Article Type : Case Report
Authors : Gaurav G, Gupta DK, Gupta N, Priyanka G and Wadhwa S
Keywords : COVID-19; Maxillary necrosis; Thromboembolism; Pneumonia
In
late 2019, a novel coronavirus whichemerged as the cause of a cluster of
pneumonia cases, in Wuhan, a city of the Hubei Province in China has now
designated as severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) [1].
Around the world it has spreaded quickly resulting in a pandemic [2]. The
disease caused by the SARS-CoV-2 was named as coronavirus disease 2019
(COVID-19) by World Health Organization (WHO) [3], COVID-19 affected more than
18 million people and around 700,000 people died according to the last WHO
report [4].
The
clinical spectrum of COVID-19 ischaracterized by respiratory failure, shock
and/or multiorgan dysfunction, which ranges from mild infection with or without
pneumonia, severe pneumonia with respiratory distress and/or hypoxia in ambient
air to critical disease [5]. In patients with severe disease, the acute
respiratory distress syndrome (ARDS) is the most frequent complication (19.6% -
29%)
[6,7], resulting in a remarkable hypoxemic respiratory failure. However,
several other clinical events could aggravate the course of COVID-19 including
thromboembolic events.
Due to rich vascularity, maxilla rarely undergoes necrosis
and if at all necrosis occur it is due to bacterial, viral, or fungal
infections. Necrosis mainly infects immunocompromised patients. The infection
begins in the nose and paranasal sinuses due to inhalation. Either by direct
invasion or through the blood vessels the infection can spread to orbital and
intracranial structures [8].
It occurs by invading the arteries leading to thrombosis that
subsequently causes necrosis of hard and soft tissues. In
the medical literature there are no reported cases of post covid-19 dental
complications. Here we report a case of
maxillary avascular necrosis by maxilla after a thromboembolic event,
causing impaired vascular supply in an
uncontrolled diabetic patient to emphasize early diagnosis of this potentially
fatal disease. Early diagnosis and prompt treatment can reduce the mortality
and morbidity of this lethal disease.
A 32-year-old diabetic male reported with inability in chewing from one side since few days. Patient gave history of positive Covid-19 hospitalization a month back and recovered well in 10 days thereafter. On intra-oral clinical examination, multiple pus drainage sinuses were seen on the left maxillary teeth alveolus starting from left upper central incisor to left upper second molar. All teeth were grade two mobiles but non carious (Figure 1a-c).
Figure 2: Routine Blood examination done.
All routine blood examinations came normal (Figure 2). 3D CT scans were done which showed irregular osteolytic lesions in whole of the left maxillary alveolus involving maxillary sinus as well (Figure 3a-c).
Figure 3a,b,c: 3D CT scan done.
Looking at the severity of lesion it was planned to extract the involved teeth, curettage the lesion, removed the complete inflamed bone and sinus lining under local anesthesia. Mucoperiosteal flap was raised till healthy bone is seen, mobile teeth were removed &along with it the necrotic bone came out (Figure 4a-e).
Figure 4a-f: Mucoperiosteal flap was raised till healthy bone is seen, mobile teeth were removed &along with it the necrotic bone.
Infected sinus lining was completely removed (Figure 5).
Figure 5: Removal infected sinus lining.
Iodoform dressing was packed in the cavity for hemostasis followed by 3.0 black silk wherein primary closure of the defect was achieved (Figure 6a,b). Three specimens were collected from inflamed soft tissue, necrosed bone and maxillary sinus lining and sent for histopathological examination.
Figure 6 a,b: Iodoform dressing was done with suturing.
On third day, iodoform dressing was removed (Figure 7a,b).
Figure 7a,b: On third day,
iodoform dressing was removed and sutured back.
Figure 8a,b: Uneventful 10th day healing.
Routine anti-microbial, anti-inflammatory drugs were prescribed, and patient was told to follow strict oral hygiene measures. On 10th day, all sutures were removed, the healing was uneventful (Figure 8a,b). The histopathological reports of the lesion & bone showed signs of inflammation while sinus lining showed rhinoscleroma, possibly because of Kelbsiella bacteria which is involved in pneumonia. The presence of any fungus or malignancy was completely ruled out (Figure 9a-c).
Figure 9a,b,c: Histopathological
report of retrieved bone ,sinus and granulation tissue.
We
reported a case of patient who has clinically recovered from COVID-19 but has
developed multiple pus discharging sinuses along mobility in teeth with
inability to chew. CT of face showed irregular osteolysis of maxillary alveolar
process & other parts of maxilla with maxillary sinus involvement, possibly
an avascular necrosis. This is highly unfortunate and unprecedented with no
reported cases in literature. Firstly, several studies reported the occurrence
of thromboembolic phenomenon in patient settings, especially in ICU patients
with severe or critical COVID-19 [9-14]. Secondly, we retrieved only ten
articles in a PubMed search for papers concerning thromboembolic events after COVID-19
cases [15,16].
Patients
with mild COVID-19 recover within two weeks, according to the last WHO report
[17]. But a few studies reported cases of mild COVID-19 which complicate with
venous thromboembolism events even in the active phase of the disease [15-22].
The
normal immunologic response of patients to infections can get altered in
uncontrolled diabetes mellitus. Such patients have altered polymorphonuclear
leukocyte response with decreased granulocyte phagocytic ability. Reports have
suggested that immunocompromised patient's serum has reduced to inhibit
Rhizopus invitro, which makes them suitable hosts to opportunistic infections
[23].
The
pathogenesis of COVID-19-related hypercoagulable state is evolving. In some
severe COVID-19 cases, an intense and uncontrolled inflammatory response seen
to contribute to thrombosis, especially in the microvasculature due to
thromboinflammation [24]. A subgroup of critical COVID-19 patients exhibits
clinical and laboratory features related to a hyperinflammatory syndrome
resembling a secondary haemophagocyticlymphohistiocytosis (SHL) such as
unremitting fever, hyperferritinemia, hypertriglyceridemia and ARDS [25]. In
these cases, increased levels of proinflammatory cytokines, such as interleukin
(IL)-1B, interferon-gamma (IFN-?), inducible protein 10 (IP10), monocyte
chemoattractant protein 1 (MCP1) and tumor necrosis factor-alpha (TNF-?), were
observed [7]. This inflammatory response causes damage to the vascular
endothelium, compromising its thrombo-protective state [24]. Several of
coagulation abnormalities seen in SARS-CoV-2-infected patients due to
activation of coagulation cascade caused by inflammation and endothelial
injury.
Clinical
and laboratory features compatible with a SHL were not exhibited by any of our
patients. Still damage to the endothelium caused by inflammation in the
pathogenesis of APE in these cases cannot be ruled out.
According
to the literature, studies showed more male predilection than women. One of the
explanations for the male sex risk factor for severe COVID-19 is the possible
sex-related differences on the immune response to SARS-CoV-2 infection [26].
The reason behind this theory is that women produce less inflammatory cytokines
after infection, which is linked with their shorter disease duration and higher
survival rates [27]. It has also been noted that male ratio is more prone for
thrombosis when hospitalized with COVID-19 [11].
Differential
diagnosis can be malignant salivary gland tumor arising from the accessory
glands of the palate, squamous cell carcinoma of maxillary sinus as chronic
ulcers with raised margins causing exposure of underlying bone, other features
can be antral carcinoma, which is local pain, swelling, epiphora, diplopia,
numbness, epistaxis, or nasal discharge. In our patient no symptoms suggestive
of any malignancy [8].
Extranodal
NK T-cell lymphoma (nasal type angiocentric lymphoma or midline lethal
granuloma) characteristically occurs in midline, affecting the oronasal region.
Patients may report nasal stuffiness, pain, and palatal swelling in the initial
stages. Patients develop progressive areas of ulceration that can lead to bone
necrosis and perforation latterly. Wegener’s granulomatosis is an uncommon
condition characterized by a necrotizing granulomatous condition of respiratory
tract, widespread vasculitis and necrotizing glomerulonephritis with common
presenting signs and symptoms include nasal stuffiness and epistaxis with or
without complain of fever, sinusitis, rhinorrhea, arthralgia, and weight loss
[8].
In
strawberry gingivitis, gingiva has a peculiar erythematous hyperplasia. It
causes oral-antral fistula by destructing underlying palatal and alveolar bone.
Due to extension of infections such as acute necrotizing ulcerative gingivitis
(ANUG) from the gingiva to bone, necrosis of bone occurs. But in the reported
case nothing as such was seen [8].
Thromboembolism is a
potential complication of mild COVID-19 cases and may occur late in the course
of the disease or even after its recovery, when the symptoms related to the
acute illness have already disappeared. These thromboembolic events can result
into avascular necrosis because of impaired vascular supply in certain parts of
body like maxilla in this case. Hence according to all the above events more
literature and study is required about the prevention of thromboembolic events
in selected group of patients with COVID-19.