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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 32  |  Issue : 1  |  Page : 60-64

Cone beam computed tomography: A diagnostic aid in rhinomaxillary mucormycosis following tooth extraction in patient with diabetes mellitus


Department of Oral Medicine and Radiology, Vydehi Institute of Dental Sciences, Bengaluru, Karnataka, India

Date of Submission21-Jan-2020
Date of Decision06-Mar-2020
Date of Acceptance15-Mar-2020
Date of Web Publication17-Apr-2020

Correspondence Address:
Dr. Shilpa Padar Shastry
Department of Oral Medicine and radiology, Vydehi Institute of Dental Sciences, NO. 82, EPIP Area, Whitefield, Bengaluru - 560 066, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_12_20

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   Abstract 


Mucormycosis is a fatal, opportunistic deep fungal infection caused by genera Rhizopus, and compromised systemic health of the patient predisposes to this condition. Rhino maxillary or rhino nasal form of mucormycosis begins after the inhalation of fungal spores and invasion into the nasal mucosa, and spreads to the paranasal sinuses. This infection can also extend into orbits and intracranium causing fatal rhino orbito-cerebral mucormycosis. Hence early intervention is very significant in terms of prognosis. The aim of current paper is to report a case of rhinomaxillary mucormycosis involving palate, nasal and maxillary sinus in an uncontrolled diabetic patient following tooth extraction highlighting the etiopathogenesis, spread of this infection and cone beam computed tomography (CBCT) features.

Keywords: Diabetes mellitus, mucormycosis, palate, solitary ulcer


How to cite this article:
Shastry SP, Murthy PS, Jyotsna T R, Kumar NN. Cone beam computed tomography: A diagnostic aid in rhinomaxillary mucormycosis following tooth extraction in patient with diabetes mellitus. J Indian Acad Oral Med Radiol 2020;32:60-4

How to cite this URL:
Shastry SP, Murthy PS, Jyotsna T R, Kumar NN. Cone beam computed tomography: A diagnostic aid in rhinomaxillary mucormycosis following tooth extraction in patient with diabetes mellitus. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2020 May 30];32:60-4. Available from: http://www.jiaomr.in/text.asp?2020/32/1/60/282602




   Introduction Top


Mucormycosis (Zycomycosis or Phycomycosis) is an acute fulminant, fatal, opportunistic deep fungal infection caused by genera Rhizopus, Rhizomucor, and Absidia. This condition was first described by Plataut AP in 1885.[1] It is known to occur in patients with compromised systemic health as in diabetic mellitus (60-80%), leukemia, prolonged corticosteroid therapy, chronic renal failure, antineoplastic therapy, immunosuppressive therapy, deferroxamine therapy, protein calorie malnutrition owing to the impaired immunity in these patients.[2],[3],[4],[5]

Mucormycosis is categorized into rhinocerebral (most common form), pulmonary, cutaneous, gastrointestinal or disseminated. Rhinocerebral form can be further subdivided depending on the tissues being affected as rhino nasal or rhino maxillary, rhino orbital and rhino orbito cerebral.[6] The rhino nasal mucormycosis begins as the fungal spores are inhaled and invades the nasal mucosa, and spreads to the paranasal sinuses. The infection also spreads from the sinuses into the mouth producing painful, necrotic ulceration of the hard palate.[7],[8] The infection may involve the orbit extending directly from paranasal sinuses or via the angular vein, lacrimal or ethmoidal vessel resulting in rhinoorbital mucormycosis. Rhinocerebral mucormycosis sets in when intracranial extension occurs from invasion by way of superior orbital fissure, ophthalmic vessels, cribriform palate and sometimes, through carotid artery.[9] Hence early intervention is very significant in terms of prognosis as 76-81% survival rate has been observed in patients who began treatment within 6 days, while a treatment delay of more than 12 days reduces this rate to 36-42%.[6] The aim of current paper is to report a case of rhinomaxillary mucormycosis involving palate, nose and maxillary sinus in an uncontrolled diabetic patient highlighting the etiopathogenesis, spread of this infection and CBCT features. To the best of our knowledge, CBCT characteristics of mucormycosis is rarely been reported. However, usefulness of CBCT in diagnosis of mucormycosis is may be limited, especially if there are intracranial extensions.


   Case Report Top


A 52-year-old male patient complained of an ulcer in the roof of the mouth since one and a half months. Pain was associated with ulcer from past one month, which was continuous and severe in intensity. Patient underwent extraction of maxillary right second premolar (tooth number 15), following which there was a non-healing socket since one and a half months. This extraction was done in a private dental clinic due to periodontal involvement and patient did not provide details of any investigations that were done before the extraction. After one week of extraction, there was formation of an ulcer in the hard palate which extended from the socket with foul smell from the ulceration. The ulcer was increasing in size and turned blackish in the last 15 days. Ulceration was followed by mobility of adjacent tooth (tooth number 13 and 14) and subsequent exfoliation of these teeth. There was also a history of bleeding from the nose in the past one week. There was no associated pus discharge, burning sensation or history of trauma. The patient was diagnosed as type II diabetic 1 year back and was currently under oral hypoglycemic medication. Patient was a chronic smoker for 15 years, smoked about 3-4 cigarettes per day.

On examination, clotted blood was seen in the right and left nostril region [Figure 1]. An irregularly shaped ulcer measuring about 5 × 4 centimeters in size was noted on hard palate extending from the incisive papilla to 1 cm anterior to junction of soft palate and hard palate; anteriorly extending from marginal palatal gingiva of 13 to marginal palatal gingiva of 22; posteriorly extending from 1 cm medial to alveolar ridge of 14, 15 region crossing midline to 1 cm medial marginal palatal gingiva of 24, 25 region [Figure 2] Another ulceration of 5 mm × 5 mm was noted 5 mm lateral to the mid palatine region on right alveolar ridge. Denuded bone was also seen in the ulcerative region. Floor of the ulcer was black with thin erythematous lining. There was also greyish white discoloration found around the ulcer. The ulcer was severely tender. Non-healing sockets seen in relation to 15, 14, and 13. Maxillary anterior teeth (Tooth number 11, 12, 21 and 22) were mobile. Lymph nodes of neck were nonpalpable.
Figure 1: Clotted blood noted in the right and left nostril region (Black arrow)

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Figure 2: Ulcer on hard palate with blackish floor (black arrow) and another ulceration of 5 mm lateral to the mid palatine region (white arrow)

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Based on the clinical findings of blackish ulcer on palate and medical history of diabetes mellitus, mucormycosis of hard palate was given as provisional diagnosis and patient was subjected to radiological and serological investigations. However other deep fungal infection like aspergillosis and bacterial infection like tuberculosis were considered in differential diagnosis.

Digital panoramic radiograph showed [Figure 3] haziness over right and left maxillary sinus, tooth sockets of right maxillary canine and first premolar.
Figure 3: Digital panoramic radiograph showing haziness over right and left maxillary sinus, extracted tooth sockets

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CBCT of the maxilla was performed [Figure 4], [Figure 5], [Figure 6].
Figure 4: (a and b) Complete opacification of left and right maxillary sinus; (c) air entrapment (dotted line); breach in the floor and posterior wall of right sinus (yellow and red arrow)

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Figure 5: Hypertrophied nasal conchae (Yellow arrow); Destruction of lateral wall of nose (red Arrow)

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Figure 6: (a) Hypertrophy of the left superior, middle and inferior concha on the left side; (b) Sphenoid sinus; (c and d) Irregular destruction of cortices in the alveolar region

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Maxillary sinus

Complete opacification of left and right maxillary sinus with air entrapment was evident [Figure 4]a and b]; medial wall of the left and right maxillary sinus were destroyed; floor of the maxillary sinus was breached in the right side [Figure 4]c, but intact on the left side; Similarly, right posterior wall of the maxillary sinus was breached [Figure 4] but, it was intact on left side; roof of the maxillary sinus was intact on both left and right side.

Nasal cavity

Hypertrophy of nasal conchae with disruption of lateral wall of the nose was evident in CBCT [Figure 5]. There was complete blockage of left sinus osteum; deviated nasal septum was present; hypertrophy of the left superior, middle and inferior concha on the left side; hypertrophy of the inferior concha on the right side [Figure 6]a.

Ethmoid sinus

Obliteration of left ethmoid sinus was seen, but right ethmoid sinus did not show any opacification.

Sphenoid sinus

Walls of sphenoid sinus were intact without any opacification [Figure 6]b.

Alveolar bone

Irregular outline of extraction sockets with disruption in the buccal and palatal cortical plates were apparent [Figure 6]c and d].

Patient's random blood sugar levels was very high (360 mg/dl), fasting sugar level was 148 mg/dl and Postprandial sugar level was 220 mg/dl. Treatment was initiated with systemic antifungal therapy (Amphotericin B, 1 mg/kg/day for 2 weeks). Patient was also monitored and referred to the general physician for glycaemic control. Surgical debridement was carried out under general anaesthesia, along with the extraction of maxillary central and lateral incisors (teeth were mobile and had poor prognosis). Histopathologically, broad aseptate fungal structures in stroma as well as infiltrating to the blood vessels were seen, suggestive of mucormycosis [Figure 7]. Postoperatively, slough around the surgical margins along with oronasal communication was evident [Figure 8]. Immediate postoperative paranasal sinus view was taken, showing haziness in the left and right maxillary sinus, endotracheal intubation tube and absence of lateral wall of the nose [Figure 9]. The patient was followed up for 3 months and there there was complete healing of the ulcer and formation of oronasal communication [Figure 10]. Patient was telephonically contacted after 2 years, who reported to us informing that he did not develop any ulceration or other symptoms, except for persisting oronasal communication [Figure 11].
Figure 7: Histopathology (7a - 10x magnification, 7b - 20x magnification)

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Figure 8: Postoperative extraoral and intraoral photographs

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Figure 9: Postoperative paranasal sinus view

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Figure 10: Intraoral picture after 3 months of Surgery with oronasal communication

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Figure 11: Photograph sent by patient 2 years after the surgery

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   Discussion Top


In normal hosts, Rhizopus is a saprophytic organism and these are eliminated by mononuclear and polymorphonuclear phagocytes by the generation of oxidative metabolites and cationic peptides.[10] This oxidative capacity is lost in patients with diabetic ketoacidosis. Moreover, reduced chemotaxis and phagocytic efficiency in these patients permit the growth of innocuous organisms. The hyperglycemic state also promotes fungal growth and are thus are highly susceptible to mucormycosis.[11] There is also evidence that increased availability of iron increases the risk of mucormycosis. Therefore, due to elevated available serum iron in patients with diabetic ketoacidosis may prone them to mucormycosis.[12]

Once the fungus begins to grow, it invades the human host through attaching to the endothelial cell lining of blood vessels leading to haematogenous spread, thrombosis, bleeding or infarction. This leads to formation of mucor thrombus through fibrin reaction, thus causing vascular occlusion, ischemia and infarction. This explains the formation of a black necrotic eschars that are formed on the nasal or palatine mucosa, characteristic feature of mucormycosis. There is also an evidence of area of ulceration or an extraction socket in the mouth in an immunocompromised patient can also be a port of entry for mucormycosis in maxillofacial region.[1],[9] In our case, the patient gave a history of extraction followed by development of ulceration, mobility and exfoliation of teeth.

Diagnosis of mucormycosis requires thorough clinical history and evaluation of underlying medical illness. Moreover, presence nasal or palatal necrosis raises strong suspicion of this condition. Radiographic evaluation benefits to reveal bony erosions, extent of sinus involvement, as well as presence of orbital infiltrations and intracranial involvement.

CBCT shows bony erosion, involvement of sinus, and nasal cavity, mucosal thickening as in our case. These features may be seen in other diseases, but if CBCT shows bony erosion and sinus involvement in an immunocompromised patient, invasive fungal sinusitis should be one of the differential diagnosis.

However, invasive fungal infections with intracranial and orbital extensions are incompletely evaluated in CBCT. Besides, soft tissue infiltration with fat stranding are unlikely to be recognized in CBCT in such cases, magnetic resonance imaging (MRI) or Multidetector computed tomography (MDCT) are better imaging modalities.[13] Therefore, CBCT may have a role in the early stages of mucormycosis, providing detailed information about extensions into all the sinuses; but if there orbital and intracranial extensions, MDCT or MRI would be the imaging of choice. In our case, absence of any such symptoms of invasive involvement, we performed CBCT, which was more economical, less radiation exposure to the patient and readily available in our institute. CBCT also revealed irregular socket healing with disrupted cortical outline, which was otherwise not apparent in panoramic radiograph. Moreover, intact sinus walls in the CBCT and absence of clinical symptoms ruled out intracranial or orbital extension of the disease.

Confirmatory diagnosis is based on demonstration of the organism in the tissue of a biopsy specimen which reveals presence of broad, nonseptate hyphae with branching at 90 degrees[9] Prompt diagnosis and early initiation of treatment can prevent mortality. Bist SS, et al. recommended four main concomitant treatment strategies, that is rapid improvement of underlying medical conditions, systemic antifungal therapy, surgical debridement and use of hyperbaric oxygen as adjuvant therapy.[9] Amphotericin-B is the mainstay of antifungal therapy in a dose of 1 mg/kg bodyweight with regular monitoring of the kidney functions as Amphotericin-B is known to be nephrotoxic. Aggressive surgical debridement until one encounter the normal bleeding tissue is essential to remove all necrotic tissue.


   Conclusion Top


The present case emphasizes that dental professionals may encounter many patients whose systemic health is compromised. In such scenario, dental treatment such as extraction may promote the growth of opportunistic fungi leading to fatal disease like mucormycosis. Therefore, careful history and appropriate investigations are very essential. Moreover, once the diagnosis is done, immediate treatment and care should be instituted to prevent the mortality. CBCT aided us to recognize the maxillary and nasal extension allowing the early intervention leading to better prognosis of the patient. Hence CBCT gives us detailed description of maxilla, nasal cavity and alveolar involvement. Correlation of clinical and radiographic features helped us arrive at early diagnosis. However, if there is intracranial involvement, advising CBCT will be of limited use, in that case CT and MRI will give better features and soft tissue contrast.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Samantha DR, Senapati SN, Sharma PK, Shruthi BS, Paty PB, Sarangi G. Hard palate perforation in acute lymphoblastoba leukemia due to mucormycosis- A case report. Indian J Hematol Blood Transfus 2009;25:36-9.  Back to cited text no. 1
    
2.
Mowat AG, Baum J. Chemotaxis of polymorphonuclear leucocytes from patients with diabetes mellitus. New Engl J Med 1971;284:621-7.  Back to cited text no. 2
    
3.
Pagano L, Ricci P, Tonso A, Nosari A, Cudillo L, Montillo M, et al. Mucormycosis in patients with haematological malignancies: A retrospective clinical study of 37 cases. Br J Haematol 2003;99:331-6.  Back to cited text no. 3
    
4.
Fain JK, Markowitz A, Khilanani PV. Case report of localized mucormycosis following intramuscular corticosteroid. Am J Med Sci 1978;275:209-16.  Back to cited text no. 4
    
5.
Daly AL, Velazquez LA, Barkley SF, Kauff CA. Mucormycosis: Association with deferroxamine therapy. Am J Med 1989;87:468-71.  Back to cited text no. 5
    
6.
Ugurlu SK, Selim S, Kopar A, Songu M. Rhino-orbital mucormycosis: Clinical findings and treatment outcomes of four cases. Turk J Ophthalmol 2015;45:169-74.  Back to cited text no. 6
    
7.
Regezi JA, Sciubba JJ. Ulcerative conditions. In: Regezi JA, Sciubba JJ, editors. 2nd ed. Oral Pathology Clinical Pathological Correlations. Philadelphia: W B Saunders; 1993. p. 52-60.  Back to cited text no. 7
    
8.
Petrikkos G, Skiada A, Sambatakou H, Toskas A, Valopoulos G, Giannopoulou M, et al. Mucormycosis: Ten years experience at a tertiary care centre in Greece. Eur J Clin Microbiol Infect Disease 2003;22:753-6.  Back to cited text no. 8
    
9.
Bist SS, Varshney S, Bisht M, Gupta N, Bhatia R. Isolated palate ulcer due to mucormycosis. Indian J Otolaryngol Head Neck Surg 2008;60:79-82.  Back to cited text no. 9
    
10.
Diamond RD, Haudenschild CC, Erickson NF. Monocyte mediated damage to rhizopus oryzae hyphae in vitro. Infect Immun 1982;38:292-7.  Back to cited text no. 10
    
11.
Manjunatha BS, Das N, Sutariya RV, Ahmed T. Mucormycosis of the hard palate masquerading as carcinoma. Clin Pract 2012;2:66-8.  Back to cited text no. 11
    
12.
Chen YX, He XE, Zhou H, Wang M, Su SO. Rapidly progressive rhino-orbito-cerebral mucormycosis in a patient with type 2 diabetes: A case report. Exper Ther Med 2017;13:1054-6.  Back to cited text no. 12
    
13.
Miracle A, Guldner C. Diagnosis of sinus pathology using cone beam computed tomography. In: Sarment D, editor. Cone Beam Computed Tomography Oral and Maxillofacial Diagnosis and Applications. Wiley. Blackwell. Inc Iowa USA. 2014 p. 65-7.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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