UNIVERSIDADE ESTADUAL PAULISTA

“JÚLIO DE MESQUITA FILHO”

Instituto de Ciência e Tecnologia

Campus de São José dos Campos

CASE REPORT DOI: https://doi.org/10.4322/bds.2024.e3811

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in

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Maxillary osteomyelitis associated with COVID-19: mucormycosis or not?

A series of cases

Osteomielite maxilar associada ao COVID-19: mucormicose ou não? Uma série de casos

Mayank VERMANI1 , Megha CHOPRA2 , Neeti SWARUP3 , Ankita PAL4 

1 - Sarvodaya Hospital. Hisar, Haryana, India.

2 - MRIIRS: Manav Rachna International Institute of Research and Studies, Faculty of Dental Sciences, Department of Prosthodontics and

Crown & Bridge. Faridabad, Haryana, India.

3 - University of California, School of Dentistry, Center for Oral and Head/Neck Oncology Research. Los Angeles, USA.

4 - Department of Prosthodontics and Crown & Bridge, Santosh Dental College and Hospital, Santosh Deemed to be University, Ghaziabad.

How to cite: Vermani M, Chopra M, Swarup N, Pal A. Maxillary osteomyelitis associated with COVID-19: mucormycosis or not?

A series of cases. Braz Dent Sci. 2024;27(1):e3811. https://doi.org/10.4322/bds.2024.e3811

ABSTRACT

Aim: A series of cases have been presented involving the oral cavity focusing on the presentation, diagnosis

and treatment of mucormycosis that can form a basis for successful therapy. Background: The management of

severe coronavirus disease (COVID-19) in conjunction with comorbidities such as diabetes mellitus, hematological

malignancies, organ transplants, and immunosuppression have led to a rise of mucormycosis which is an

opportunistic infection. Cases Description: The various forms that have been enlisted till date are rhino-

cerebral, rhino-orbital, gastrointestinal, cutaneous, and disseminated mucormycosis. From the dentistry and

maxillofacial surgery perspective, the cases depicting extension of mucormycosis into the oral cavity have been

less frequently recorded and thus, require a detailed study. The patients that reported to our private practice had

non-tender swelling, draining sinuses and mobility of teeth. A similarity was observed in the clinical signs both

in osteomyelitis and mucormycosis. Thus, a histopathological examination was used to establish the denitive

diagnosis. Conclusion: Mucormycosis is a life threatening pathology that requires intervention by other branches

to make an early diagnosis and commence the treatment. The characteristic ulceration or necrosis is often

absent in the initial stage and thus, histopathological examination and radiographic assessment are required

to formulate a denitive diagnosis. Early intervention is a necessity to avoid morbidity. The treatment involves

surgical debridement of the necrotic infected tissue followed by systemic antifungal therapy. Mucormycosis

has recently seen a spike in its prevalence, post the second-wave of coronavirus pandemic in India. It was

seen commonly in patients with compromised immunity, diabetes mellitus, hematological malignancies, or on

corticosteroid therapy. Mucormycosis invading the palate mostly via maxillary sinus has been less frequently

described. In the post-COVID era the features associated with mucormycosis involving oral cavity, should warrant

a possible differential diagnosis and managed appropriately.

KEYWORDS

Eschar; Immunomodulation; Mucormycosis; Palatal ulceration; Rhino-Cerebral.

RESUMO

Objetivo: Apresentar uma série de casos com enfâse na apresentação, diagnóstico e tratamento da mucormicose

oral, assim como uma revisão sistemática que sirva como base para estabelecimento de terapias de sucesso.

Introdução: A forma severa da infecção por coronavirus (COVID-19) associada a diabetes mellitus, doenças

hematológicas malignas, transplante de órgãos e imunossupressão levaram a um aumento das infecções

oportunistas de mucormicose. Descrição dos Casos: As diversas apresentações clínicas que foram descritas

até o momento são a rinocerebral, rino-orbital, gastrointestinal, cutânea e mucormicose disseminada.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

INTRODUCTION

The Coronavirus pandemic (COVID-19),

despite having several prevention and treatment

measures, continues to cause a significant

problem worldwide [1]. According to Raut and

Huy [2], the second wave of COVID-19 has

affected India substantially, with the highest

number of daily reported cases being slightly

more than 0.4 million in the rst week of May’

2021. Kumar et al. [3] and Mahalaxmi et al. [4]

presented mucormycosis as an impending threat,

as the treatment of the same was a major challenge

for the clinician, and that was supposedly

associated with the coronavirus-disease. The

rst case of COVID-19 associated mucormycosis

(CAM) (Rao et al. [5]) was reported in Chile but

most cases were reported in India and were linked

to the Delta variant (B.1.617.2) [5].

‘Mucormycosis’ belongs to the order Mucorales

of the class Zygomycetes, which are ubiquitous,

especially in soil, decaying vegetation and organic

matter such as leaves, compost piles, and animal

dung [2,6]. The ‘black fungus’, is a rare but potentially

fatal infection if there is a lack of treatment. It is also

called mucormycosis and is transmitted by fungal

spores of the Mucorales order [1].

In the second wave, the incidence of

mucormycosis has risen more rapidly in

comparison to the rst wave with around 15,000

cases during the end of May ‘2021 [7]. These fungi

are usually harmless, but can become pathogenic

in certain circumstances like immunosuppression,

diabetic acidosis, steroid therapy and, in transplant

patients [8]. In susceptible patients, there is marked

tissue necrosis of the adjacent structures, followed

by rapid progression and angioinvasion from the

nasal and sinus mucosa into the orbit and brain.

This is known as the Rhino-orbital mucormycosis

and Rhino-cerebral mucormycosis respectively [4].

Currently, the necrosis of hard and soft tissues

of the oral cavity is also being noted, that often

presents as a palatal perforation and is known as

rhino-maxillary mucormycosis [9,10].

The clinical manifestation, diagnosis,

treatment and prognosis of this COVID-19

associated mucormycosis has made the surgeon’s

skeptic as not much is available in literature

at present. Mucormycosis invading the palate

mostly via maxillary sinus has been less frequently

described. In the post-COVID era the features

associated with mucormycosis involving oral cavity,

should warrant a possible differential diagnosis

and managed appropriately. The present article

reports a series of patients having rhino-maxillary

mucormycosis with the aim to direct the attention

towards the clinical presentation and pathogenesis

of mucormycosis and an emphasis on the diagnosis

and treatment part.

CASE DESCRIPTION

A 44-year old afebrile female patient

reported to the department of dentistry, with

a painless and diffused gingival swelling and a

pururlent discharge from the left buccal sinus

with multiple mobile teeth (Grade II mobility)

in the left upper quadrant. (21, 22, 23, 24 and

25) since last 10 days. (Dental and medical

history related to COVID-19 tabulated in Table I).

No que concerne a odontologia e a cirurgia maxillofacial, os casos que apresentam extensão de mucormicose

para cavidade oral tem sido menos reportados e assim requerem mais estudos. Os pacientes que compareceram a

nossa clínica apresentavam aumento de volume endurecido, drenagem de uidos dos seios maxilares e mobilidade

dentária. Clinicamente tanto a osteomielite quanto a mucormicose apresentaram-se de forma semelhante. Assim,

análise histopatológica foi utilizada para estabelecimento do diagnóstico denitivo. Conclusão: A mucormicose

é uma patologia grave que requer intervenção precoce para estabelecimento do tratamento. A ulceração e

necrose características usualmente estão ausentes nos estágios iniciais da lesão, assim análise histopatológica e

radiográca são necessárias para o diagnóstico nal. Intervenção precoce é necessária para diminuir a morbidade.

O tratamento envolve o debridamento cirúrgico da área necrosada seguida de terapia antifúngica sistêmica.

Recentemente, houve um aumento nos casos de mucormicose, após a Segunda onda da pandemia de COVID-19

na índia. Os casos acometiam principalmente pacientes imunocomprometidos, com diabetes mellitus, doenças

hematológicas malignas e em uso de corticosteróides. A mucormicose invadindo o palato pelos seios maxilares

foi raramente descrita. Na era pós-COVID a mucormicose envolvendo a cavidade oral deve entrar no painel de

diagnósticos diferenciais para que o tratamento adequado possa ser instituído precocemente.

PALAVRAS-CHAVE

Necrose; Imunomodulação; Mucormicose; Úlcera palatina; Rinocerebral.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Table I - Characteristics of seven cases reported

Case ID Present

Complaint

Clinical

Findings

Medical

Condition

Haematological/

Serological

Findings

CT/ Radiographic

Findings

Diﬀerential

Diagnosis

Histological

Findings Treatment

Hb1AC/ D

Dimer/CRP

Case 1:

(44/F)

Gingival

swelling,

draining buccal

sinuses, multiple

mobile teeth

(21,22,23,24,25)

Access

opening with

respect to

25, draining

sinuses

present on

the buccal

aspect of

21-25 region,

gingival

swelling

Reported

negative history

for Covid-19

and Diabetes

(DM incidental

ﬁnding)

Hb1AC: 8.1

(raised)

Ill-deﬁned osteolytic

area is seen in

alveolar arch of

maxilla (mainly

towards left side),

ﬂoor of left maxillary

sinus & part of hard

palate, adjacent soft

tissue thickening

was also seen.

Mucosal thickening

seen in left maxillary

sinus-obliteration of

left mucormycosis

unit, mild mucosal

thickening in

left frontal and

ethmoidal sinus.

Endo-perio

lesion,

necrotizing

periodontitis,

osteomyelitis

and chronic

granulomatous

fungal infection

H/E section

demonstrates

necrotic

material,

haematoxylin

stained non

septate

branching

fungal hyphae

Mobile teeth

were extracted,

avascular

alveolar bone

debrided

D Dimer:

1.29(raised)

Post-Operative

Regimen: i.v.

administration

of Liposomal,

lipid

amphotericin

(5mg/kg/day).

CRP: 59.9 (raised) *Blood sugar

levels were

monitored.

Case 2:

(43/M)

Mobile left

upper teeth

(21-28)

Mobility with

respect to

(21-28), i.e,

all the teeth

of upper left

quadrant

Already a

diagnosed

case of

mucormycosis-

was treated

by medial

maxillectomy—

after some

days mobility

observed

and came to

the dental

department, he

had a history of

COVID 1 month

back, was also

hospitalized

during this

(Oxygen support

and steroidal

therapy but no

diabetes)

Mucosal thickening

seen in bilateral

frontal, ethmoid

(left> right) and

maxillary sinus,

thinning of medial

wall of left orbit

with suspicious focal

erosion. Extensive

fat stranding in

the left peri antral

region and adjacent

infratemporal fossa

with mild relative

thickening of the

lateral pterygoid and

temporalis muscles.

Periodontal

infection,

Periapical

lesion,

Osteomyelitis

of maxilla

H/E section

demonstrates

caseous

necrosis,

hematoxylin

stained non

septate

branching

fungal hyphae

with mixed

eosinophilic

and

neutrophilic

inﬁltrate

Surgical

debridement,

extraction of

mobile teeth &

Caldwell Luc

operation was

performed.

*Resected

infected

mass was a

typical case of

Black Fungus

(as it was black

in color)

Case 3:

(62/M)

Non-healing

palatal ulcer

from past 15

days—inability

to open the

mouth

O/E a palatal

ulcer was

found on the

right side in

association

with mobile

16, 17. The

ulcer was

(1*2 cm2)

with irregular

borders: the

ulcer was

covered by

necrotic

slough and

on the lateral

aspect a part

of underlying

bone was also

exposed. Two

ﬁnger mouth

opening. The

ulcer was non

tender but

the patient

complaint of

bad taste.

Reported

positive history

for COVID-

19 a month

back—was

hospitalized for

the same (with

oxygen support

and steroidal

therapy) Also,

was a diabetic.

RBS: 250 mg/dl Mucosal thickening is

seen in right maxillary

and sphenoid sinus

on right side causing

partial opaciﬁcation

of sinuses, Fat

stranding is seen in

right anterior and

posterior periantral

soft tissues. Soft

tissue density

is seen in right

pterygopalatine fossa

with fat stranding.

Fat stranding is

also seen in right

masticator space and

in the region of right

inferior orbital ﬁssure.

Periodontal

infection,

Periapical

lesion,

Osteomyelitis

of maxilla,

Osteomyelitis

(Bacterial,

fungal,

parasitic)

Histopathological

examination

did not test

positive for

mucormycosis---

though the clinical

representation

and medical

history were

same as that

of the other

mucormycosis

patients.

Surgical

resection and

debridement.

Post-operative

regimen was

same as above.

CT: Computed Tomographic Findings. * Post-operative regimen

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Case ID Present

Complaint

Clinical

Findings

Medical

Condition

Haematological/

Serological

Findings

CT/ Radiographic

Findings

Diﬀerential

Diagnosis

Histological

Findings Treatment

Case 4:

(57/M)

C/C of mobile

teeth in right

upper back

tooth region

since 3 days

Mobile teeth

(11, 12, 13, 14,

15, 16) and pus

ﬁlled buccal

sinus that had

pus discharge

on the right

side--- In this

case the mobile

teeth were

extracted—the

underlying

bone was

necrotic

which created

suspicion of

osteomyelitis.

The sample

was sent for

histopathological

examination

along with

the extracted

teeth where

the specimen

tested

positive for

mucormycosis.

Patient

recalled—now

the discharging

(pus) was also

seen on left

side

No history of

COVID and

Diabetes.

There is evidence

of mottled density

involving alveolar

process of maxilla

on right side. Focal

cortical thinning is

seen involving lateral

wall of right maxillary

sinus. Note is made

of mucosal thickening

in bilateral maxillary

sinuses (areas of

polypoidal mucosal

thickening in left

maxillary sinus) with

blockage of right

osteo-meatal unit.

Soft tissue thickening

is seen in right

buccal space with fat

stranding in overlying

subcutaneous plane

Periodontal

infection,

Periapical

lesion,

Osteomyelitis

of maxilla

H/E section

demonstrates

necrotic

material,

hematoxylin

stained non

septate

branching

fungal hyphae,

with occasional

round to ovoid

hematoxylin

stained

structure,

indicative

of conidia

with mixed

eosinophilic

and

neutrophilic

inﬁltrate

Inferior

maxillectomy

was done.

Post-operative

regimen same

as above.

Case 5:

(70/M)

Discoloration

present on the

palate since 15

days which was

progressively

increasing

Discolouration

was present

on the palate.

It was non

tender. (Pt had

HRCT)

Reported

positive history

for COVID 20

days back—was

hospitalized for

the same for

7 days (with

oxygen support

and steroidal

therapy), Also,

was a diabetic

(220mg/dl)

RBS: 220mg/dl Mucosal thickening

with partial

opaciﬁcation is seen

in right maxillary

sinus suggestive

of sinusitis. There

is obliteration of

right osteo-meatal

units. Mild mucosal

thickening is seen in

right ethmoid and left

maxillary sinuses. The

nasal septum is mildly

deviated towards

right side. Concha

bullosa is seen on

right side. Soft tissue

thickening is seen on

right submandibular

and visualized upper

cervical region.

Osteomyelitis

of maxilla

H/E section

demonstrates

necrotic

material,

hematoxylin

stained non

septate

branching

fungal hyphae,

with occasional

round to ovoid

hematoxylin

stained

structure,

indicative

of conidia

with mixed

eosinophilic

and

neutrophilic

inﬁltrate

Treatment not

done (ﬁnancial

reasons)

Case 6:

(56/F)

Ulcer with red

erythematous

boundary with

necrotic slough

seen on the

alveolus of right

maxillary tooth

region

Ulcer with red

erythematous

boundary

and necrotic

slough over

the avascular

bone and they

were seen in

association

with mobile

teeth

(Grade 2 mobile:

13, 14 and 15;

Grade 3 mobile:

16 and 17

No COVID

history, but was

a diabetic since

3 years and was

on medication

Positive for

mucormycosis

(epithelial

ulceration,

inﬂammatory

granulation

tissue giant cell

reaction and

aseptate fungal

hyphae)

Extraction of

mobile teeth

followed by right

hemimaxillectomy.

Post-operative

regimen same

as above.

Case 7:

(46/M)

Palatal

discoloration

(whitish

coloration) that

just started on

the left lateral

slope of palate

that the patient

noticed 3 days

back

Whitish

discoloration

that had

just started

present on

the lateral

slope of

palate at the

junction of

hard and soft

palate

Reported

positive history

for COVID

25 days back--

was hospitalized

for the same for

15 days (with

oxygen support

and steroidal

therapy), Also,

was a diabetic

(221mg/dl)

FBS: 221 mg/dl Minor salivary

gland,

Epidermoid

cyst,

Pleomorphic

adenoma

Histological

examination

was

positive for

mucormycosis

Systemic anti-

fungal therapy:

(Tab.

Isavuconazole-

200mg TID

on Day 1 and

2 and then

200 mg/day for

3-6 months).

CT: Computed Tomographic Findings. * Post-operative regimen

Table I - Continued...

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

After two days of the commencement of the root

canal treatment (RCT), the mobility increased

further and the patient was referred to us.

On investigating the past medical history, the

patient gave negative history for COVID-19

and Diabetes Mellitus. This demanded further

laboratory investigations where, the patient was

diagnosed with diabetes mellitus type II. The

CRP (C - reactive protein) and D-Dimer were

also raised. The patient presented with severe

immunosuppression and a detectable viral

load, that together lead to the formulation of a

provisional diagnosis of necrotizing periodontitis

and a differential diagnosis of endo-perio lesion,

osteomyelitis and chronic granulomatous fungal

infection. (Figure 1) (https://drive.google.com/

le/d/1wA33wXR0BImPxEHiGbOvd2hkxnZt5

a1Q/view?usp=drive_link):

For conrmation, excisional biopsy was done

and the specimen was sent for histopathological

examination. The biopsy conrmed the presence

of necrotic material and hematoxylin-stained

non septate branching fungal hyphae that were

suggestive of mucormycosis. Also, adjacent soft

tissue thickening was seen.

For the management of patient, aseptic

conditions were maintained and under local

anaesthesia the buccal mucoperiosteal ap was

raised where a complete loss of buccal cortical

plate was seen. The mobile teeth were extracted

and the avascular alveolar bone was debrided with

the use of a round bur until fresh bleeding was

observed. Copious irrigation was done and site

was sutured. The intravenous administration of

Liposomal/ lipid Amphotericin (5 mg/kg/day for

5 weeks) was prescribed and the blood sugar levels

were monitored during this time. The patient was

recalled for suture removal after 7 days. (Clinical,

radiological, histological, surgical treatment

pictures presented in Figure 1).

In another case, the patient was diagnosed

with COVID-19 and mucormycosis. Caldwell-Luc

operation was performed for debridement and

the resected infected mass was typical ‘black’

color (Figure 2) (https://drive.google.com/

le/d/1yKH0nF9CqwcCMAAiuC9qk7J4281jA-rY/

view?usp=drive_link).

On the contrary, a 62-year old patient did not

test positive for mucormycosis though the clinical

and medical history were similar to it which made

us skeptic about the diagnosis and, a provisional

diagnosis of Osteomyelitis was made that could

be bacterial, fungal or parasitic. Also, the patient

gave a positive history for COVID-19 and diabetes

(Random Blood Sugar was 250 mg/dl) and he was

hospitalized for the same with oxygen support and

steroidal therapy (Figure 3).

Figure 1 - Clinical presentation of Case 1: (44/F). A. Sinus opening can be demarcated well between the central and lateral incisor region in

the left quadrant. B. 3D volume imaging shows Ill-deﬁned osteolytic area seen in the anterolateral wall of left maxillary sinus, loss of buccal

cortical bone can be appreciated. C: H &E stained sample (20X)revealed non-septate branching fungal hyphae along with necrotic material.

D: Extraction of mobile teeth and debridement of avascular bone (demonstration of the surgical bed after debridement). E: Surgical site

sutured.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Another patient 57 years old with no

COVID-19 history was mucormycosis positive in

histopathological staining.

The detailed ndings and description of the

cases reported is tabulated in Table I.

DISCUSSION

COVID-19 has already claimed over a million

lives worldwide. SARS-CoV-2 are a large group

of viruses that cause illness in humans chiey

Figure 2 - Clinical presentation of Case 2: (43/ M). The patient was already a diagnosed case of mucormycosis (was treated by medial maxillectomy).

A. Clinical presentation of the quadrant where the teeth were mobile. B. Axial section representing thickening of right maxillary sinus mucosal

lining. C. H&E stained sample (10x) revealed caseous necrosis, hematoxylin stained non septate branching fungal hyphae with mixed eosinophilic

and neutrophilic inﬁltrate. D. Presentation at the time of surgery. Characteristic ‘black eschar’ seen. E. Resected infected mass which has a typical

‘black color’ characteristic of mucormycosis. F. Surgical bed after resection of the infected mass. G. Sutured surgical site.

Figure 3 - Clinical presentation of Case 3: (62/ M). A. Intraoral photograph of the patient shows an ulcerative lesion (ulcer measuring 1 * 2 cm2

with irregular borders, covered with necrotic slough present on the lateral aspect of the palate on the right side (underlying bone was also

exposed). B. H&E stained sample (20x) demonstrated occurrence of mixed inﬂammatory inﬁltrate, with presence of giant cells. The sample

did not test positive for mucormycosis, though the clinical representation and medical history were same as that of the other mucormycosis

patients. C. Resected infected mass. D. Surgical bed post resection of infected mass. E. Post-operative picture.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

through the airborne route and droplet release

when the infected person coughs, sneezes and

talks. COVID management ranges from palliative

care at home for mild and moderate cases to

respiratory support in the form of supplemental

oxygen therapy at 5L/min and titration of ow

rates to reach >90% SpO2 mark for the most

severe cases [11,12].

Anti-inammatory or immunomodulatory

therapy comprised of Methylprednisolone (in

doses of 1 to 2 mg/kg IV in 2 divided doses) or

0.2-0.4 mg/kg of Dexamethasone prescribed

usually for a duration of 5 to 10 days. Also,

glucocorticoids and particularly, Remdesivir

has proven to decrease mortality in hypoxemic

patients [13,14].

Extending the total equivalence dose of

immunomodulatory therapy produced major

adverse effects such as hyperglycaemia and

multiple secondary infections. Particularly they

lead to deterioration of the immune system and

predisposes the patients to an increased risk of

contracting fungal like Rhizopus, Mucor and

Thamnostylum. This risk is attributed to the

disrupted neutrophil function and depletion in

native inammatory response [15].

Mucormycosis can be classied into various

clinical forms based on the anatomical site

of involvement like rhino-orbito-cerebral,

pulmonary and cutaneous [13]. Initiation of

the rhino-orbito cerebral or the pulmonary

type starts with inhalation of the fungus

through nose into the paranasal sinus. It has

a tendency to grow along the blood vessels

via the elastic lamina leading to extensive

endothelial damage resulting in the formation

of thrombi and ischaemic necrosis that results

in the characteristic ‘black color’. This necrosed

tissue creates an environment for the fungus

to proliferate. In cases with rhino-orbital

involvement, like involvement of nasal and

sinus walls, a poor blood supply can lead to the

invasion of the orbit via the venous channels and

freely communicating foramen. The involvement

of orbit and fungal invasion through orbital apex

can involve the cranium and ultimately kills the

host [14].

A predisposing factor that has been subjected

as the cause for mucormycosis is the presence

of iron in the host that predisposes patients

with diabetic ketoacidosis to mucormycosis.

Other conditions that make a patient vulnerable

are comorbidities like uncontrolled diabetes

mellitus, diabetic ketoacidosis and neutropenia

and those who had prolonged stay in hospitals

as in India [16,17]. It can be postulated that as

the numbers were on a high during this second

peak, there must have been a sterilization failure

considering the oxygen ventilation masks as the

resources were limited (Figure 4) [18].

The generalized presentation comprises

of one-sided facial pain, numbness or swelling;

toothache, loosening of teeth (as in the case

series in this article: the major chief complaint

was sudden mobility of teeth); blurred or double

vision with pain, fever, skin lesion, thrombosis

and necrosis, in some cases, chest pain, pleural

effusion, hemoptysis and worsening of respiratory

symptoms [19]. Similar oral findings were

described by Mohanty et al. [20], rhino-maxillary

mucormycosis frequently presents as osteomyelitis

of the maxillae which is associated with facial

swelling, black discoloration, mobility of teeth

and palatal perforations. The rhinomaxillary

form is considered to be a less fatal form of

the rhinocerebral type of mucormycosis. The

case series presented in this article aims at

the diagnosis and treatment of this rhino-

maxillary form of mucormycosis having palatal

involvement.

Figure 4 - Risk factors predilection for mucormycosis in India Post-

Covid-19.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

The management of mucormycosis mainly

involves monitoring and controlling diabetic

ketoacidosis, reduction of steroids, discontinuation

of immunomodulatory drugs, extensive surgical

debridement to remove all the necrotic debris

material, and maintenance of adequate systemic

hydration, and antifungal therapy for at least 4-6

weeks [21].

Of the seven cases reported so far, two of

them belonged to the rural background and the

rest were from urban setup in India. The mean

age was 54 years and 5 were men. Five patients

had a positive history for COVID-19 and one

patient did not report any history for COVID-19

but on investigation D-Dimer and CRP, both were

raised; that are generally raised post-COVID-19

(the patient did not go for examination of post-

COVID-19 antibody titre). The ve patients that

gave a positive COVID-19 history had acute

respiratory distress syndrome (ARDS) and they

were hospitalized for oxygen support, where the

ventilation mechanisms also may have acted as

a risk factor for mucormycosis.. Six cases out of

seven reported, presented with symptoms such as

mobility of teeth, purulent sinuses, palatal ulcers

with irregular borders covered with a necrotic

slough (the ulcers were generally non-tender)

and palatal discolorations that were suggestive

of mucormycosis. The clinical presentation in

one of the cases was a non-tender palatal ulcer

with irregular borders and exposed avascular

bone presentation exactly like that of a typical

mucormycosis patient but the histology reports

did not reveal fungus presence.

Differential diagnosis of the lesion should

include squamous cell carcinoma, osteomyelitis

(bacterial/ fungal/ parasitic), periodontal infection,

periapical lesion, palatal minor salivary gland,

epidermoid cyst, pleomorphic adenoma and chronic

granulomatous infection like tuberculosis [22,23].

The lesion appears as a persistent chronic ulcer

with raised margins suggestive of Squamous Cell

Carcinoma, however, when this lesion is present

with a history of diabetes and immunosuppression,

then a diagnosis of fungal infection is favoured,

which is later confirmed by histopathological

investigation.

Six out of the seven reported cases were

treated with surgical debridement of the necrotic

avascular alveolar bone along with the extraction

of teeth with mobility. Copious irrigation was

done throughout this procedure. Amphotericin-B

was prescribed and daily monitoring of blood

sugar and other vitals was done. Hyperbaric

oxygen therapy, granulocyte colony-stimulating

factor (G-CSF) and topical application of

Amphotericin-B could have been used as adjuncts

in the treatment but were not used in any of the

cases [24]. Though mucormycosis has a high

mortality rate, the six patients who underwent

treatment have survived and are responding well.

CONCLUSION

An increase in number of mucormycosis cases

has been noted post-second wave of COVID in India.

It is a life-threatening and an aggressive pathology

that involves inter professional cooperation to make

an early diagnosis and prompt treatment. The trio

of diabetes, excessive use of corticosteroids in the

COVID-19 scenario appears to be the main causal

of mucormycosis. The characteristic feature of

ulceration or necrosis is often absent in the initial

stages of infection. The definitive diagnosis is

formulated in assistance with advanced radiological

techniques such as CT, and histopathological

investigation. The treatment ranges from surgical

debridement of the dead, infected and necrotic

tissue to systemic antifungal therapy in the form

of amphotericin-B or the more effective liposomal

amphotericin-B. Dentists and surgeons at their

end, have a chance to mark a diagnosis and treat

this fatal rhino-maxillary form of mucormycosis to

reduce the morbidity and mortality rate.

Author’s Contributions

The author’s contributions are as follows:

MV: Conceptualization, Manuscript

Design, Manuscript Writing, Review. MC:

Conceptualization, Literature Search, Manuscript

Writing and Review. NS: Conceptualization,

Investigation, Manuscript Preparation. AP:

Manuscript Review.

Conict of Interest

No conicts of interest declared concerning

the publication of this article.

Funding

The authors declare that no nancial support

was received.

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Regulatory Statement

This study was conducted in accordance with

all the provisions of the local human subjects

oversight committee guidelines and policies of:

Institutional Ethics Committee of Sarvodaya

Multispeciality & Cancer Hospital.

The approval code for this study is: ECR/999/

Inst/HR/2021. ANS: The policies and approval

code have been updated in the above comments.

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Megha Chopra

(Corresponding address)

MRIIRS: Manav Rachna International Institute of Research & Studies, Faculty of

Dental Sciences, Department of Prosthodontics and Crown & Bridge, Faridabad,

Haryana, India.

Email: drmeghachopra@gmail.com

Date submitted: 2023 Feb 17

Accept submission: 2023 Sep 22

Braz Dent Sci 2024 Jan/Mar;27 (1): e3811

Vermani M et al.

Maxillary osteomyelitis associated with COVID-19: mucormy cosis or not? A series of cases

Vermani M et al. Maxillary osteomyelitis associated with COVID-19:

mucormycosis or not? A series of cases

Appendix 1. Supplementary les.

Case 1: https://ojs.ict.unesp.br/index.php/cob/article/view/3811/4694

Case 2: https://ojs.ict.unesp.br/index.php/cob/article/view/3811/4695

Case 4: https://ojs.ict.unesp.br/index.php/cob/article/view/3811/4696

Case 6: https://ojs.ict.unesp.br/index.php/cob/article/view/3811/4697