Rhino-orbital entomophthoramycosis in pediatric patients: Report of two cases.
Ontology highlight
ABSTRACT:
SUBMITTER: Ordonez-Ortega J
PROVIDER: S-EPMC8927958 | biostudies-literature | 2022 Jun
REPOSITORIES: biostudies-literature
Publications
Rhino-orbital entomophthoramycosis in pediatric patients: Report of two cases.
Ordoñez-Ortega Javier J Borbolla-Pertierra Ana María AM Sánchez-Carrillo Yolotl Y Costta-Michuy María de Los Ángeles MLÁ Mirabal-García Mónica M Luengas-Bautista Marlen M Palacios-Reyes Deborah D
Medical mycology case reports 20220307
Here we present two cases of entomophthoramycosis in young children, these were the first cases ever diagnosed at our center. Both cases represented a diagnostic and treatment challenge. Surgical treatment was key in the management of both patients. Even though atypical disease was encountered, clinical response was obtained after surgical excision and antifungal treatment for a few months. ...[more]
Similar Datasets
Project description:BackgroundOrbital apex syndrome is a symptom complex of visual loss and ophthalmoplegia resulting from a disease involving the orbital apex. It can be caused by inflammation, infection, and malignancies. Mucormycosis is an infection caused by filamentous saprophytes of the order Mucorales. It is ubiquitous, and the infection can occur from ingesting contaminated food, inhaling spores, or injecting the disrupted skin or wounds. It is mainly a disease of the immunocompromised, affecting patients with poorly controlled diabetes, organ transplant recipients, and patients with hematological malignancies. We present the case of a man with orbital apex syndrome resulting from rapidly spreading rhino-ocular cerebral mucormycosis, who had a poor outcome despite an aggressive combined medical and surgical treatment. This is an unusual cause of orbital apex syndrome.Case presentationA 46-year-old Bangladeshi man presented to the emergency department with a history of toothache and pain in the left eye. On examination, the patient had a left-sided periorbital edema, ptosis, and proptosis. He had complete ophthalmoplegia and absent direct pupillary response in the left eye. Magnetic resonance imaging of the brain and orbit showed bilateral ethmoidal, left frontal, maxillary, and sphenoidal sinusitis with left orbital cellulitis. His left orbit was surgically decompressed and histology confirmed mucormycosis. Despite aggressive treatment, the patient had only a partial improvement in his symptoms.ConclusionsMucormycosis is a rare disease that can easily be misdiagnosed, leading to delayed treatment and disease dissemination. Clinicians must be suspicious of mucormycosis in patients presenting with multiple cranial nerve palsy.
| S-EPMC11575186 | biostudies-literature
Project description:There has been a surge of rhino-orbital mucormycosis cases in India in the wake of the second wave of the COVID-19 pandemic. It has been widely suggested that dysglycemia resulting from diabetes which is a common comorbidity in COVID-19 patients, and indiscriminate steroid use has resulted in this surge. We report a series of 13 cases of rhino-orbital mucormycosis in COVID-19 patients admitted to our center between mid-April and early June 2021. The cases showed a male preponderance, two patients had loss of vision, and four of them showed intracranial extension of disease. Twelve patients had received steroids and 12 had preexisting or newly diagnosed diabetes, both steroid use and diabetes being the most common identified risk factors. Considering other possible risk factors, immunosuppressed state, antiviral or ayurvedic (Indian traditional) medications, and oxygen therapy were not associated with a definite risk of mucormycosis, because they were not present uniformly in the patients. We propose that COVID-19 itself, through molecular mechanisms, predisposes to mucormycosis, with other factors such as dysglycemia or steroid use increasing the risk.
| S-EPMC8832906 | biostudies-literature
Project description:BackgroundViral or bacterial infections can trigger auto-immune inflammatory reactions and conditions in children. Self-reactivity arises due to similarities in molecular structures between pathogenic microorganisms and regular body structures with consequent immune-cross reactions. Reactivation of latent Varicella Zoster Virus (VZV) infections can cause neurological sequalae, including cerebellitis, post-herpetic neuralgias, meningo/encephalitis, vasculopathy and myelopathy. We propose a syndrome caused by auto-immune reactivity triggered by molecular mimicry between VZV and the brain, culminating in a post-infectious psychiatric syndrome with childhood VZV infections.Case presentationTwo individuals, a 6-year-old male and 10-year-old female developed a neuro-psychiatric syndrome 3-6 weeks following a confirmed VZV infection with intrathecal oligoclonal bands. The 6-year-old male presented with a myasthenic syndrome, behavior deterioration and regression in school, he was poorly responsive to IVIG and risperidone, however had a pronounced response to steroid treatment. The 10-year-old female presented with marked insomnia, agitation, and behavioral regression as well as mild bradykinesia. A trial of neuroleptics and sedatives resulted in a mild unsustained reduction in psychomotor agitation and IVIG was also unsuccessful, however the patient was very responsive to steroid therapy.ConclusionPsychiatric syndromes with evidence of intrathecal inflammation temporally related to VZV infections that are responsive to immune modulation have not been described before. Here we report two cases demonstrating neuro-psychiatric symptoms following VZV infection, with evidence of persistent CNS inflammation following the resolution of infection, and response to immune modulation.
| S-EPMC9969201 | biostudies-literature
Project description:Although voriconazole, a triazole antifungal, is a safe drug, treatment with this agent is associated with certain adverse events such as hepatic, neurologic, and visual disturbances. The current report presents two cases, one a 9-year-old boy and the other a 17-year-old girl, who experienced neurologic side effects associated with voriconazole therapy. Our aim is to remind readers of the side effects of voriconazole therapy in order to prevent unnecessary investigations especially for psychological and ophthalmologic problems. The first case was a 9-year-old boy with cystic fibrosis and invasive aspergillosis that developed photophobia, altered color sensation, and fearful visual hallucination. The second case was a 17-year-old girl with cystic fibrosis and allergic bronchopulmonary aspergillosis, and she experienced photophobia, fatigue, impaired concentration, and insomnia, when the dose of voriconazole therapy was increased from 12 mg/kg/day to 16 mg/kg/day. The complaints of the two patients disappeared after discontinuation of voriconazole therapy. Our experience in these patients reminded us of the importance of being aware of the neurologic adverse events associated with voriconazole therapy in establishing early diagnosis and initiating prompt treatment. In addition, although serum voriconazole concentration was not measured in the present cases, therapeutic drug monitoring for voriconazole seems to be critically important in preventing neurologic side effects in pediatric patients.
| S-EPMC4895037 | biostudies-other
Project description:IntroductionThe aim of this study was to report a case of ROCM related to nasogastric intubation who was survived by liposomal amphotericin B (LAmB) combination therapy in situ without orbital exenteration.Case presentationA 44-year-old woman presented with a 1-week history of rapidly enlarging swelling on the right nose, cheek, and lower eyelid after underwent gastrointestinal decompression. The lesions were derived from the nasal area where the nasogastric tube had been placed. Based on the biopsy results and clinical manifestations, ROCM was diagnosed. Immediate combination therapy with intravenous LAmB and micafungin and multisection debridement of the right facial region were applied. Postoperative treatment included cleaning, irrigating, and local dressing of the wound area using LAmB. LAmB was also used daily as binocular eye drops against deep infection on the eyeballs. The patient recovered well 4 months later and remained free of disease after 40 months of follow-up.ConclusionThis case adds to our knowledge on the potential risk of nasogastric intubation for mucormycosis infection. Nasogastric tube may be the source of infection associated with ROCM. This report evaluates the beneficial effect of LAmB combination therapy in situ for cleaning, irrigating, local wound dressing, and eye drops on lesion areas. The combination of LAmB as cleaning, irrigating, local dressing solution, and eye drops to control intraocular and intraorbital ROCM infection has not been previously reported to our knowledge. These methods provide multiple choices to substitute for orbital exenteration on the survival of ROCM patients.
| S-EPMC10846874 | biostudies-literature
Project description:BackgroundMucormycosis, is a rare yet potentially life-threatening fungal infection common in immunocompromised patients. Despite optimal care, mucormycosis in haemato-oncological patients often results in poor outcomes. This case series details the presentations and unique challenges faced during the management of patients with acute myeloid leukemia who developed rhino-cerebral mucormycosis.Case presentationWe present three cases of rhino-cerebral mucormycosis in patients with acute myeloid leukemia: two females aged 35 and 29, and one male aged 42. Symptoms manifested during chemotherapy induction, with all patients experiencing symptoms suggestive of rhino, orbital, or cerebral infection in a background of severe neutropenia (ANC < 0.5). Nasal endoscopy revealed necrotic tissue in all cases, with contrast-enhanced computer tomography (CECT) confirming invasive fungal infection. Rhizopus species were isolated in cultures from the two female patients, and histopathological evidence of fungal invasion was noted in one. Prompt treatment with liposomal Amphotericin B combined with surgical debridement with functional endoscopic sinus surgery (FESS) and treatment of neutropenic sepsis resulted in the survival of two patients, though one succumbed during treatment.ConclusionsThis case series highlights the importance of early clinical suspicion and treatment of mucormycosis in hematological malignancies. Due to mild and atypical presentations and lack of confirmation by microbiological and histological methods, a multifaceted diagnostic approach combining clinical, laboratory, and imaging modalities is essential. A multidisciplinary treatment approach with the management of concomitant complications like neutropenic sepsis is crucial for better outcomes.
| S-EPMC11670406 | biostudies-literature
Project description:PurposeTo describe the rare entity of concurrent herpetic acute retinal necrosis (ARN) and orbital inflammation.ObservationsTwo cases of ARN with simultaneous orbital inflammation are described. A 40-year old male presented with a painful left eye and hand motion visual acuity (VA). Both panuveitis and orbital inflammation with involvement of the sclera and optic nerve were observed. He was initially treated with oral steroid therapy, after which the orbital inflammation improved but the panuveitis remained. A diagnostic anterior chamber paracentesis was positive for HSV-2 by PCR. He was started on systemic antivirals, but ultimately developed a retinal detachment. The second patient was an 18-year old female with hand motion VA in the left eye. Panuveitis and severe conjunctival chemosis were observed. MRI demonstrated dacryoadenitis with preseptal inflammation. The patient was initially started on oral steroid therapy, which alleviated the orbital inflammation but not the intraocular inflammation. An anterior chamber diagnostic paracentesis was positive for HSV-1, after which the patient underwent vitrectomy for a retinal detachment. The patient was started on systemic antiviral therapy.Conclusions and importanceHerpetic disease should remain on the differential for cases of concurrent intraocular and orbital inflammation. Early recognition of this process may help prevent severe vision loss. It is important to recognize that orbital inflammation secondary to herpetic disease may be diverse in its presentation.
| S-EPMC6393694 | biostudies-literature
Project description:PurposeTo present the different clinical manifestations of rhino-orbital mucormycosis (ROM) co-infection in severe COVID-19 patients.Study designProspective observational clinical study METHODS: Among 32,814 patients hospitalized with the diagnosis of COVID-19 between March 2020 and December 2020 in our center, eleven microbiologically confirmed ROM co-infection cases in severe COVID-19 patients were evaluated.ResultsThere were nine men and two women with a mean age of 73.1 ± 7.7 years. Eight patients had uncontrolled type 2 diabetes with a mean diagnosis duration of 12.1 ± 4.4 years. All patients had COVID-19-associated acute respiratory distress syndrome and received corticosteroids. The mean time interval between COVID-19 diagnosis and ROM diagnosis was 14.4 ± 4.3 days. Seven patients (63.6%) had orbital apex syndrome, and four patients (36.4%) presented with orbital cellulitis. Endophthalmitis was detected in 54.5% of patients, and two of these patients developed retinoschisis. CT scan/MRI revealed sino-orbital involvement in all patients, and three of these had cerebral involvement at initial presentation. All patients received intravenous and retrobulbar liposomal amphotericin B and had undergone radical debridement of involved sinuses. Intravitreal liposomal amphotericin B injected in patients with endophthalmitis. Despite all measures, 63.6% of patients expired.ConclusionsSevere COVID-19 is associated with a significant incidence of ROM with higher mortality rates due to immune dysregulation and the widespread use of steroids. Physicians should be aware of the possibility of this infection in patients with COVID-19. An aggressive multidisciplinary approach can help to reduce mortality.
| S-EPMC8165350 | biostudies-literature
Project description:PurposeMucormycosis is a severe fungal infection caused by species of the order Mucorales. Early and accurate diagnosis is a prerequisite in the management of the disease. In the present study, we evaluated and compared two PCR-based techniques for the diagnosis and identification of mucormycosis in patients with rhino-orbital mucormycosis (ROM) post-COVID-19.MethodsDiagnosed clinically and radiologically, 25 patients of ROM were included in the study and endoscopically or blind collected nasal swabs or orbital tissues were submitted for microbiological evaluation (direct microscopy + culture) and PCR using primers targeting two different loci (ITS and 28S rDNA region) for diagnosis. All PCR products were further processed for species identification using Sanger sequencing whenever possible.ResultOf the 25 samples included in the study, 16 samples were positive for presence of fungal filaments by Smear suggestive of Mucorales sp., but only 7/25 grew in culture. ITS-based PCR was able to identify mucormycosis in 7/25 (28%) samples and 28S rDNA PCR showed positivity for 19/25 (76%) samples. Rhizopus oryzae was found to be the predominant species in our study. The sensitivity and specificity of 28S rDNA PCR compared to culture were found to be 85.71% and 27.78%, respectively, while for ITS-based PCR, they were 42.86% and 77.78%, respectively.Conclusions28S rDNA-based PCR is a reliable and sensitive method for early diagnosis of mucormycosis. Molecular techniques have shown a promising future to provide quick and effective treatment by accurately identifying the aetiologic agent.
| S-EPMC9684940 | biostudies-literature
Project description:In this report, we present a case of rhino-orbital mucormycosis in a 57-year-old female with poorly controlled diabetes mellitus. The only mold cultured at 25°C, 37°C, and 40°C from a specimen of the nasal crust was identified phenotypically and independently using nuclear ribosomal DNA sequence data as Thamnostylum lucknowense. To our knowledge, this report presents the first data implicating this mucoraceous fungus as a mycotic agent of human infection.
| S-EPMC3318503 | biostudies-literature