Brain Diseases/etiology* ; Humans ; Leukemia ; Mucormycosis/complications* ; Nose Diseases

Adult ; Cerebrospinal Fluid Rhinorrhea ; surgery ; Endoscopy ; adverse effects ; Female ; Humans ; Mucormycosis ; etiology

We report a case of mucormycosis induced by Cunninghamella spp. infection in a ten-year-old girl with acute lymphoblastic leukemia, who developed fever and respiratory symptoms after chemotherapy and was diagnosed with invasive fungal disease. Peripheral blood DNA sequences were analyzed using metagenomic next-generation sequencing (mNGS), and by comparison with the Pathogens Metagenomics Database (PMDB), we identified Cunninghamella spp. with sequence number 514 as the pathogen. The patient was treated with amphotericin B combined with posaconazole and showed a favorable response. We searched Pubmed, Embase, CNKI, and Wanfang database for reports of cases of Cunninghamella spp. infection in children and retrieved 22 reported cases (including 12 males) with a median age of 13.5 (3-18) years. In these 22 cases, hematological malignancy was the most common underlying condition (19/22), and most of patients experienced an acute onset and rapid progression with respiratory symptoms (14/20) and fever (16/20) as the most common symptoms. CT imaging often showed unilateral lesions with varying imaging findings, including pulmonary nodules or masses, infiltrative changes, and pleural effusion. Definite diagnoses were established in 18 of the cases, and 4 had probable diagnoses; the lungs and skin were the most frequent organs compromised by the infection. A definite diagnosis of Cunninghamella spp. infection still relied on histopathological examination and fungal culture, but the molecular techniques including PCR and mNGS had shown potentials in the diagnosis. Almost all the cases received antifungal treatment after diagnosis (21/22), and 13 patients also underwent surgeries. Death occurred in 9 (42%) of the cases at a median of 19 (4-54) days after onset of the signs or symptoms. The patients receiving antifungal therapy combined with surgery had a high survival rate (9/13, 69%) than those with antifungal therapy alone (3/8, 37%). Invasive fungal disease is a common complication in immunoco-mpromised patients, but Cunninghamella spp. infection is rare and has a high mortality rate. In cases highly suspected of this disease, active diagnosis and early treatment are critical to improve the survival outcomes of the patients.
Adolescent ; Amphotericin B/therapeutic use* ; Antifungal Agents/therapeutic use* ; Child ; Cunninghamella ; Female ; Humans ; Male ; Mucormycosis/etiology*

Child, Preschool ; Female ; Humans ; Male ; Mucormycosis ; drug therapy ; etiology ; Precursor Cell Lymphoblastic Leukemia-Lymphoma ; complications ; Remission Induction

PURPOSE: To report a case of bilateral ophthalmic artery occlusion in rhino-orbito-cerebral mucormycosis. METHODS: Reviewed clinical charts, photographs, and fluorescein angiography RESULTS: An 89-year-old man with poorly controlled diabetes developed sudden bilateral ptosis, complete ophthalmoplegia of the right eye, and superior rectus palsy of the left eye. Brain and orbit magnetic resonance imaging showed midbrain infarction and mild diffuse sinusitis. On the 2nd day of hospitalization, sudden visual loss and light reflex loss developed. There were retinal whitening, absence of retinal arterial filling, and a total lack of choroidal perfusion on fluorescein angiography of the right eye. The left eye showed a cherry red spot in the retina and the absence of retinal arterial filling and partial choroidal perfusion on fluorescein angiography. On rhinologic examination, mucormyosis was noticed. Despite treatment, visual acuity and light reflex did not recover and he died 4 days after admission. CONCLUSIONS: Bilateral ophthalmic artery occlusion can occur in rhino-orbital-cerebral mucormycosis.
Aged, 80 and over ; Arterial Occlusive Diseases/diagnosis/*etiology ; Brain Diseases/*complications/diagnosis/microbiology ; Fatal Outcome ; Functional Laterality ; Humans ; Magnetic Resonance Imaging ; Male ; Mucormycosis/*complications/diagnosis/microbiology ; Ophthalmic Artery/*pathology ; Orbital Diseases/*complications/diagnosis/microbiology ; Paranasal Sinus Diseases/*complications/diagnosis/microbiology

PURPOSE: To report a case of bilateral ophthalmic artery occlusion in rhino-orbito-cerebral mucormycosis. METHODS: Reviewed clinical charts, photographs, and fluorescein angiography RESULTS: An 89-year-old man with poorly controlled diabetes developed sudden bilateral ptosis, complete ophthalmoplegia of the right eye, and superior rectus palsy of the left eye. Brain and orbit magnetic resonance imaging showed midbrain infarction and mild diffuse sinusitis. On the 2nd day of hospitalization, sudden visual loss and light reflex loss developed. There were retinal whitening, absence of retinal arterial filling, and a total lack of choroidal perfusion on fluorescein angiography of the right eye. The left eye showed a cherry red spot in the retina and the absence of retinal arterial filling and partial choroidal perfusion on fluorescein angiography. On rhinologic examination, mucormyosis was noticed. Despite treatment, visual acuity and light reflex did not recover and he died 4 days after admission. CONCLUSIONS: Bilateral ophthalmic artery occlusion can occur in rhino-orbital-cerebral mucormycosis.
Aged, 80 and over ; Arterial Occlusive Diseases/diagnosis/*etiology ; Brain Diseases/*complications/diagnosis/microbiology ; Fatal Outcome ; Functional Laterality ; Humans ; Magnetic Resonance Imaging ; Male ; Mucormycosis/*complications/diagnosis/microbiology ; Ophthalmic Artery/*pathology ; Orbital Diseases/*complications/diagnosis/microbiology ; Paranasal Sinus Diseases/*complications/diagnosis/microbiology

Gastrointestinal mucormycosis is an uncommon opportunistic fungal infection often presents in immunocompromised patients. Direct invasion of the intestinal walls by spores from ingested food is the main pathogenetic mechanism of this disease, which usually takes place in stomach and colon. Early diagnosis is critical, especially in vascular invasive types, due to its high mortality rate close to 100%. In the past when appropriate diagnostic tools were not available, mucormycosis were frequently found with autopsy. The advance in current endoscopic technology has increased diagnostic rate and made successful management available with appropriate treatments such as debridement of contaminated tissues. In this case report, we discussed a case of ileal mucormycosis diagnosed by colonoscopy and treated with anti-fungal agent successfully.
Amphotericin B/therapeutic use ; Antifungal Agents/therapeutic use ; Colonoscopy ; Humans ; Ileal Diseases/*diagnosis/microbiology/therapy ; Ileum/pathology ; Immunocompromised Host ; Leukemia, Myeloid, Acute/*complications/drug therapy ; Male ; Mucormycosis/*diagnosis/etiology/therapy ; Tomography, X-Ray Computed ; Young Adult

Peritonitis is a common problem in patients undergoing peritoneal dialysis. However, peritonitis due to Cunninghamella (C.) bertholletiae, a fungus of the class Zygomycetes, is rare. We present a case of fungal peritonitis in a patient on continuous ambulatory peritoneal dialysis due to kidney rejection. Direct examination of the patient's peritoneal fluid showed fungal hyphae, and the culture was identified as C. bertholletiae. A cumulative dose of 1,600 mg fluconazole was given to the patient intraperitoneally over a one-week period. When his condition had stabilised, oral antifungal treatment was administered for two weeks. After removal of the Tenckhoff catheter, the patient was discharged with arteriovenous fistulation for haemodialysis. Zygomycosis due to C. bertholletiae is often fatal and non-responsive to systemic antifungal therapy. This case is the first from India with a successful outcome, and highlights the importance of early detection and intervention for successful outcome of peritonitis caused by C. bertholletiae.
Antifungal Agents ; administration & dosage ; Cunninghamella ; isolation & purification ; Drug Administration Routes ; Fluconazole ; administration & dosage ; Follow-Up Studies ; Graft Rejection ; complications ; Humans ; Kidney Failure, Chronic ; complications ; therapy ; Kidney Transplantation ; Male ; Middle Aged ; Mucormycosis ; drug therapy ; etiology ; microbiology ; Peritoneal Dialysis, Continuous Ambulatory ; adverse effects ; Peritonitis ; drug therapy ; etiology ; microbiology

INTRODUCTIONInvasive fungal pan-sinusitis can present atypically with severe acute visual loss with minimal anterior orbital inflammation. We describe 2 such cases with a background of uncontrolled diabetes.

CLINICAL PICTURERespective clinical presentations of orbital apex and cavernous sinus syndromes were associated with isolation of Aspergillus galactomannan and Rhizopus.

TREATMENTUrgent extensive surgical debridement and systemic antifungal is necessary.

OUTCOMEClinical improvement of the ocular motor nerves can be expected within 2 months of treatment but visual loss is usually permanent.

CONCLUSIONUnderlying pansinusitis is an important differential for acute visual loss, especially in uncontrolled diabetics. Early treatment determines outcome.

Adult ; Antifungal Agents ; therapeutic use ; Antigens, Fungal ; analysis ; Aspergillosis ; complications ; diagnosis ; therapy ; Aspergillus ; immunology ; isolation & purification ; Debridement ; methods ; Diabetes Complications ; blood ; complications ; therapy ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Humans ; Magnetic Resonance Imaging ; Male ; Middle Aged ; Mucormycosis ; complications ; diagnosis ; therapy ; Rhizopus ; immunology ; isolation & purification ; Sinusitis ; complications ; diagnosis ; therapy ; Tomography, X-Ray Computed ; Vision, Low ; diagnosis ; etiology ; therapy