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Contrast-enhanced magnetic resonance imaging (MRI), axial view. A strongly enhanced lesion was observed in the nasal septum, ethmoid sinuses, bilateral orbital apexes, and cavernous sinus
Source publication
Background
Orbital apex syndrome is a localized type of orbital cellulitis, where mass lesions occur at the apex of the cranial nerves. Although nasal septal abscess is uncommon, the organism most likely to cause nasal septal abscess is Staphylococcus aureus, and fungal septal abscesses are rare. Here we present an extremely rare and serious case o...
Context in source publication
Context 1
... Tool). The organism detected by PCR and tissue cultures was S. apiospermum. Subsequent enhanced magnetic resonance imaging (MRI) revealed strongly enhanced lesions of the nasal septum, ethmoid sinuses, bilateral orbital apexes, and cavernous sinus. The dura along the anterior and bilateral middle cranial fossa was thickened and well- enhanced. (Fig. 4). The diagnosis was orbital apex syn- drome and hypertrophic pachymeningitis secondary to fungal nasal septal abscess caused by S. apiospermum. Drug susceptibility testing (Table 1) led us to change the antifungal drip to voriconazole (500 mg/day). Since voriconazole had no apparent effect on the patient's symptoms after four days, we ...
Citations
... Scedosporium species are human pathogenic moulds. They are the agents of eumycetoma [1][2][3][4][5], a chronic deep fungal infection of the skin and subcutaneous tissues, and are responsible for a broad spectrum of localised and life-threatening disseminated infections in immunocompetent and immunocompromised individuals [6][7][8][9][10] affecting numerous organs of the body ( Figure 1A), including the bones and joints [11][12][13], the central nervous system [14][15][16][17], the eyes [18][19][20][21][22][23][24], the lungs [14,[25][26][27][28][29][30][31][32][33], the sinuses [34][35][36], and other body sites [37][38][39] of cystic fibrosis patients [8,28,[40][41][42], haematopoietic stem cell and solid organ transplant recipients [31,[43][44][45][46][47], hospitalised patients with COVID-19 [48], victims of near-drowning following natural disasters [14,15,30,49,50], and persons with traumatic injuries [51][52][53][54][55]. Recently assigned to the high-(eumycetoma-causative agents) and moderate-priority pathogen groupings by the World Health Organisation [56], Scedosporium diseases have an overall all-cause mortality rate of 42-46% [56]. ...
Scedosporium species are human pathogenic fungi, responsible for chronic, localised, and life-threatening disseminated infections in both immunocompetent and immunocompromised individuals. The diagnosis of Scedosporium infections currently relies on non-specific CT, lengthy and insensitive culture from invasive biopsy, and the time-consuming histopathology of tissue samples. At present, there are no rapid antigen tests that detect Scedosporium-specific biomarkers. Here, we report the development of a rapid (30 min) and sensitive (pmol/L sensitivity) lateral-flow device (LFD) test, incorporating a Scedosporium-specific IgG1 monoclonal antibody (mAb), HG12, which binds to extracellular polysaccharide (EPS) antigens between ~15 kDa and 250 kDa secreted during the hyphal growth of the pathogens. The test is compatible with human serum and allows for the detection of the Scedosporium species most frequently reported as agents of human disease (Scedosporium apiospermum, Scedosporium aurantiacum, and Scedosporium boydii), with limits of detection (LODs) of the EPS biomarkers in human serum of ~0.81 ng/mL (S. apiospermum), ~0.94 ng/mL (S. aurantiacum), and ~1.95 ng/mL (S. boydii). The Scedosporium-specific LFD (ScedLFD) test therefore provides a potential novel opportunity for the detection of infections caused by different Scedosporium species.
... Scedosporium species are human pathogenic moulds. They are the agents of eumycetoma [1][2][3][4][5], a chronic deep fungal infection of the skin and subcutaneous tissues, and are responsible for a broad spectrum of localised and life-threatening disseminated infections in immunocompetent and immunocompromised individuals [6][7][8][9][10] affecting numerous organs of the body ( Figure 1A), including the bones and joints [11][12][13], the central nervous system [14][15][16][17], the eyes [18][19][20][21][22][23][24], the lungs [14,[25][26][27][28][29][30][31][32][33], the sinuses [34][35][36], and other body sites [37][38][39] of cystic fibrosis patients [8,28,[40][41][42], hematopoietic stem-cell and solid organ transplant recipients [31,[43][44][45][46][47], hospitalised patients with COVID-19 [48], victims of near-drowning following natural disasters [14,15,30,49,50], and in persons with traumatic injuries [51][52][53][54][55]. Recently assigned to the high (eumycetoma-causative agents) and moderate priority pathogen groupings by the World Health Organisation [56], Scedosporium diseases have an overall all-cause mortality rate of 42-46% [56]. ...
Scedosporium species are human pathogenic fungi, responsible for chronic, localised, and life-threatening disseminated infections in both immunocompetent and immunocompromised individuals. Diagnosis of Scedosporium infections currently relies on non-specific CT, lengthy and insensitive culture from invasive biopsy, and time-consuming histopathology of tissue samples. At present, there are no rapid antigen tests that detect Scedosporium-specific biomarkers. Here we report the development of a rapid (30 min) and sensitive (pmol/L sensitivity) lateral-flow device (LFD) test, incorporating a Scedosporium-specific IgG1 monoclonal antibody (mAb), HG12, which binds to extracellular polysaccharide (EPS) antigens of between ~15 kDa and 250 kDa secreted during hyphal growth of the pathogens. The test is compatible with human serum, and allows the detection of the Scedosporium species most-frequently reported as agents of human disease (Scedosporium apiospermum, Scedosporium aurantiacum, and Scedosporium boydii), with limits of detection (LOD) of the EPS biomarkers in human serum of ~0.81 ng/mL (S. apiospermum), ~0.94 ng/mL (S. aurantiacum), and ~1.95 ng/mL (S. boydii). The Scedosporium-specific LFD (ScedLFD) test therefore provides a potential novel opportunity for the detection of infections caused by different Scedosporium species.
... Research in 2014 (19) showed that the pathogenic fungi of ROCM have increased to 37 species in 12 orders, but the list expanded to 47 species in 15 orders with the addition of Tilletiopsis minor (68), Saksenaea erythrospora (69), Pleurostomophora richardsiae (70), Acremonium, Phoma sp. (71), Apophysomyces ossiformis (72), and Scedosporium apiospermum (73), Aspergillus nominae (74), Mucor menace (75), and Lichtheimia ornate (76) ( Table 1). ...
Background
Extranodal natural killer/T-cell lymphoma, nasal type is a syndrome of middle face destruction with an association to Epstein-Barr virus. Fungi have been recovered from the diseased tissue now and then but were often seen as a lymphoma-associated secondary infection. However, there are ENKTL-NT cases with the recoveries of fungi and complete recovery with antifungal therapy, which are quite similar to rhino-orbital-cerebral mycosis (ROCM) that often confuses the physicians.
Methods
We searched Medline for English-language manuscripts limited to “human” and “case reports,” “letters,” “reviews,” and “clinical conferences” from 1966 to 2022. We used MeSH terms “lymphoma, extranodal nk-t-cell” [MeSH Terms] or “lethal midline granuloma” [MeSH Terms], in combination with MeSH terms “microbiology” [subheading] or “microbiology” [all fields] or “fungi” [all fields] or “fungi” [MeSH Terms] for ENKTL-NT with infections. We used MeSH terms “Mycoses” in combination with “Nose” [Mesh] OR “Orbital Diseases” [Mesh] for rhino-orbital-cerebral fungal infections.
Results
We appraised 149 included articles and extracted references related to ENKTL-NT and/or ROCM. Themes and subcategories were subsequently derived. Our findings revealed that ROCM and ENKTL-NT are characterized by progressive and destructive ulcers in the midline face or rhino-orbital structures. ROCM is mainly caused by fungi in the order of Mucorales, and ENKTL-NT is usually associated with Epstein-Barr virus and sometimes fungi. Radiologically, both are characterized by non-specific features of sinusitis, soft tissue infection, and necrosis. Pathologically, ROCM and ENKTL-NT share the same characteristics of inflammation, necrosis, and granuloma. ROCM is characterized by the detection of fungi in tissue, while ENKTL-NT is typically positive for NK/T-cell markers and cytotoxic granule-associated proteins, proliferation, and vascular damage of angioinvasion, which could be incited by Mucor irregularis and Rhizopus arrhizus in patients and mice.
Conclusion
ENKTL-NT and ROCM share many similarities in clinical presentations, radiology, and histopathology, and might have the same etiology. This may explain why the two diseases are tangled together in the reported cases, and suggests the role that the fungi may play in the development of these ENKTL-NT/ROCM diseases. The reason why ENKTL-NT and ROCM are sometimes confused is that the main pathogens of ROCM, Mucor irregularis and Rhizopus arrhizus , are the fungal causative agents of ENKTL-NT.
... There are several other reports of Scedosporium spp. involving the orbit in the literature spanning the last 40 years and most suffered from complications following the infection [6,8,9]. Loh et al. described the most similar situation to the current case involving progressive OAS in the setting of fungal pansinusitis of S. apiospermum following extraction of abscessed teeth. ...
Scedosporium apiospermum is a ubiquitous, highly resistant opportunistic fungus found in sewage and polluted waters and may infect the paranasal sinuses. Orbital Apex Syndrome may occur following trauma, surgery, or infection. An 80-year-old male with diabetes mellitus and mild dementia underwent uncomplicated, bilateral functional endoscopic sinus surgery for chronic sinusitis with polyposis. Initial pathology was reported as non-invasive bacterial and fungal species. On postoperative day 4, he had sudden right vision loss and abducens nerve palsy. Imaging noted violation of the lamina papyracea and inflammation of the optic nerve without compression. Medical therapy was begun and the patient developed sudden vision loss of the left eye. The patient then underwent emergent surgical decompression of both optic nerves. A final culture from the original surgery of S. apiospermum was made on postoperative day 10. Aggressive medical therapy was continued and the patient ultimately expired from complications of medical therapy and other underlying conditions. Trauma to the delicate bony walls of the orbit during sinus surgery in an immunocompromised patient who is unknowingly colonized with S. apiospermum can lead to the rapid spread of this highly neurotoxic organism.
... The first case was a 51-yearold woman with a history of asthma and recent treatment with oral steroids who presented a fungal septum abscess after endoscopic surgery performed for bilateral maxillary sinus fungus balls [3]. The second case was a 59-year-old man with uncontrolled diabetes who developed a fungal nasal septum abscess with a complication of orbital apex syndrome [4]. Two cases of fungal nasal septum abscess caused by atypical fungal stains such as Fusarium verticillioides and Aspergillus thermomutatus were reported in hematopoietic stem cell transplant recipients 5,6]. ...
The fungal nasal septum abscess is a rare localized invasive form of fungal rhinosinusitis. Rare cases have been described in the literature. In this article, we intend to describe a new case of fungal nasal septum abscess caused by Aspergillus flavus in diabetic patient after sinonasal surgery. A 53-year-old woman with a history of uncontrolled type 2 diabetes and asthma developed a nasal septum abscess after a sinonasal endoscopic surgery which was performed for nasal polyposis. Needle aspiration of the abscess was performed and the pus cultures were positive for Aspergillus flavus. The patient was treated with antifungal drugs and surgical drainage of the abscess. A clinical and biological improvement was observed. Her case has been followed up for 18 months, and there hasn't been any recurrence of the infection. The fungal nasal septum abscess should be suspected in patients who do not respond adequately to standard treatment of nasal septum abscess, especially patients with risk factors of fungal rhinosinusitis.
... It is not possible to find information of Scedosporium infections with neck soft tissue invasion similar to this patient. It is a cause, however of sinusitis and otomycosis [22][23][24][25][26] which makes it unlikely that this fungus is the causal agent in this case. ...
... In 2017, Ippei Kishimoto et al. reported only the second case of S. apiospermum-related OAS secondary to a fungal nasal septal abscess in a patient with uncontrolled diabetes. In their study, S. apiospermum was detected by the polymerase chain reaction and tissue cultures [10]. Despite treatment with antifungal drugs and surgical resection of the lesion, this patient suffered bilateral vision loss in common with the present case. ...
Orbital apex syndrome (OAS) is a localized orbital cellulitis at the orbital apex that can cause vision loss from optic neuropathy and ophthalmoplegia involving multiple cranial nerves. Herein, we report a rare and rapidly progressive case of OAS secondary to fungal pansinusitis caused by Scedosporium apiospermum in an immunocompromised patient following the extraction of abscessed teeth.
A 48-year-old man with diabetes mellitus who had failed to adhere to his treatment presented with complaints of a right-sided headache and toothache for two weeks, with nausea and vomiting for two days prior to presentation. The patient was treated for septic shock secondary to the dental abscesses. Non-contrast brain computed tomography (CT) showed no significant intracranial abnormalities other than pansinusitis. Four days later, dental extraction was performed. The patient reported progressive painless blurring of the vision in his right eye following the dental extractions and was referred to the ophthalmology department. Subsequent examinations revealed decreased optic nerve function and ophthalmoplegia in his right eye and dental caries in the upper molars, with a mucopurulent discharge from the right sphenoid region. The clinical diagnosis was OAS. Pus near the orbital apex was drained surgically. Methicillin-resistant Staphylococcus aureus was isolated from the pus and a nasal swab. Tissue culture from the septal wall yielded S. apiospermum. The patient’s condition deteriorated, despite intensive antibiotic and antifungal treatment and repeated surgical debridement. The disease progressed rapidly to his left eye. Sixty-seven days after the inital presentation, his visual acuity (VA) of both eyes was classified as no perception of light (NPL). The patient discharged himself from the hospital (at own risk discharge) and subsequently failed to attend a scheduled appointment in the ophthalmology clinic.
If immunocompromised patients present with OAS, fungal infections should be ruled out. Prompt and aggressive treatment using a multidisciplinary approach is mandatory in cases of potentially life-threatening and vision-threatening fungal infections.
Although Scedosporium species and Lomentospora prolificans are uncommon causes of invasive fungal diseases (IFDs), these infections are associated with high mortality and are costly to treat with a limited armamentarium of antifungal drugs. In light of recent advances, including in the area of new antifungals, the present review provides a timely and updated overview of these IFDs, with a focus on the taxonomy, clinical epidemiology, pathogenesis and host immune response, disease manifestations, diagnosis, antifungal susceptibility, and treatment. An expansion of hosts at risk for these difficult-to-treat infections has emerged over the last two decades given the increased use of, and broader population treated with, immunomodulatory and targeted molecular agents as well as wider adoption of antifungal prophylaxis. Clinical presentations differ not only between genera but also across the different Scedosporium species. L. prolificans is intrinsically resistant to most currently available antifungal agents, and the prognosis of immunocompromised patients with lomentosporiosis is poor. Development of, and improved access to, diagnostic modalities for early detection of these rare mold infections is paramount for timely targeted antifungal therapy and surgery if indicated. New antifungal agents (e.g., olorofim, fosmanogepix) with novel mechanisms of action and less cross-resistance to existing classes, availability of formulations for oral administration, and fewer drug-drug interactions are now in late-stage clinical trials, and soon, could extend options to treat scedosporiosis/lomentosporiosis. Much work remains to increase our understanding of these infections, especially in the pediatric setting. Knowledge gaps for future research are highlighted in the review.
Scedosporium apiospermum (S. apiospermum) is typically reported to be involved in superficial and subcutaneous fungal infections but overlooked in invasive infections, which is associated with a high mortality rate. It poses a diagnostic challenge due to its confusable characteristics to other hyaline hyphomycetes. Here, we reported a psoriasis patient with an invasive S. apiospermum infection. The patient presents an abscess at the intermuscular space of the left hip and an increased C-reactive protein level. Pus culture showed white-greyish, cottonlike colonies with aerial mycelium and terminal oval conidia, suggesting S. apiospermum. This rare fungus was rapidly confirmed by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry and RNA sequencing. The patient was successfully treated with voriconazole with no recurrence of the abscesses despite delayed treatment. This is the first such case infection report from China that described an unusual case of intermuscular space abscesses due to S. apiospermum. This report highlights the possibility of fungal infections in deeper tissue, as well as the necessity of thorough evaluation and microbiological diagnosis for invasive infections, particularly in immunocompromised patients.
Scedosporium sp. is a genus of filamentous fungi that is infrequently related to localized or disseminated infections in either immunocompromised patients, or immunocompetent patients with penetrating contaminated trauma. We report the case of a 31-year-old patient with a history of penetrating gunshot wound that required enucleation of his left eye, who 12 years after initial trauma presents necrosis of the anophthalmic socket with implant extrusion and lysis of inferior eyelid, finally identifying the causal agent to be Scedosporium sp. The patient received medical treatment with systemic voriconazole, and wide surgical debridement, with a satisfactory evolution. Fungal infections of anophthalmic sockets are infrequent, requiring a high diagnostic suspicion and aggressive treatment to avoid potentially devastating consequences.
