Rhinocerebral mucormycosis and Trichosporon asahii fungemia in a pediatric patient with acute lymphoblastic leukemia: a rare coinfection

ABSTRACT Mucormycosis is a rare life-threatening opportunistic infection, with rhinocerebral mucormycosis (ROCM) being the most common presentation. Trichosporon asahii is an emerging pathogen that often causes fatal infections in patients with underlying hematologic malignancies due to its high drug resistance. We report a rare case of concomitant rhinocerebral mucormycosis and T. asahii fungemia secondary to Pseudomonas aeruginosa sepsis in a patient with neutropenia and acute lymphoblastic leukemia. A boy aged one year and two months was diagnosed with B-cell acute lymphoblastic leukemia on January 10 and underwent three courses of regular chemotherapy. He experienced neutropenia for 154 days and was hospitalized for vomiting, diarrhea and fever for 3 days. The day after hospitalization, Pseudomonas aeruginosa was isolated by blood culture and ceftazidime/avibactam was administered. Extracorporeal Membrane Oxygenation (ECMO) was used to provide continuous extracorporeal respiration and circulation for the patient. On day 8, the patient developed T. asahii fungemia. On day 10, he presented with necrotizing skin caused by Rhizopus delemar. He was treated with liposomal amphotericin B for Rhizopus delemar and voriconazole for T. asahii infection. Unfortunately, his health deteriorated and he died on day 11 due to the rapid progression of the infection and multiple organ failure. The management and treatment of such a complex infection requires a multidisciplinary approach and close monitoring of the patient's condition. Therefore, it is imperative to continue to research and report rare cases such as this to further understand the complexities of mucormycosis and trichosporidiosis coinfection and improve patient outcomes.

Imaging and clinical perspectives of rhino-orbito-cerebral mucormycosis: An epidemic born out of a pandemic

Purpose: To study the imaging and clinical perspectives of a recent epidemic of rhinocerebral mucormycosis during the COVID-19 (coronavirus disease) pandemic in India. Materials and Methods: This retrospective study included clinical and imaging data of 24 diagnosed cases of acute rhinocerebral mucormycosis who were presented to our hospital in May 2021. The clinical profile and imaging data of the patients were documented. Results: 18 patients (75.0%) had moderate to severe COVID-19 illness with a history of oral or intravenous corticosteroid intake. 19 patients (79.2%) had presented with hyperglycemia at the time of admission. Nasal discharges with or without black crusts and facial swelling (79.2%) were the most commonly reported complaints, followed by headaches or facial pain (62.5%). All cases demonstrated the involvement of one or more paranasal sinuses in imaging, with predominantly unilateral involvement. Extra sinusoidal involvement was commonly seen, with abnormalities seen in retromaxillary and masticator space, orbit, face, oral cavity, pterygopalatine fossa, and deep spaces of the neck, cavernous sinus, and brain. Conclusion: An upsurge of mucormycosis cases during the COVID-19 pandemic appears to be closely related to uncontrolled hyperglycemia and corticosteroid therapy. Computed tomography (CT) and magnetic resonance (MR) imaging are invaluable in establishing an early diagnosis, defining the extent of disease, and helping in immediate surgical planning.

Paciente endodóntico con mucormicosis rinocerebral: reporte de un caso / Endodontic patient with rhinocerebral mucormycosis: clinical case report

Se presenta en la Clínica de Endodoncia de la División de Estudios de Postgrado e Investigación (DEPeI) de la Facultad de Odontología (UNAM) una paciente diagnosticada con mucormicosis rinocerebral; en este artículo se presentan las variaciones en el manejo clínico de esta paciente y la importancia de mantener el mayor número de órganos dentales en boca para una mejor estabilidad del obturador palatino, tanto para las funciones de fonación como de masticación adecuadas.

A patient previously diagnosed with cerebral mucormycosis attended the Endodontics Clinic of the Graduate and Research School, National School of Dentistry, National University of Mexico (UNAM). This article presents variations in the clinical handling of the patient; stress is made on the importance of preserving the greatest number of teeth in the mouth to thus achieve better stability of the palatal obturator and establish suitable phonation and mastication functions.

Prosthetic rehabilitation after orbital exenteration: A case series.

Orbital exenteration is executed by the ophthalmic surgeon to treat various neoplasms or non-malignant diseases. But it leads to several functional, esthetic and psychological problems for the patients. Orbital prosthesis is a good alternative for cosmetic and psychological rehabilitation, if reconstructive surgery is not possible or not desired by the patient. In the following article, diff erent materials and retentive aids for fabrication of an orbital prosthesis given in the literature along with few novel methods have been discussed for four patients who underwent orbital exenteration. Factors that an ophthalmic surgeon should consider during surgery, which may later on help the prosthodontist to obtain good cosmetic results, are also discussed briefly. Remarkable results can be obtained if both work as a team for one common goal i.e. improvement of quality of life of the patient after orbital exenteration.

Rhinocerebral Mucormycosis with Isolated Sixth Nerve Palsy in An Immunocompetent Patient

We report a case of rhinocerebral mucormycosis in a 31 year old immunocompetent female presenting initially like acute rhinosinusitis with nasal stuffiness, severe headache, vomiting who soon developed isolated right lateral rectus palsy. Computed tomography(CT) scan of the Post-Nasal Spaces(PNS) showed an ill defined expansile heterogenous density mass in the sphenoid with extension into the ethmoids, nasal cavity, optic canal, superior orbital fissure, clivus and right temporal lobe with signal void in Magnetic Resonance Imaging(MRI). The debris and polypoid mucosa obtained on nasal endoscopy revealed mucormycosis on histopathologic examination. The patient was managed with urgent surgical debridement and medical management.

A case of successful posaconazole salvage therapy for rhinocerebral mucormycosis after failure of amphotericin B / 대한내과학회지

A 58-year-old man with uncontrolled type 2 diabetes mellitus developed rhinocerebral mucormycosis. The infection progressed to intracranial extension despite more than 5 weeks of treatment with amphotericin B. The patient then received oral posaconazole, 800 mg/d, in divided doses for 6 months. Salvage treatment with the new azole antifungal posaconazole resulted in dramatic clinical improvement as early as 1 week after the initiation of therapy. Oral posaconazole continued through 24 weeks of treatment, with marked clinical, mycological, and radiological improvements and no adverse events. Here we review the medical literature on rhinocerebral mucormycosis, which is a rapidly progressive and often fatal infection. The treatment of choice is amphotericin B, which failed in our patient. Our case report suggests that posaconazole appears to be a well tolerated and effective salvage treatment option for rhinocerebral mucormycosis, including disseminated disease.

Massive Cerebral Infarction Due to Rhinocerebral Mucormycosis

Rhinocerebral mucormycosis is rare, but fatal infection of the nasal cavity and sinuses. It can spread to the orbits and cranium within days, and prognosis is directly associated with length of time before diagnosis and treatment. Rhinocerebral mucormycosis can cause cerebral infarction via carotid artery occlusion. Therefore, neurosurgeon is paramount in making the proper management. We recently encountered a case of rhinocerebral mucormycosis with massive cerebral infarction. The clinical and radiological details of this case are presented here with a brief review of the literature.

A Case of Rhinocerebral Mucormycosis Misdiagnosed as Herpes Zoster / 대한피부과학회지

Mucormycosis is an opportunistic fungal infection frequently associated with diabetes mellitus or an immunocompromised status. Rhinocerebral mucormycosis, the most frequent form of mucormycosis, is characterized by facial pain, swelling, fever, blood-tinged rhinorrhea and shows a rapidly progressive course. Herein, we describe an immunocompromised and diabetic woman with rhinocerebral mucormycosis initially misdiagnosed as herpes zoster.

Vancomycin-Induced Agranulocytosis and Rhinocerebral Mucormycosis in a Patient with Continuous Ambulatory Peritoneal Dialysis-Associated Peritonitis / 대한신장학회잡지

Vancomycin-induced agranulocytosis is a rare side effect, but the increased use of vancomycin may disclose a more frequent occurrence. And it is a life-threatening complication, especially in end stage renal disease (ESRD) patients. We describe a 62-year-old patient with continuous ambulatory peritoneal dialysis (CAPD), who developed agranulocytosis after 3 weeks intraperitoneal administration of vancomycin for treatment of peritonitis caused by methicillin-resistant Staphylococcus haemolyticus. The agranulocytosis was resolved with granulocyte colony-stimulating factor (G-CSF) therapy and by the discontinuation of vancomycin. But, the patient developed subsequent rhinocerebral mucormycosis with invasion to skull base associated prolonged neutropenic period and expired in spite of surgical resection and intravenous administration of amphotericin. This case serves as a reminder to clinicians that patients receiving long-term treatment with vancomycin should have their white blood cell count monitored and vancomycin-induced agranulocytosis should be corrected promptly.

A Case of Rhinocerebral Mucormycosis Presenting with Facial Palsy / 대한피부과학회지

Mucormycosis is an opportunistic fungal infection affecting patients with diabetes or immune-compromised status, caused by species of the Mucoraceae family. Rhinocerebral mucormycosis, the most frequent form of mucormycosis, is characterized by a rapidly progressive and usually fatal course. Common initial symptoms of rhinocerebral mucormycosis include facial pain, facial swelling, headache, fever and blood-tinged rhinorrhea, however, facial palsy is relatively rare. We describe a diabetic female considered rhinocerebral mucormycosis presenting with facial palsy.

The Clue for Early Diagnosis and Prediction of Intracranial Involvement in Rhinocerebral Mucormycosis

BACKGROUND: Rhinocerebral mucormycosis (RCM) is an uncommon and fatal clinical syndrome resulting from an opportunistic infection caused by a fungus of the order Mucorales in immunocompromized patients. The mortality and morbidity in the patients with intracranial involvement is invariably high, and it was reported that most survivors had early diagnosis and received aggressive treatment. Therefore, we retrospectively reviewed four patients of pathologically confirmed mucormycosis to find out the clues for early diagnosis of RCM and for prediction of fatal intracranial involvement. METHODS: The clinical, radiological features and histo-pathological involvement sites in the patients with intracranial involvement were compared to those without intracranial involvement. RESULTS: All the patients had uncontrolled diabetes and were admitted with orbital involvement. On the precise physical examination, mucosal involvement was found in all patients; three of them in only nasal mucosa and one in oral palatal mucosa. Despite the vigorous antifungal therapy, two patients had intracranial involvement and expired. MRI finding of extensive sphenoid sinusitis adjacent cavernous sinus preceded the intracranial involvement of RCM, which was not found in the patients without intracranial involvement. CONCLUSIONS: Our findings suggest that early observation of oral or nasal mucosal changes might be an important clinical clue for differentiation of RCM from other causes of rhino-oculo-cerebral symptoms in uncontrolled diabetic patients, and that extensive sphenoid sinusitis might be an important radiological feature for predicting the fatal intracranial involvement of RCM.

Rhinocerebral Mucormycosis with Selective Cranial Nerve Palsy / 대한이비인후과학회지

Rhinocerebral mucormycosis, a rare opportunistic invasive fungal infection, is known to be fatal and aggressive. It is characterized by an aggressive necrotizing infection spreading from the nose to the paranasal sinuses, orbit, and the brain. This disease is most often associated with diabetic ketosis but can be seen with uremic acidisis, leukemia, malnutrition, AIDS, steroid, antimetabolic or antibiotic therapy, severe burn, septicemia, and treatment with immunosuppressive medications. Early clinical recognition of this potentially fatal disease is essential before irreversible changes occur. We report in this study about a 40-year-old man with poorly controlled diabetes mellitus who developed mucormycosis which involved unilateral paranasal sinus, orbit, and selective cranial nerves (especially, hypoglossal nerve). Despite of treatment he died from an abruptly developed acute respiratory distress syndrome that might be caused by mucormycotic hematogenous dissemination 33 days after admission.

A Case of Rhinocerebral Mucormycosis Caused by Mucor Species / 대한의진균학회지

Mucormycosis refers to infections caused by the order Mucorales of the class Zygomycetes. They are acute, rapidly developing, often fatal, opportunistic infections of immunocompromised patient. We report a case of rhinocerebral mucormycosis in a 73-year-old man with a 20-year history of poorly controlled diabetes mellitus. He had black necrotic ulcers on the hard palate, left blepharoptosis, left visual loss, left facial palsy and left hemiparesis. Brain and sino-orbital MRI scan showed protrusion of left eyeball, sinusitis on the both maxillary and ethmoid sinus and left middle cerebral artery infarction. Swab of black necrotic ulcer on the hard palate yielded large fungal hyphae on KOH preparation and inoculation of necrotic tissue on Sabouraud dextrose agar revealed a rapid growth of white to gray cottony colonies that soon turned gray to dark brown. Slide culture showed globose sporangia and no rhizoid or apophysis. These morphologic characteristics were those of the Mucor species. Histopathologic examination of the palatal lesion showed large, non-septated fungal hyphae in the dermis. The patient was treated with amphotericin B 50 mg/day for 45 days but died 114 days after admission.

Two Cases of Rhinocerebral Mucormycosis with Perineural Extension to the Pons

Rhinocerebral mucormycosis is a very rare, but mostly fatal fungal infection, usually found in diabetic or other immunocompromised hosts. It has a characteristic pattern of spread, invasion of vessel walls with subsequent local thrombotic infarction or direct invasion. But we found two cases contrast to these typical patterns of spread. Our cases initially originated in the nasal cavity and progressed to the cavernous sinus in usual manner, but after then, extended along the trigeminal nerve into the pons. MRI demonstrated the perineural spread, which was confirmed by pathology.

Two Cases of Rhinocerebral Mucormycosis with Perineural Extension to the Pons

Rhinocerebral mucormycosis is a very rare, but mostly fatal fungal infection, usually found in diabetic or other immunocompromised hosts. It has a characteristic pattern of spread, invasion of vessel walls with subsequent local thrombotic infarction or direct invasion. But we found two cases contrast to these typical patterns of spread. Our cases initially originated in the nasal cavity and progressed to the cavernous sinus in usual manner, but after then, extended along the trigeminal nerve into the pons. MRI demonstrated the perineural spread, which was confirmed by pathology.

Recurrent rhinocerebral mucormycosis: A case report

A case of recurrent rhinocerebral mucormycosis that has occurred during an induction chemotherapy for acute megakaryocytic leukemia in a 10 year-old boy is reported. He had suffered from high fever, proptosis, right eye ball pain and necrotic inflammation of hard palate during the chemotherapy of leukemia. CT scan of the paranasal sinus showed inflammatory change of right ethmoid and maxillary sinuses, and right orbital cystic mass which displaced medial rectus muscle. Pathologic examination of the inflammatory mass revealed mucormycosis with characteristic hyphae invading vessel walls. He was managed with 2 times of extensive debridement of necrotic tissue and currettage of cystic mass, and intravenous amphotericin-B for 80 days with apparent improvement. Seven months after discharge from the hospital, necrosis of posterior nasal septum and hard palate was noted for second time. It was managed again with 2 times of extensive debridement. Since this last operation he is on follow-up for 16 months uneventfully and is on therapy with low dose Ara-C in continuous remission.

A Case of Rhinocerebral Mucormycosis in a Diabetic Woman

We report a case of rhinocerebral mucormycosis in a 63-year-old female with a 1-year history of poorly controlled diabetes mellitus. She had black necrotic ulcers on the hard palate, bloody nasal discharge, swelling of the left side of her face, left blepharoptosis, proptosis, and conjunctival injection. Histopathological examination of the palatal lesion showed large, nonseptate, right-angled branching fungal hyphae in the dermis, and Rhizopus species was isolated on Sabouraud's agar media. The patient was treated with a combination of amphotericin B and surgical debridements but died of asphyxia one month after admission. This is a relatively uncommon, opportunistic infection occurring in a diabetic woman, and only several cases are reported in the Korean literature up to date.