4
    
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Auluck A. Maxillary necrosis by mucormycosis. a case report and literature review. Med Oral Patol Oral Cir Bucal 2007;12:E360-4.  Back to cited text no. 5
    
6.
Chakrabarti A, Das A, Sharma A, Panda N, Das S, Gupta KL, et al. Ten years′ experience in zygomycosis at a tertiary care centre in India. J Infect 2001;42:261-6.  Back to cited text no. 6
    
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Greenberg MS. Ulcerative vesicular and bullous lesions. In: Greenberg MS, Glick M, editors. Burket′s Oral Medicine Diagnosis and Treatment. Elsevier; 2003. p. 79.  Back to cited text no. 7
    
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Aggarwal P, Saxena S, Bansal V. Mucormycosis of maxillary sinus. J Oral Maxillofac Pathol 2012;11:66-9.  Back to cited text no. 8
    
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Brown OE, Finn R. Mucormycosis of the mandible. J Oral Maxillofac Surg 1986;44:132-6.  Back to cited text no. 9
    
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Spellberg B, Edwards J Jr, Ibrahim A. Novel perspectives on mucormycosis: Pathophysiology, presentation, and management. Clin Microbiol Rev 2005;18:556-69.  Back to cited text no. 10
    
11.
Jayalakshmi SS, Reddy RG, Borgohain R, Subramanyam C, Panigrahi M, Sundaram C, et al. Predictors of mortality in rhinocerebral mycosis. Neurol India 2007;55:292-7.  Back to cited text no. 11
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Kontoyiannis DP, Lewis RE. Invasive zygomycosis: Update on pathogenesis, clinical manifestations, and management. Infect Dis Clin North Am 2006;20:581-607, vi.  Back to cited text no. 12
    
13.
Lee FY, Mossad SB, Adal KA. Pulmonary mucormycosis: The last 30 years. Arch Intern Med 1999;159:1301-9.  Back to cited text no. 13
    
14.
Shetty SR, Punya VA. Palatal mucormycosis: A rare clinical dilemma. Oral Surg 2008;1:145-8.  Back to cited text no. 14
    
15.
Prasad K, Lalitha RM, Reddy EK, Ranganath K, Srinivas DR, Singh J. Role of early diagnosis and multimodal treatment in rhinocerebral mucormycosis: Experience of 4 cases. J Oral Maxillofac Surg 2012;70:354-62.  Back to cited text no. 15
    
16.
Chakrabarti A, Das A, Mandal J, Shivaprakash MR, George VK, Tarai B, et al. The rising trend of invasive zygomycosis in patients with uncontrolled diabetes mellitus. Med Mycol 2006;44:335-42.  Back to cited text no. 16
    
17.
Ibrahim AS, Gebermariam T, Fu Y, Lin L, Husseiny MI, French SW, et al. The iron chelator deferasirox protects mice from mucormycosis through iron starvation. J Clin Invest 2007;117:2649-57.  Back to cited text no. 17
    
18.
Chamilos G, Lewis RE, Kontoyiannis DP. Lovastatin has signifi cantactivity against zygomycetes and interacts synergistically with voriconazole. Antimicrob Agents Chemother 2006;50:96-103.  Back to cited text no. 18
    
19.
Gil-Lamaignere C, Simitsopoulou M, Roilides E, Maloukou A, Winn RM, Walsh TJ. Interferon- gamma and granulocyte-macrophage colony-stimulating factor augment the activity of polymorphonuclear leukocytes against medically important zygomycetes. J Infect Dis 2005;191:1180-7.  Back to cited text no. 19
    
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Safdar A. Antifungal immunity and adjuvant cytokine immune enhancement in cancer patients with invasive fungal infections. Clin Microbiol Infect 2007;13:1-4.  Back to cited text no. 20
    
21.
Tragiannidis A, Groll AH. Hyperbaric oxygen therapy and other adjunctive treatments for zygomycosis. Clin Microbiol Infect 2009;15 Suppl 5:82-6.  Back to cited text no. 21
    
22.
Cornely OA, Maertens J, Winston DJ, Perfect J, Ullmann AJ, Walsh TJ, et al. Posaconazole vs. fluconazole or itraconazole prophylaxis in patients with neutropenia. N Engl J Med 2007;356:348-59.  Back to cited text no. 22
    
23.
Trifilio S, Singhal S, Williams S, Frankfurt O, Gordon L, Evens A, et al. Breakthrough fungal infections after allogeneic hematopoietic stem cell transplantation in patients on prophylactic voriconazole. Bone Marrow Transplant 2007;40:451-6.  Back to cited text no. 23
    


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