People’s Journal of Scientific Research / Volume 16 / Issue 1 / Jan-June 2023
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Mucormycosis- A Case Report
Tushar Phulambrikar, Sanjana Moses, Tanvi Dosi
Department of Oral Medicine and Radiology, Sri Aurobindo College of Dentistry, Indore
ABSTRACT:
Mucormycosis, an aggressive and opportunistic fungal infection caused by Rhizopus sp., Mucor, and
Lichtheimia, poses a significant challenge in the post-COVID era. Previously considered a rare
occurrence, mucormycosis has witnessed a surge in cases, particularly affecting the nose, paranasal
sinuses, and cerebral tissue. These fungal pathogens exhibit a destructive behaviour, eroding small
blood vessels and leading to thrombosis, ischemia, and tissue necrosis. Patients with compromised
systemic health, such as diabetes mellitus, leukemia, and immunosuppressive therapy, are particularly
susceptible to this infection due to impaired immunity.
The various clinical manifestations of mucormycosis are categorized into rhinocerebral, pulmonary,
cutaneous, gastrointestinal, and disseminated forms. Within the rhinocerebral form, subdivisions based
on the affected tissues further refine the classification like rhino-orbital, rhino-sino-orbital, rhino-
orbito-cerebral.
Fungal culture remains a cornerstone for identifying the causative organism, while magnetic resonance
imaging is the gold standard for radiological evaluation, offering detailed imaging of the affected
regions. Computed tomography scans also play a crucial role in the diagnostic pathway.
With dental practitioners encountering an increasing number of mucormycosis cases, Cone Beam
Computed Tomography has emerged as a valuable diagnostic tool. Recent advancements have led to the
development of diagnostic criteria based on CBCT findings, aiding in the accurate and timely diagnosis
of mucormycosis. We report a case of mucormycosis affecting maxilla highlighting the importance of
CBCT in addition to conventional diagnostic methods thereby improving its management and clinical
outcome.
KEYWORDS: mucormycosis; COVID-19; CBCT.
Address for correspondence : Dr Tushar Phulambrikar, HOD, Department of Oral Medicine and Radiology, Sri Aurobindo College of
Dentistry, Indore-Ujjain Highway, Indore-453555 (Madhya Pradesh)
E-mail: drtushar@hotmail.com
Submitted: 15.06.2023, Accepted: 19.06.2023, Published: 26.06.2023
INTRODUCTION:
Mucormycosis is an opportunistic fulminant
fungal infection caused by Rhizopus sp., which most
commonly affects nose, paranasal sinuses and cerebral
tissues. Rhizopus along with mucor and lichtheimia
account for about 70 - 80 % of all cases of
mucormycosis. These fungi have the tendency to erode
and invade small blood vessels and lead to thrombosis,
ischemia and tissue necrosis[1]. It is known to occur in
patients with compromised systemic health as in
diabetes mellitus, leukemia, prolonged corticosteroid
therapy, chronic renal failure, antineoplastic therapy,
immunosuppressive therapy, deferoxamine therapy,
protein calorie malnutrition owing to the impaired
immunity in these patients[2]. Mucormycosis is
categorized into rhinocerebral (most common form),
pulmonary, cutaneous, gastrointestinal and
disseminated. Rhinocerebral form can be further
subdivided depending on the tissues affected as- rhino-
nasal or rhino-maxillary, rhino-orbital, rhino-cerebro-
orbital[3].
Diagnosis is based on the history, clinical
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presentation and investigations. Fungal culture is done
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People’s Journal of Scientific Research / Volume 16 / Issue 1 / Jan-June 2023
Phulambrikar T et al. Mucormycosis
Figure 1: Intraoral clinical presentation showing enlarged gingiva and palatal changes in the region of 16, 17.
to visualize the causative organism and the gold
standard in radiological evaluation is Magnetic
Resonance Imaging (MRI), followed by Computed
Tomography (CT) scan[4].
In the post COVID era, there has been a surge
of cases of mucormycosis which was a seldom
occurrence earlier[5]. As Dental practitioners, we
encountered a myriad of cases and evaluated them for
the signs and symptoms and conducted investigations,
in this case, Cone Beam Computed Tomography
(CBCT). The recent development of diagnostic criteria
based on CBCT findings aids in the diagnostic process.
We present a case of Mucormycosis affecting
maxilla in a diabetic patient with a history of COVID-
19. A 54 year old male patient reported to the
department of Oral Medicine and Radiology at Sri
Aurobindo College of Dentistry, with the chief
complaint of pain in the right side of his jaw since 7
days. The patient was apparently alright a week ago
when he started experiencing pain in the right quadrant
of maxilla which was dull, gnawing and continuous in
nature which aggravated on chewing. The patient's
medical history was significant for a recent COVID-19
i n f e c t i o n 3 m o n t h s a g o a c c o m p a n i e d b y
hospitalisation in the ICU for 10 days. Corticosteroids
along with antiviral drugs were administered to the
patient, after which he was discharged. His medical
history revealed that he had diabetes for 4 years and
was under medication. His foregoing reports were
suggestive of his blood sugar levels being consistently
>140 mg/dl.
On extra oral examination, there was no gross
facial asymmetry, no pain or tenderness on palpation.
The patient was evaluated for any sinus abnormality
through palpation which yielded a negative result. On
intra oral examination, swelling on the marginal and
attached gingiva was observed which was non
suppurative, fibrous and non tender on palpation and
gentle probing [Figure 1]. On further examination,
there was a diffuse slight tumescence on the right
posterior part of hard palate corresponding to the teeth
16 and 17 with mild tenderness on palpation and no pus
discharge. The teeth in the first quadrant had no
mobility, but examination of maxilla revealed the
presence of segmental mobility in the first quadrant.
Based on the history and clinical findings, a
provisional diagnosis of mucormycosis of the maxilla
was made and differentials included osteomyelitis,
chronic granulomatous infection, and deep fungal
infections.
A CBCT scan was advised and it indicated loss
of cortical plate in the region of the first quadrant. On
further perusal, the scan revealed an isodensity in the
lumen of right and left maxillary sinuses suggestive of
sinusitis. A radiographic diagnosis of Mucormycosis
involving maxillary arch and invasive fungal sinusitis
was made [Figure 2,3 & 4].
Phulambrikar T et al. Mucormycosis
People’s Journal of Scientific Research / Volume 16 / Issue 1 / Jan-June 2023
71
hypoattenuation in the lumen of the maxillary sinus.
Figure 2: Shows an axial section of CBCT which depicts the loss of buccal cortical plate along with osteopenia in the region of 15, 16, 17.
Figure 3: Sagittal section of the scan gives an insight of the bone destruction. Figure 4: Axial section of the scan shows the isodensity or soft tissue
Figure 5: Post operative picture showing the sutured area of edentulous space Figure 6: Post operative OPG.
Diagnostic Criteria for Rhinomaxillary mucormycosis:
6]
A.
Involvement of at least one maxillary sinus
B.
Osteolytic changes in maxillary alveolar bone
with or without involvement of other
mentioned bones
C.
Presence or absence of dental findings
DISCUSSION:
M u c o r m y c o s i s i s a n o p p o r t u n i s t i c ,
angioinvasive and deep fungal infection affecting the
nasal cavity, paranasal sinuses, involving eye and
ultimately the brain. It most commonly affects
immunocompromised patients but healthy patients are
not an exception. Infection arises after inhalation
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People’s Journal of Scientific Research / Volume 16 / Issue 1 / Jan-June 2023
Phulambrikar T et al. Mucormycosis
through the nose and hence affects the nasal cavity and
then spreads to the surrounding tissues[1].
In 1855, Kurchenmeister described a case of
Mucormycosis in a patient of neoplastic lung on the
basis of its histoplasmology which was probably the
first authentic human case.
In 1876, Furbringer described pulmonary
mucormycosis for the first time which was caused by
Absidia.The first case in humans was reported in 1885
by Paultauf[2].The first publication was done by Gregory
et al, of the first observation of rhino-orbital cerebral
mucormycosis in 1943.The first report was by Harris in
1955 of the first known survivor[4].
According to WHO, the incidence rate of
mucormycosis globally varies from 0.005 to 1.7 per
million population in 2020[7]. A 10-year study from
Tamil Nadu showed an annual incidence of 18.4 cases
per year during 20052015[8]. Another study from Tamil
Nadu reported 9.5 cases per year during 20152019[8]. A
multicentre study across India reported 465 cases from
12 centres over 21 months; the study reported an annual
incidence of 22 cases per year, and an average of 38.8
cases for each participating centre[8]. Chakrabarti et al
showed an increasing trend of mucormycosis, with an
annual incidence of 12.9 cases per year during 1990
1999, 35.6 cases per year during 20002004, and
50 cases per year during 20062007. The overall
numbers increased from 25 cases per year (19902007)
to 89 cases per year (20132015)[9]. Though invasive
aspergillosis is given importance among invasive
mould infections in intensive-care units (ICUs), a
multicentre s tudy in Indian ICUs reported
mucormycosis in a considerable (14%) number of
patients[9]. Without population-based estimates, it is
difficult to determine the exact incidence and
prevalence of mucormycosis in the Indian population.
The computational-model-based method estimated a
prevalence of 14 cases per 100,000 individuals in India.
In India, prevalence of mucormycosis is about 80 times
higher than the prevalence in developed countries[7].
In India, Diabetes mellitus has been the most
common risk factor linked with mucormycosis[9]. Other
causes include haematological malignancy and
chemotherapy, haematopoietic stem cells, and solid-
organ transplant recipients on immunosuppressive
therapy, with iron overload, on peritoneal dialysis,
extensive skin injury, human immunodeficiency virus
(HIV) infection, and voriconazole therapy[10].
Along with these, COVID-19 disease has a
propensity to cause extensive pulmonary disease and
subsequent alveolo-interstitial pathology. This by itself
may predispose to invasive fungal infections of the
airways including the sinuses and the lungs.
Furthermore, there is an alteration of the innate
immunity due to COVID-19-associated immune
dysregulation characterized by decreased T cells,
including CD4 and CD8 cells[11]. Other factors like
steroid administration, immunomodulating drugs like
tocilizumab, and high doses of Vitamin C, oxygen
therapy, and prolonged hospitalization predispose the
development of mucormycosis.
Initial clinical assessment calls for an
investigation; MRI being the modality of choice to
evaluate the extent of disease and prognostication. Next
in line being CT although it has been observed that
CBCT is one of the most underrated imaging modalities
in the early diagnosis of mucormycosis as evidenced by
the paucity of literature. As of late, CBCT has been
considered as the examination of choice in various
instances, since it gives high resolution imaging,
diagnostic consistency and risk benefit assessment[6].
In the case mentioned in this report, we
evaluated the patient through proper history, assessing
the clinical signs and symptoms, subjecting the patient
to available modality i.e. CBCT and applying the
diagnostic criteria based on CBCT findings to
formulate a diagnosis.
The clinical findings included gingival
enlargement, diffuse swelling involving hard palate and
segmental mobility. In severe cases, a necrosed
ulcerative area forms over the palate owing to the
thrombosis which eventually causes exposure of the
non-vital bone[10].
CBCT images show loss of cortical bone along
with obliteration of right and left maxillary sinus
suggestive of invasion of sinus lumen.
In cases where the infection has spread through
the other paranasal sinuses, ocular involvement takes
place through the thin lamina papyracea of the ethmoid
bone into the orbit resulting in rhino-sino-orbital
mucormycosis[10].
The treatment for mucormycosis infections
involves the utilization of Amphotericin B, which is the
initial FDA-approved drug for this purpose.
Amphoterecin B is available in different lipid
formulations, including liposomal preparation, lipid
complex, and colloidal dispersion. These formulations
enhance the effectiveness of the drug and improve its
administration to the affected areas, thereby assisting in
the treatment of mucormycosis infections, administered
in the dose of 10 mg/kg/day.[12] A dosage of 5mg/kg/day
is recommended in COVID-19 associated Mucormy-
cosis. An oral suspension of Posaconazole
800mg/24hrs in 2 divided doses is advised for 12-13
weeks[13].
People’s Journal of Scientific Research / Volume 16 / Issue 1 / Jan-June 2023
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Phulambrikar T et al. Mucormycosis
Necrosis and thrombosis occurring during
mucormycosis can lead to inadequate delivery of
antifungal medications, making it crucial to consider
the removal of affected tissue as an essential aspect of
care for complete eradication of the infection. It is
important to acknowledge that predicting surgical
outcomes in mucormycosis cases is challenging due to
biases in patient selection. Reports have indicated that
surgical management in patients with rhino-orbito-
cerebral mucormycosis yields better results compared
to non-surgical treatment, enabling local control of the
infection.
CONCLUSION:
Early detection and aggressive management
are paramount in eradicating mucormycosis. Thorough
clinical assessment, meticulous patient history, and
comprehensive investigations are prerequisites for
accurate diagnosis. In settings with limited resources,
CBCT serves as an invaluable resource for visualizing
radiographic images. This case report underscores the
significance of astute clinical and radiographic
examination in facilitating an early and expeditious
diagnosis of mucormycosis. By emphasizing the
pivotal role of these diagnostic approaches, healthcare
providers can augment their capacity to promptly
identify and treat mucormycosis, ultimately leading to
improved patient outcomes.
Financial Support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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