A rare entity to major outbreak: a case report on mucormycosis
Dhaman Gupta1, Tanvi Dosi2,&
1Department of Public Health Dentistry, Sri Aurobindo College of Dentistry, Indore, Madhya Pradesh, India, 2Department of Oral Medicine and Radiology, Sri Aurobindo College of Dentistry, Indore, Madhya Pradesh, India
Tanvi Dosi, Department of Oral Medicine and Radiology, Sri Aurobindo College of Dentistry, Indore, Madhya Pradesh, India
Mucormycosis is relatively uncommon, fulminant, progressive, life threatening fungal disease which is most often seen in debilitating patients with immunocompromised condition. Mucormycosis cases are seen in patients with the use of systemic steroids in the treatment of severely affected COVID-19 cases and also in the patients with uncontrolled diabetes which causes immunosuppression are being reported with mucormycosis. The main symptoms of this disease include pain on the temporal and the orbital region of the affected side which could be throbbing or lancinating type, mobility of the teeth, jaw pain and often swelling is present which could be extraoral and intraoral both or sometimes only intraorally. The diagnostic approach in such cases is done with the help of clinical diagnosis, histopathology and with advanced imaging like cone beam computed tomography, magnetic resonance imaging and computed tomography. We here used cone beam computed tomography imaging that revealed haziness in the sinuses and breach in cortical bone of the affected area which confirmed the diagnosis of mucormycosis. Early treatment planning like administration of antifungal drugs and surgical debridement will be life saving in such a deadly disease.
Mucormycosis is a fungal disease associated with long term morbidity and mortality [1,2]. It is caused by fungi of the order mucorales . After candidiasis and aspergillosis, mucormycosis is the third most common fungal infection [4,5]. This disease was described by Furbinger in 1976 and first case was published by Arnold Paltauf in 1885 . Around 27 species and eleven genera under mucorales are associated with infections in humans. Mostly mucormycosis is caused by rhizopus arrhizus followed by apophyromyces, lichtheimia, mucor, rhizomucor and cunninghamella species . Port of entry of mucormycosis is by direct contamination of open oral wounds or by inhalation of spores . Mucormycosis commonly affects orbit, nose, paranasal sinuses and central nervous system . COVID-19 pandemic continues to be a major problem around the world. Though various options have been used but systemic steroids found to be effective in the survival of COVID-19 . But the use of systemic steroids can lead to fungal or secondary bacterial infection . High prevalence of diabetes patients are seen in India . Systemic steroid and underlying condition like uncontrolled diabetes after recovering from COVID-19 are well known risk factors for mucormycosis reported in literature. This paper reports two such cases.
Patient and observation
Information for the patient: a 58-year-old male, reported to the outpatient department, having pain in the right maxillary quadrant. Pain was continuous and lancinating in nature and radiated to temporal region resulting in a headache. Patient suffered from COVID-19 a month ago. Patient took medications for 5 days along with steroid therapy. There was history of diabetes since 4 years.
Clinical results: on extra-oral examination, there was a diffuse swelling on the left side of the face which was tender on palpation. Pertaining to the pain, patient had undergone extraction of right maxillary third molar, 20 days ago with no pain relief; ruling out the possibility of the tooth being the cause of pain. Intra-oral examination revealed mobility in all the teeth of first quadrant. Other intraoral findings included inflammation of palatal gingiva extending up to the mid-palatine raphe which was tender when palpated and exhibited the cardinal signs of inflammation.
Diagnostic procedure: cone beam computed tomography (CBCT) revealed; breach in the buccal and palatal cortical bone and illustrated scattered hypodense areas in the right anterior and posterior region. In conjunction with these findings, there was also evidence of discernible haziness and bone destruction along the floor, lateral wall and medial wall of the right and left maxillary sinus (Figure 1, Figure 2, Figure 3).
Therapeutic intervention and follow-up: patient was admitted in the other hospital for surgical debridement and antifungal drug amphotericin B was administered for the patient´s recovery.
Information for the patient: a 60-year-old male, reported in the department with the complaint of pain in the right upper posterior teeth region with a diffuse swelling in right facial region. Patient had a history of COVID-19 with 20% lung involvement for which he was admitted. The treatment received included, oxygen therapy and steroid administration for approximately five days. He had diabetes since 7 years. Around 15 days later, he started experiencing pain and mobility in maxillary posterior tooth region which was throbbing type and continuous in nature radiating towards right temporal and orbital region.
Clinical results: on examination, diffuse extra oral swelling was present on the right side of face. Extension of the swelling superiorly extending upto infraorbital rim and inferiorly till the inferior border of mandible which was soft in consistency and tender on palpation. On intraoral examination, gingival enlargement was observed with blackish discoloration on facial gingiva posteriorly. Mobility was noted in anterior as well as posterior maxillary teeth.
Diagnostic procedure: cone beam computed tomography (CBCT) was advised as the suitable radiographic imaging modality which revealed scattered hypodensity present in anterior as well as posterior maxillary region along with loss of buccal cortical bone seen in posterior aspect as seen in the axial plane. Paranasal air sinuses involving right maxillary, sphenoid and ethmoid sinuses encased a hazy component giving soft tissue appearance with breaching in medial and lateral wall of maxillary sinus (Figure 4, Figure 5, Figure 6).
Therapeutic intervention and follow-up: to attain the better result patient was advised for surgical debridement and antifungal drug amphotericin B which was administered in the other hospital.
World is already facing COVID-19 pandemic, emergence of new outbreak of mucormycosis. Mucormycosis is a rare life threatening black fungal infection associated in patients recovered from COVID-19 . Most of the time, pathogens are found in soil, fruits, manure and dust. It can be obtained from oral cavity, throat, nasal passage and sometimes in healthy patients´ stool also . Port of entry is by direct contamination through skin laceration and by inhalation of spores through mouth or nose . Sino-orbital or pansinusitis type of mucormycosis is found commonly in patients with uncontrolled diabetes and those who are administered with systemic steroids [16,17]. Normally fungi are avirulent; they become virulent when the patients´ immunity becomes low generally happens in immunocompromised patients .
Diabetes mellitus also increases fungal proliferation. Patients recovered from COVID-19 having uncontrolled diabetes are known to have significantly low host defence mechanism thus reducing phagocytic and chemotaxis efficiency. Diabetes patients produce keto-reductase enzyme which allow mucormycosis to utilize ketone bodies. Diabetes also alters the activity of transferrin which binds the iron resulting in growth of mucormycosis . Systemic steroids are inexpensive and have shown beneficial effects in COVID-19 patients but also increase the risk of secondary infections . Therefore, both uncontrolled diabetes and steroid use in covid recovering patients are associated factors for pansinusitis or rhino-sinusitis. The symptoms presenting in both the cases of pansinusitis were pain in temporal and orbital region resulting in headache, swelling near infra orbital rim, mobility of teeth, inflammation, enlargement and blackish discoloration of facial gingiva and mild palatal swelling. It initiates from oral and nasal mucosa spreading to all the paranasal sinuses.
To detect the spread and propagation of mucormycosis cone beam computed tomography (CBCT) imaging was used. CBCT revealed areas of hypo density and breach in buccal and cortical bone with evidence of discernible haziness, bone destruction along the floor, lateral and medial wall of right and left maxillary sinus. Sinus thickening and haziness of ethmoid and sphenoid paranasal sinuses were also observed. Surgical debridement and anti fungal drugs like amphotericin B can be administered in such cases to attain the maximum for the patient´s recovery.
Mucormycosis is a relatively uncommon, fulminant, progressive and life threatening fungal disease mostly occurring in the debilitating patients with immuno compromised condition. It is usually associated with patients having uncontrolled diabetes and the use of systemic steroids in the treatment of severely affected COVID-19 cases. Early diagnosis and treatment of mucormycosis which include antifungal drugs and surgery can improve the survival and outcome.
The authors declare no competing interests.
Dhaman Gupta and Tanvi Dosi: concept of the study, review of literature, data collection, data entry, data analysis and final manuscript writing and overall final manuscript preparation. All the authors have read and agreed to the final version of the manuscript.
Figure 1: three-dimensional view showing bony destruction in right maxillary quadrant
Figure 2: axial cone beam computed tomography (CBCT) view shows loss of buccal cortical bone
Figure 3: bone destruction and haziness in maxillary sinus shown in axial cone beam computed tomography (CBCT) imaging
Figure 4: three-dimensional view showing bony destruction in right maxillary quadrant
Figure 5: maxillary and sphenoid sinuses showing haziness
Figure 6: ethmoid sinus showing mild haziness and thickening of sinus lining
- Oswal NP, Gadre PK, Sathe P, Gadre KS. Mucormycosis of mandible with unfavorable outcome. Case Rep Dent. 2012;2012:257940. PubMed | Google Scholar
- Reddy IS, Rao NR, Reddy VMS, Rao R. Primary cutaneous mucormycosis (zygomycosis) caused by apophysomyces elegans. Indian J Dermatol Venereol Leprol. 2008;74:367-70. PubMed | Google Scholar
- Revannavar SM, Supriya PS, Samaga L, Vineeth VK. COVID-19 triggering mucormycosis in a susceptible patient: a new phenomenon in the developing world. BMJ Case Rep. 2021;14(4):e241663. PubMed | Google Scholar
- Pandilwar PK, Khan K, Shah K, Sanap M, Anoop Unnikrishnan KS, Nerurkar S. Mucormycosis: a rare entity with rising clinical presentation inimmunocompromised hosts. Int J Surg Case Rep. 2020;77:57-61. PubMed | Google Scholar
- Mekki SO, Hassan AA, Falemban A, Alkotani N, Alsharif SM, Haron A et al. Pulmonary mucormycosis: a case report of a rare infection with potential diagnostic problems. Case Rep Pathol. 2020 Jan 6;2020:5845394. PubMed | Google Scholar
- Skiada A, Pavleas I, Drogari-Apiranthitou M. Epidemiology and diagnosis of mucormycosis: an update. J Fungi (Basel). 2020 Nov;6(4):265. PubMed | Google Scholar
- Prakash H, Chakrabarti A. Global epidemiology of mucormycosis. J Fungi (Basel). 2019 March 21;5(1):26. PubMed | Google Scholar
- Bakathir AA. Mucormycosis of the jaw after dental extractions: two case reports. Sultan Qaboos Univ Med J. 2006 Dec;6(2):77-82. PubMed | Google Scholar
- Nilesh K, Vande AV. Mucormycosis of maxilla following tooth extraction in immunocompetent patients: reports and review. J Clin Exp Dent. 2018 March 1;10(3):e300-e305. PubMed | Google Scholar
- Garg D, Muthu V, Sehgal IS, Ramachandran R, Kaur H, Bhalla A et al. Corona virus disease (COVID-19) associated mucormycosis (CAM): case report and systematic review of literature. Mycopathologia. 2021;186(2):289-298. PubMed | Google Scholar
- Veisi A, Bagheri A, Eshaghi M, Rikhtehgar MH, Rezaei Kanavi M, Farjad R. Rhino-orbital mucormycosis during steroid therapy in COVID-19 patients: a case report. Eur J Ophthalmol. 2021 April 10;11206721211009450. PubMed | Google Scholar
- Mehta S, Pandey A. Rhino-orbital mucormycosis associated with COVID-19. Cureus. 2020 Sep 30;12(9):e10726. PubMed | Google Scholar
- Patil S, Sarate D, Chopade S, Khade M, Dhage S, Kangate S. Emerging challenge of mucormycosis in post-COVID patients. IAR J Med Cse Rep. 2021 May 10;2(3):7-10. Google Scholar
- Kim J, Fortson JK, Cook HE. A fatal outcome from rhinocerebral mucormycosis after dental extractions: a case report. J Oral Maxillofac Surg. 2001 Jun;59(6):693-7. PubMed | Google Scholar
- Sugar AM. Mucormycosis. Clin Infect Dis. 1992 Mar;14 Suppl 1:S126-9. PubMed | Google Scholar
- Papadogeorgakis N, Parara E, Petsinis V, Vourlakou C. A case of successfully treated rhino cerebral mucormycosis: dental implications. Int J Dent. 2010;2010:273127. PubMed | Google Scholar
- Ahmadikia K, Hashemi SJ, Khodavaisy S, Getso MI, Alijani N, Badali H et al. The double-edged sword of systemic corticosteroid therapy in viral pneumonia: a case report and comparative review of influenza-associated mucormycosis versus COVID-19 associated mucormycosis. Mycoses. 2021 Feb 16;10.1111/myc.13256. PubMed | Google Scholar