Primary odontogenic onset invasive mucormycosis-an under recognized clinical entity.

OBJECTIVES: The primary source of facial mucormycosis is through inhalation of fungal sporangiospores, resulting in invasive disease in paranasal sinuses. However, dental onset mucormycosis has not been well documented in literature. The aim of this study was to describe the clinical characteristics and outcomes of patients with odontogenic onset mucormycosis. METHODS: From a large cohort of mucormycosis involving the face between July 2020 and October 2021, we selected patients who had dental symptoms at onset and predominant alveolar involvement with little to no paranasal sinus disease as shown by baseline imaging. All patients had a confirmed diagnosis of mucormycosis through histopathology, with or without the growth of Mucorales in fungal culture. RESULTS: Out of 256 patients with invasive mucormycosis of the face, 8.2% (21 patients) had odontogenic onset. Uncontrolled diabetes was a common risk factor, affecting 71.4% (15/21) of the patients, while recent COVID-19 illness was noted in 80.9% (17/21) of patients. The median duration of symptoms at presentation was 37 days (IQR, 14-80 days). The most common symptoms were dental pain with loose teeth (100%), facial swelling (66.7% [14/21]), pus discharge (28.6% [6/21]), and gingival and palatal abscess (28.6% [6/21]). Extensive osteomyelitis was found in 61.9% (13/21) of the patients, and 28.6% (6/21) had oroantral fistulas. The mortality rate was low, at 9.5% (2/21), with only 9.5% (2/21) of the patients having brain extension and 14.2% (3/21) in the orbit. CONCLUSION: This study suggests that odontogenic onset invasive mucormycosis may be a separate clinical entity with its own distinct clinical features and prognosis.

Differences in clinical and imaging presentation of maxillary sinus fungus ball with and without intralesional hyperdensity.

Maxillary sinus fungal balls (MSFBs) mostly occur in older individuals and demonstrate female predominance. Early diagnosis is important to avoid treatment delays. Intralesional hyperdensity (IH) indicates the presence of heavy metal deposition within fungal hyphae and has been the most specific characteristic of MSFB on computed tomography (CT). For those without IH on CT, the diagnosis of MSFB remains challenging. This study aimed to characterize clinical presentation of MSFB with and without IH and to study factors contributing to MSFB with no IH formation. We retrospectively identified 588 patients with MSFB. The clinical characteristics and CT findings were reviewed. Patients with unilateral MSFB had a mean age of 57.4 years and demonstrated female predominance (64.63%). The female-to-male ratio was highest at 51-60 years (2.02) and rose to 2.60 in MSFB with IH only. Compared to those with IH, MSFB without IH was significantly more common in males (OR = 2.49), in those with diabetes mellitus (DM) (OR = 1.87), adjacent maxillary odontogenic pathology (OR = 1.75). Complete opacification on CT was less common in MSFB without IH (OR = 0.60). Patients with MSFB without IH were more likely to have DM, no female predominance, adjacent maxillary odontogenic pathology, and partial opacification of the sinus, compared to those with IH. These may be helpful in better understanding of the formation of MSFBs without IH, early identification of them and prevention of post-operative recurrence.

A rare entity to major outbreak: a case report on mucormycosis.

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.

Imaging findings using a combined MRI/CT protocol to identify the "entire iceberg" in post-COVID-19 mucormycosis presenting clinically as only "the tip".

AIM: To analyse combined computed tomography (CT) and magnetic resonance imaging (MRI) characteristics of invasive rhino-orbital mucormycosis (IROM) in post-COVID-19 infection patients for accurate diagnosis and delineation of the extent of involvement. MATERIALS AND METHODS: A retrospective analysis was undertaken of 50 patients who developed IROM post-COVID-19 infection who underwent combined CT/MRI evaluation. RESULTS: The age range of the 50 affected patients was 23-73 years. Out of these, 41 were diabetic. CT/MRI showed predominant involvement of the maxillary (n=26) and ethmoid (n=19) sinuses. Extension of disease to the orbit (n=35), cavernous sinus (n=18), hard palate (n=15), skull base (n=8), and intracranial involvement (n=3) was seen. Perineural spread of the disease was analysed along all divisions of the trigeminal nerve and its branches. MRI showed T2-hypointense soft-tissue thickening with heterogeneous contrast enhancement with corresponding hyperdensities on CT diagnosing the presence of fungal elements. CONCLUSION: Clinicians should be aware of the possibility of IROM post-COVID-19 infection. Conjunctive use of CT, which depicts bone destruction and other reactive bony changes along with MRI, which reveals characteristic findings of soft-tissue thickening of the involved sinuses with extension of disease to the orbits, cavernous sinus, dura, hard palate, skull base, and intracranial structures. Accurate diagnosis and early recognition of the disease and its extension with appropriate use of these techniques helps to initiate appropriate and timely treatment, which is vital to prevent a fatal outcome.

Coronavirus disease (COVID-19) complicated by rhino-orbital-cerebral mucormycosis presenting with neurovascular thrombosis: a case report and review of literature.

Among the ever-increasing literature of the coronavirus disease 2019 (COVID-19), there have been reports on several complications in association with severe acute respiratory syndrome-coronavirus-2 (SARS-CoV-2), such as secondary bacterial and fungal infections. We report a 61-year-old woman with a past history of diabetes mellitus who presented to our hospital suffering from COVID-19 infection. During the course of her hospitalization, the patient developed chemosis and proptosis in both eyes, ultimately leading to a diagnosis of invasive rhino-orbital-cerebral mucormycosis and cerebrovascular thrombosis. This study strengthens the possible association between the occurrence of COVID-19 and invasive mucormycosis infection, providing new impetus for further investigations to substantiate this correlation.

CT and MRI Findings of Invasive Mucormycosis in the Setting of COVID-19: Experience From a Single Center in India.

An increasing incidence of rhinoorbitocerebral mucormycosis (ROCM) among patients with COVID-19 has recently been reported in India. We report the imaging findings for 25 patients with COVID-19 and invasive ROCM at a single hospital in India. Findings included sinus wall erosions (n = 20), air within bony sinus structures (n = 11), and focal mucosal nonenhancement (n = 8). Orbital, vascular, and intracranial complications were also observed. Radiologists should recognize the increasing incidence of ROCM among patients with COVID-19 to facilitate early diagnosis.

Combined Endonasal-Transorbital Approach to Manage the Far Lateral Frontal Sinus: Surgical Technique.

In recent decades, the ever-expanding use of endoscopes and development of dedicated instrumentation have reshaped the panorama of surgical approaches to the frontal sinus.1 Nonetheless, the far lateral portion of the sinus might still represent a concern, especially in cases with unfavorable or distorted anatomy.2,3 We report the case of a 52-year-old man, referred to our department for recurrent episodes of left orbital swelling and supraorbital headache, 3 months after Draf III frontal sinusotomy for marsupialization of multiple frontal mucoceles. Computed tomography and magnetic resonance imaging scans were consistent with persistent inflammatory tissue in the far lateral left frontal sinus. Revision surgery was performed, adopting a combined endonasal orbital transposition3 and superior eyelid transorbital approach.4,5 The postoperative course was uneventful, and the microbiologic and histologic examinations demonstrated noninvasive Aspergillus fumigatus infection. The radiologic control showed patency of the frontal recess and complete clearance of the sinus. The patient is asymptomatic after 16 months (Video 1). The transorbital approach is effective in managing orbital and frontal sinus diseases,6 and the combination with the endonasal route grants complete access to the frontal sinus, even in cases of high pneumatization and lateral extension.4,7 Multiportal transorbital approaches represent additional techniques in the rhinologist's surgical armamentarium, which can overcome the limits of a single port approach.8,9 Reports on their use providing technical hints and critical considerations are to be encouraged to ease and stimulate the surgical training in this field.

Treatment of Paranasal Sinus Fungus Ball: A Systematic Review and Meta-Analysis.

OBJECTIVES: Paranasal sinus fungus ball is a common non-invasive mycosis with excellent long-term surgical treatment results. The present systematic review and meta-analysis were undertaken to define current treatment concepts and success rates in paranasal sinus fungus ball treatment. METHODS: Systematic searches were performed in multiple databases with criteria designed to include all studies published until May 2020 focusing on paranasal sinuses fungus ball treatment in humans. We selected studies including at least 10 patients, specifying treatment modalities, providing a minimum 6-month follow-up, and objectivating treatment success. After duplicate removal, abstract and full-text selection, and quality assessment, we reviewed eligible articles for treatment modalities and success rates. Success rates were pooled in a random effect meta-analysis and compared according to the use of intraoperative sinus lavages and postoperative antibiotics. RESULTS: Among 740 unique citations, 14 studies were deemed eligible. Most (n = 11) were retrospective case series. All studies relied on endoscopic sinus surgery. Intraoperative lavages were proposed in 10 studies and postoperative antibiotics in 7 (for all patients in 5 studies and for selected patients in 2). No significant heterogeneity was observed between results (Cochran's Q P = .639, I2 test = 0). Treatment success rate was 98.4% (95% confidence interval 97.4%-99.3%). Intraoperative sinus toilette and postoperative antibiotics didn't significantly improve the success rate. CONCLUSION: Endoscopic sinus surgery shows excellent results in fungus ball treatment. Further prospective studies might help further reducing antibiotics prescriptions in these patients and improve their management.

[IManagement of patients with relapsed primary naso-sinusal tuberculosis]. / Gestion de la rechute d'une tuberculose naso-sinusienne primaire.

Primary naso-sinusal tuberculosis (TB) is a relatively rare or exceptional disorder characterized by polymorphic or non-specific clinical manifestation. Diagnosis is based on anatomo-pathological examination and mycobacteriology test of biopsy specimen. Predictor of good outcome is early conventional anti-tuberculous antibiotic therapy. However, our study reports recurrence at this rare site in an immunocompetent patient despite early suitable TB treatment and good adherence with therapy. Relapse was correlated with underdosing of rifampicin. This study highlights the diagnostic, etiological and therapeutic management of this relapse. Our experience could help clinicians to better manage this uncommon condition.

Fungus ball of the maxillary sinus: Retrospective study of 48 patients and review of the literature.

BACKGROUND: Maxillary fungus ball (FB) is the most frequent paranasal localization. OBJECTIVE: To review clinical presentation, surgery and results of treatment in our series of patients with maxillary FB. To review the literature concerning treatment of maxillary FB. PATIENTS AND METHODS: 48 patients with a diagnosis of maxillary FB were treated with endoscopic sinus surgery (ESS) alone or in association with external approaches. Before surgery all patients received computed tomography (CT), nasal endoscopy and dental examination. All the patients were followed for 1 year after surgery. Studies concerning surgical treatment of maxillary FB from 2006 were reviewed. RESULTS: The mean age of patients was 53.6 ±â€¯11.9 years. 20 patients (41.6%) did not present any symptom, 19 patients (39.7%) had nasal symptoms, 3 patients (6.2%) had facial pain, 6 patients (12.5%) had a combination of both. Endoscopic examination was positive in 31 patients (64.6%), 17 patients (35.4%) showed negative findings. Logit regression model demonstrated that clinical symptoms contribute to the prediction of a positive endoscopic examination. 25 patients (52.1%) presented odontogenic factors. Complete clinical and radiological resolution of FB was observed in 46 patients (95.8%). CONCLUSIONS: Comparing our sample to the studies reviewed we may concluded that odontogenic factors were frequently reported and should be treated at the same time of maxillary FB. ESS alone or in association with external approaches is an effective treatment for patients with maxillary FB.

Pediatric Mucormycosis: A 10-Year Systematic Review of Reported Cases and Review of the Literature.

Mucormycosis is a severe infection that affects a variety of patients, including immunocompromised children and neonates. Given improved survival rates from advances in the treatment of malignancies, the population at risk for mucormycosis is increasing. We conducted a systematic review of cases of mucormycosis in children in the English-language literature reported between August 2008 and June 2017 and analyzed the clinical characteristics, diagnosis, management, and outcome of those infections. The most common underlying diagnoses included neutropenia (41%), hematologic malignancy (39%), prematurity (13%), and hematopoietic stem cell transplant (11%). Sinus disease (28%) and disseminated disease (24%) were the most common presentations. Rhizopus spp were the most common organisms isolated (22%). Amphotericin B remains the backbone of treatment and was prescribed in 86% of these cases. The resulting mortality rate remains high (32%). We provide here the results of a literature review of mucormycosis in children, including its epidemiology and clinical manifestations, and describe current advances in its diagnosis and treatment.

Conversion to Chronic Invasive Fungal Sinusitis From Allergic Fungal Sinusitis in Immunocompetence.

A review of the treatment of allergic and invasive fungal sinusitis, as well as a presentation of the first recorded case of a conversion from allergic fungal sinusitis (AFS) to chronic granulomatous invasive sinusitis and the fourth case of invasive fungal sinusitis associated with Curvularia. This immunocompetent patient suffering from chronic AFS converted after repeated high-dose steroid tapers and noncompliance. AFS may present atypically and should be suspected even in immunocompetent patients with sinus disease who report new onset pain and neurologic symptoms. Clinicians should consider the potential complications associated with repeated systemic steroid administration. Laryngoscope, 129:2447-2450, 2019.

Outcomes and factors affecting them in patients with rhino-orbito-cerebral mucormycosis.

AIM: To report the frequency and factors affecting patients', globe and vision survivals in rhino-orbito-cerebral mucormycosis (ROCM). METHODS: This is a retrospective study of 63 patients (79 eyes) with biopsy-proven ROCM at a university hospital 2008-2016. Systemic and ophthalmic manifestations, imaging, management and final outcomes were recorded. Globe survival was defined as no exenteration and vision survival as final visual acuity of light perception and more. RESULTS: Mean age was 55.5 (SD 12.9) years with no gender preference. Diabetes was the most common underlying disease (68.3%). Patient survival was observed in 57.1 % (36/63). Presence of frozen eye (OR 4.6), nasal mucosal involvement (OR 7.3) and shorter duration of antifungal therapy (OR 1.03) were significantly associated with lower patient survival. Exenteration did not significantly change the survival. Globe survival was detected in 43% (34/79). Higher white blood cell (WBC) count was associated with a lower globe survival (p=0.02). Vision survival was observed in 25.3% (20/79) in whom younger age was significantly associated with a worse vision survival. CONCLUSION: Patient, globe and vision survivals were 57%, 43% and 25%, respectively. Exenteration did not affect the patients' survival. While frozen eye and nasal mucosal involvement were significantly associated with a lower survival, higher WBC count significantly increased the risk of exenteration.

Update on sinus disease in children with cystic fibrosis: advances in treatment modalities, microbiology, and health-related quality-of-life instruments.

PURPOSE OF REVIEW: There is a lack of consensus with regards to the diagnosis and treatment of sinus disease in children with cystic fibrosis. Here, we review literature from the past 18 months in order to highlight the way forward in this contentious field. RECENT FINDINGS: Most of the literature (from the past 18 months) on sinus disease in pediatric cystic fibrosis focused on treatment approaches, bacteriology and immunology, and health-related quality-of-life (HRQOL) instruments. Quality studies have demonstrated that functional endoscopic sinus surgery (FESS) is as safe in children with or without cystic fibrosis; that the microbiology of the paranasal sinus in children with cystic fibrosis is different than that of their lungs; and, that HRQOL instruments may prove useful in determining sinonasal disease severity in children with cystic fibrosis. SUMMARY: Medical and surgical approaches appear to be viable in the treatment of sinonasal disease in pediatric cystic fibrosis; the microbiology and immunology of pediatric cystic fibrosis is proving more complex and nuanced than initially believed; and, HRQOL instruments show promise in reconciling differences between observable and clinically relevant sinus disease in pediatric cystic fibrosis patients.