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Journal of Investigative Medicine High
Impact Case Reports
Volume 11: 1–5
© 2023 American Federation for
Medical Research
DOI: 10.1177/23247096231217823
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Case Report
Introduction
Severe acute respiratory syndrome coronavirus 2 (SARS-
COV-2) causes the coronavirus disease 2019 (COVID-19)
which led to a pandemic of more than 650 million cases and
6 million death as of January 2023 in the course of nearly 3
years. The most common signs of COVID-19 are respiratory
distress, gastrointestinal (GI) and hepatic diseases, and neu-
rological complications.1 This infection could develop in
fatal outcomes, especially in patients with underlying comor-
bidities, such as diabetes mellitus, hypertension, obesity, old
age, trauma, organ transplant, hematopoietic malignancies,
or cardiac, respiratory, or renal disorder.2,3
The physiopathology suggests that in infected COVID-19
patients, the absolute number of T lymphocytes, and CD4+
and CD8+ T cells are lower than healthy individuals while
various inflammatory markers, including interleukin (IL)-2,
IL-6, IL-10, tumor necrosis factor-alpha (TNF-α) are at
higher levels.4
This immune dysregulation is exacerbated in patients
with metabolic disorders, including hypertension and diabe-
tes and other chronic medical conditions, such as respiratory
system disease and cardiovascular disease as well as patients
taking drugs, such as steroids and Tociluzimab during
COVID treatment.5,6
Following COVID-19 infection, individuals are prone to
other infections, especially those infections for which cell-
mediated immunity constitutes an important host defense.
Recent studies suggest that some people may develop sinus
infections after COVID.7 The weakened inherent immunity
predisposes the patient to fungal and bacterial infections.
Fungal infections were observed in SARS patients in earlier
studies and were considered the leading cause of death in
25% to 73.7% of patients.8-10
Other studies have found a higher percentage of secondary
pulmonary infections (8%-15%) in COVID-19 patients, but
it is not clear whether it is bacterial or fungal infection.11,12
A rare kind of bacterial infection is caused by Actinomyces
israelii that could be associated with COVID infection.
The aim of the present study is to present a rare case of
actinomycosis and perform a review of literature on post-
COVID-19 occurrence of actinomycotic infection.
1217823HICXXX10.1177/23247096231217823Journal of Investigative Medicine High Impact Case ReportsMoaddabi et al
case-report20232023
1Department of Oral and Maxillofacial Surgery, Dental Research Center,
Mazandaran University of Medical Sciences, Sari, Iran
2Department of Neurosciences, Reproductive and Odontostomatological
Sciences, University of Naples “Federico II”, Naples, Italy
3Department of Oral and Maxillofacial Radiology, Dental Implants
Research Center, Dental Research Institute, School of Dentistry, Isfahan
University of Medical Sciences, Isfahan, Iran
*These authors contributed equally to the work.
Received August 30, 2023. Revised November 5, 2023. Accepted
November 12, 2023.
Corresponding Author:
Mariangela Cernera, University of Naples Federico II, Via Pansini 5,
80131 Naples, Italy.
Email: mariangela.cernera@icloud.com
Actinomycotic Sinomaxillary Infection
in a COVID-19 Patient: A Case Report
and Review of the Literature
Amirhossein Moaddabi, DDS, MSc1,*, Mariangela Cernera, DDS2,*,
Niccolò Giuseppe Armogida, DDS2, Parisa Soltani, DDS, MSc2,3,
and Gianrico Spagnuolo, DDS, PhD2
Abstract
Individuals with COVID-19 are prone to a variety of infections due to immune dysregulation. The present report presents
a case of actinomycotic infection in the maxillary bone and sinus region in a patient with a history of COVID-19. This case
report highlights the importance of considering bacterial infections including actinomycosis when encountering destructive
lesions resembling more prevalent fungal infections due to different therapeutic medication protocols. In addition, a literature
review of the existing reports of similar post-COVID-19 actinomycotic infection is presented.
Keywords
actinomycosis, actinomycete, COVID-19
2 Journal of Investigative Medicine High Impact Case Reports
Case Presentation
A 62-year-old Iranian female attended the office of an oral
and maxillofacial surgeon with a chief complaint of pain in
the maxillary and zygomatic region. The patient had a his-
tory of controlled hypertension and cardiac problems and
was taking the following medications: aspirin 80 mg, clopi-
dogrel 75 mg, bisoprolol 2.5 mg, indapamide 1.5 mg, and
losartan 12.5 mg. She tested positive for COVID-19 in
August 2021 using real-time polymerase chain reaction
(RT-PCR) technique. After 10 days, she felt an intense, dull,
pulsating pain in the maxillary and zygomatic region. She
claimed that the pain was alleviated only with diclofenac.
After 1 month, the patient was referred to an otolaryngolo-
gist who performed endoscopic sinus surgery. However, the
pain did not resolve and was exacerbated. The patient was
then referred to a dentist after 20 days who extracted the right
maxillary second premolar. However, the pain was not alle-
viated and the extracted socket was not healed. Finally, 2
months after the diagnosis of COVID-19, the patient was
referred to the oral and maxillofacial surgeon.
Extraoral clinical examination was normal with a normal
range of mouth opening and no extraoral swelling. In the
intraoral examination, necrotic bone was observed in the
posterior right maxillary alveolar bone. The roots of the right
maxillary first molar were exposed and inflammation was
noted in the surrounding gingiva (Figure 1). The right maxil-
lary first molar had severe mobility.
Cone-beam computed tomography (CBCT) was obtained
from the maxillary region and paranasal sinuses. In the
CBCT views, a lytic and destructive lesion was observed on
the right side of the maxilla. Destruction of the posterior
alveolar process, medial wall, anterior wall, lateral wall, and
floor of the right maxillary sinus was seen. The right maxil-
lary sinus was completely opacified with soft tissue density
and fragments of bone were also seen in the sinus. Evidence
of endoscopic sinus surgery was observed in the nasal and
paranasal regions. Loss of bone support of the involved teeth
was detected without root resorption (Figure 2).
Under general anesthesia, the right maxillary first molar
was extracted and a biopsy was obtained from the necrotic
bone and granulation tissue in the maxillary sinus and sinus
mucosa. The samples were stored in 2 different containers:
one with normal saline for culturing and another one with
formalin for histopathological examination. Bacterial smear
and culture revealed a tangled mass of elongated, rod-shaped
cells that branched into filaments about 1 to 2 µm in diame-
ter. Gram staining revealed gram-positive rods and thiogly-
colate broth growth showed non-acid fast bacteria indicating
Actinomyces. Histopathological evaluation using hematoxy-
lin and eosin staining also demonstrated areas of bone necro-
sis along with inflammatory infiltration and bacterial rods
(Figure 3).
Figure 1. Intraoral photograph depicting exposed roots of the
right maxillary first molar with surrounding inflammation.
Figure 2. Cone-beam computed tomographic images in (A)
coronal and (B) axial sections depicting the lytic and destructive
lesion on the right side of the maxilla.
Moaddabi et al 3
The patient was prepared for surgery by a team contain-
ing an oral and maxillofacial surgeon, an oral and maxillo-
facial radiologist, and an otolaryngologist. Half an hour
before the surgery, 1 g of cefazolin was administered via
intravenous injection. First, endoscopic sinus surgery was
performed to clear the nasal cavity and sinus ostium.
Afterward, curettage and removal of necrotic bone was per-
formed using intraoral open sinus surgery with buccal
advancement flap and buccal fat grafting to close the oroan-
tral connection. After the operation, cephalexin 500 was
administered every 8 hours for 1 week. Routine non-steroi-
dal anti-inflammatory drugs (NSAIDs) were administered
in case of pain. Chlorhexidine 0.2% mouthwash was also
administered 3 times a day for 1 week. The patient then
received high-dosage penicillin G for a period of 6 months
administered by the infectious diseases specialist.
The patient was followed 1 week, 1 month, 3 months, 6
months, and 8 months after the surgery. The clinical signs
and symptoms started to improve after the surgery. The sur-
gical site and the extraction socket have been completely
healed. No evidence of pus discharge and secretions and
oroantral fistula was observed in clinical examination
(Figure 4).
Discussion
Actinomycosis is a rare anaerobic bacterial infection caused
by gram-positive, non-motile, non-acid fast filamentous bac-
terial rods. Actinomyces are part of the normal flora com-
monly seen in the human oral cavity, female urogenital tract,
and GI tract. The organism has low virulence and only
invades the body to cause deep-seated infections when there
is tissue injury or following a break in the normal mucosal
barrier. Clinically, actinomycotic infection has 3 subtypes;
cervicofacial, thoracic, and abdominal. Cervicofacial type is
the most common type of actinomycotic infection. Infection
is mostly polymicrobial, established with the help of a com-
panion bacteria by inhibiting host defense, reducing oxygen
tension, or by toxin production that facilitates the inoculation
of actinomycoses.13
The infection is characterized by contagious spread,
suppurative and granulomatous inflammation leading to
multiple abscess formation, and sinus tracts that may dis-
charge yellow-colored sulfur granules. In the jaw bones, it
can result in osteomyelitis if untreated. Imaging findings are
nonspecific and are non-contributory in diagnosing fungal
or bacterial osteomyelitis, but will help in assessing the
degree of soft tissue and bone involvement. Only the biopsy
specimen of the involved tissue could confirm the diagnosis
in which the filamentous shape of the microbes that resem-
ble fungal hyphae could be observed.
There are only a few reported cases of actinomycosis sec-
ondary to COVID-19 infection in the literature (Table 1). All
of them report male patients, with a mean age of 49-year-old,
with a history of COVID-19 for 1 to 4 months before the start
of the bacterial infection and with other comorbidities, such
as type II diabetes and other predisposing conditions, includ-
ing dental extractions. Hyperglycemia in diabetic patients is
associated with impairment of immune response and inter-
ference with the wound healing process. Therefore, resis-
tance to bacterial infection is lower in diabetic individuals
compared with those without diabetes.19,20 Similarly, admin-
istration of corticosteroids can lead to immune suppression,
in turn increasing the susceptibility of the individuals to
infective diseases.21 The reported cases mostly revealed
co-infection of actinomycosis and mucormycosis in the
individuals. A systematic review in 2021 revealed 101 cases
of post-COVID-19 mucormycosis, of which 82 were from
India. Diabetes, extensive use of corticosteroids, and a
background of COVID-19 appears to increase incidence
of mucormycosis.19 Therefore, mucormycosis is one of the
most important differential diagnoses to consider in such
Figure 3. Histopathological hematoxylin and eosin staining of
the sample indicating focus of actinomycosis with surrounding
inflammatory response.
Figure 4. Intraoral photograph depicting resolution of
inflammation and favorable healing of the infection.
4 Journal of Investigative Medicine High Impact Case Reports
cases. Another bacterial infection that needs to be considered
is nocardiosis. Nocardia are gram-positive aerobic filamen-
tous bacteria with similar cytomorphologic features with
actinomycete. However, they differ most prominently in
their ability or lack thereof to retain acid-fast staining.22
Infections with the two microorganisms also may show simi-
lar clinical manifestations. In addition, nocardiosis has been
reported in COVID-19 patients.23 However, since the bacte-
rial tests in the present case revealed non-acid-fast features, a
diagnosis of actinomycosis was made.
The current report presented a case of actinomycosis in
a 62-year-old female patient with a history of controlled
hypertension and cardiac problems and showing clinical
symptoms 10 days after testing positive for COVID-19. The
radiographic appearance of bone destruction and necrosis in
the sinonasal region of COVID-19 patients rises the more
common possibility of fungal infections including mucormy-
cosis. Improper diagnostic tests and culture can lead to mis-
diagnosis and thus hinder adequate and timely treatment
with antibiotic therapy. Therefore, it is important to consider
the possibility of imitating bacterial infections, such as acti-
nomycosis in these cases.
Typical management of cervicofacial actinomycosis con-
sists of conservative debridement and intravenous antibiotic
therapy.24 The same regimen used in our case report allowed
for the resolution of infection.
Conclusion
This case reports highlights the importance of histopatho-
logical diagnosis in routine clinical practice. The prevalence
of actinomycosis in COVID-19 patients is considerably less
than mucormycosis. Definitive treatment cannot be provided
in cases of misdiagnosis, which can lead to substantial mor-
bidity and mortality. Histopathological examination of these
lesions is crucial for a definitive diagnosis and prognosis of
the lesion.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect
to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, author-
ship, and/or publication of this article.
Ethics Approval
Our institution does not require ethical approval for reporting indi-
vidual cases or case series.
Informed Consent
Written informed consent was obtained from the patient for their
anonymized information to be published in this article.
ORCID iD
Mariangela Cernera https://orcid.org/0000-0001-6356-6536
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Article
Full-text available
During the SARS-CoV-2 pandemic, few cases of Nocar-dia spp. co-infection have been reported during or after a COVID-19 infection. Nocardia spp. are gram-positive aerobic actinomycetes that stain partially acid-fast, can infect immunocompromised patients, and may cause dis-seminated disease. We report the case of a 52-year-old immunocompromised man who had Nocardia pseudobrasiliensis pneumonia develop after a SARS-CoV-2 in-fection. We also summarize the literature for no-cardiosis and SARS-CoV-2 co-infections. Nocardia spp. infection should remain a part of the differential diagnosis for pneumonia in immunocompromised hosts, regardless of other co-infections. Sulfonamide/carbapenem combina-tions are used as empiric therapy for nocardiosis; species identification and susceptibility testing are required to se-lect the optimal treatment for each patient.
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Introduction and Importance: Maxillary actinomycosis is a persistent, very rare disease produced by Actinomyces species which may include only soft tissue or bone or the two together. Actinomycotic osteomyelitis of maxilla is very infrequent when compared to mandible. Case presentation Here we are conferring a case of an elderly male patient who had history of COVID-19 infection 4 months ago, with constant complaint of non-remitting vague pain in the region of maxilla with tooth loosening and extractions. He was given a provisional diagnosis of chronic osteomyelitis of maxilla which was later on proved by histopathology as actinomycotic osteomyelitis. Clinical discussion A saprophytic fungus causes mucor mycosis, and it is quite unusual. Strawberry gingivitis is one of the signs and symptoms. Mucormycosis and post-covid oral maxillofacial problems can be improved with early diagnosis. Oral Mucormycosis should be suspected in individuals with weakened immune systems, uncontrolled diabetes or post-covid instances. Surgery and adequate antibiotic treatment are necessary to treat actinomycosis. Infection may return after a period of inactivity, so long-term follow-up is necessary. Conclusion We conclude a positive causal association between COVID-19 and actinomycosis. Maxillary osteomyelitis, a very rare infection, and in our case, the causative organism was Actinomyces Patients who have been infected should be tested for Actinomycin, which may masquerade as a head and neck illness.
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p>Actinomyces are commensals of human oropharynx and actinomycosis is considered mainly as an endogenous infection that is triggered by a mucosal lesion. Typically, the disease presents as a slowly progressive painless indurated mass evolving into multiple abscesses with draining sinus tracts sometimes expressing a typical yellow exudate with characteristic sulfur granules. The gold standard of diagnosis is histological examination and bacterial culture of the tissue. Most isolates are susceptible to beta lactams and they are the treatment of choice along with surgical management with drainage of abscesses and excision of recalcitrant fibrotic lesions and debridement of necrotic bone tissue. Here we present a case of 37-year-old male patient who has developed severe COVID-19 infection following which he developed invasive mucormycosis followed by actinomycosis. We postulate that the lymphopenia and the use of immunosuppressants used in treatment of COVID-19 lead to mucormycosis and aggressive debridement used as a strategy in treatment of mucormycosis led to colonization of actinomyces leading to cervicofacial actinomycosis.</p
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Purpose Covid-19 is a global threat that pushes health care to its limits. Since there is neither a vaccine nor a drug for Covid-19, people with an increased risk for severe and fatal courses of disease particularly need protection. Furthermore, factors increasing these risks are of interest in the search of potential treatments. A systematic literature review on the risk factors of severe and fatal Covid-19 courses is presented. Methods The review is carried out on PubMed and a publicly available preprint dataset. For analysis, risk factors are categorized and information regarding the study such as study size and location are extracted. The results are compared to risk factors listed by four public authorities from different countries. Results The 28 records included, eleven of which are preprints, indicate that conditions and comorbidities connected to a poor state of health such as high age, obesity, diabetes and hypertension are risk factors for severe and fatal disease courses. Furthermore, severe and fatal courses are associated with organ damages mainly affecting the heart, liver and kidneys. Coagulation dysfunctions could play a critical role in the organ damaging. Time to hospital admission, tuberculosis, inflammation disorders and coagulation dysfunctions are identified as risk factors found in the review but not mentioned by the public authorities. Conclusion Factors associated with increased risk of severe or fatal disease courses were identified, which include conditions connected with a poor state of health as well as organ damages and coagulation dysfunctions. The results may facilitate upcoming Covid-19 research.
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Objective: Acute Invasive Fungal Rhinosinusitis is a rare condition which recently has gained attention due to its increased occurrence in the post COVID-19 patients past the second wave. The current study retrospectively evaluates the occurrence of Acute Invasive Fungal Rhinosinusitis (Mucormycosis) in post COVID-19 (Corona Virus Disease-19) patients. Methods: A descriptive study included patients diagnosed with Acute Invasive Fungal Rhinosinusitis (Mucormycosis) after recent COVID-19 infection. 110 patients were evaluated retrospectively with histopathological confirmation of Mucormycosis. Surgical treatment was restricted to patients who tested Real Time Polymerase Chain Reaction (RT PCR) negative for COVID-19 except for three patients who were tested positive. Antifungal agents were given to patients following surgery. Results: A total of 110 patients with a mean age of 48.42 years were included. The most common risk factor was diabetes mellitus (88.2%). Sino-nasal, orbital, palatal and intracranial involvement were 57.9%, 48.5%, 12.7% and 5.6% respectively. Histopathological confirmation revealed mucormycosis. The most common reported symptoms were periorbital oedema (20.5%), headache (20.3%), gingival swelling (18.5%) facial pain (18.4%) and facial swelling (18.2%). All the patients were treated with surgical debridement and antifungal medications. The overall survival rate was 95.32%. Conclusion: Acute Invasive Fungal Rhinosinusitis is a life-threatening opportunistic infection. Patients with moderate to severe COVID-19 infection are more susceptible to it. Uncontrolled diabetes mellitus and intake of corticosteroids increase the risk of developing Acute Invasive Fungal Rhinosinusitis. Early diagnosis and timely management can improve survival rates of the patients.
Article
Coronavirus disease 2019 (COVID-19) is an infection caused by severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2). COVID-19 infection may be associated with a wide range of bacterial and fungal co-infections. Herewith a case of 46 year-old male patient of post COVID-19 developed co-infection. He had received steroid treatment and improved in last month. He is known case of diabetes type II since last one year and was on treatment. Now presented to our hospital having fever, facial pain, and swelling mid-face region. His RT-PCR test was positive. The CT scan of the nasal septum, medial walls of bilateral maxillary, ethmoid, sphenoid and frontal sinuses exteding into bilateral nasal cavities. Features suggestive of infective pathology invasive fungal rhinosinusitis On clinical, radio imaging and on histopathological findings diagnosed as maxillary mucormycosis with actinomycosis. Conclusion: We are presenting this rare case of COVID-19 associated with co-infection of mucormycosis and actinomycosis for its clinical, radio imaging, and on histopathological findings. Key words: Coronavirus Disease 2019 (COVID-19), Mucormycosis, Actinomycosis, Co-infections.
Article
Objectives Occurrence of invasive fungal respiratory superinfections in patients with COVID-19 has gained increasing attention in the latest studies. Yet, description of acute invasive fungal sinusitis with its management in those patients is still scarce. This study aims to describe this recently increasing clinical entity in relation to COVID-19 patients. Methods Prospective longitudinal study included patients diagnosed with acute invasive fungal rhinosinusitis after a recent COVID-19 infection. Antifungal agents given included amphotericin B, voriconazole and/or posaconazole. Surgical treatment was restricted to patients with PCR negative results for COVID-19. Endoscopic, open and combined approaches were utilized to eradicate infection. Follow-up for survived patients was maintained regularly for the first postoperative month. Results A total of 36 patients with a mean age of 52.92 ± 11.30 years old were included. Most common associated disease was diabetes mellitus (27.8%). Mycological analysis revealed infection with Mucor and Aspergillus species in 77.8% and 30.6% of patients, respectively. Sino-nasal, orbital, cerebral and palatine involvement was found in 100%, 80.6%, 27.8% and 33.3% of patients, respectively. The most common reported symptoms and signs are facial pain (75%), facial numbness (66.7%), ophthalmoplegia and visual loss (63.9%). All patients were treated simultaneously by surgical debridement with antifungal medications except for two patients with PCR positive swab for COVID-19. These two patients received antifungal therapy alone. Overall survival rate was 63.89% (23/36). Conclusion Clinical suspicion of acute invasive fungal sinusitis among COVID-19 patients and early management with antifungal therapy and surgical debridement is essential for better outcomes and higher survival. Level of evidence IV.