Objective To compare the clinical features and auxiliary examination results of sinus-originated intracranial aspergillosis（ICA）and rhino-orbito-cerebral mucormycosis（ROCM）. Methods A retrospective analysis was performed for the clinical data of seven patients with sinus-originated ICA and six patients with ROCM who were diagnosed and treated in our hospital from January 2018 to January 2023. Results The pathogenic factor of CNS fungal infection was observed in all patients with ROCM and 57.1% of the patients with sinus-originated ICA. Compared with the patients with sinus-originated ICA，the patients with ROCM were more likely to develop ocular symptoms and signs such as exophthalmos，periorbital swelling，and intraocular inflammation，as well as non-ocular symptoms and signs including fever，facial swelling，and facial nerve palsy. Compared with the patients with sinus-originated ICA，the patients with ROCM had significantly greater increases in blood glucose，erythrocyte sedimentation rate，C-reactive protein，and procalcitonin. Cranial magnetic resonance imaging showed that sinus-originated ICA often affected the paranasal sinuses，orbital apices，retro-orbital regions，and cavernous sinuses，while the patients with ROCM were more likely to have sinusitis，bone destruction，and mucormycosis invading cerebral vessels. As for treatment，50% of the patients with ROCM and 42.9% of the patients with sinus-originated ICA received antifungal therapy alone，while 50% of the patients with ROCM and 57.1% of the patients with sinus-originated ICA received a combination of medication and surgery. By follow-up to September 2023，50% of the patients with ROCM and 28.6% of the patients with sinus-originated ICA had no response to treatment and died. Conclusion Both sinus-originated ICA and ROCM have rapid progression，and ROCM survivors with diabetes are more likely to experience disabilities. Timely diagnosis and initiation of antifungal therapy，correction of underlying predisposing factors，and active surgical debridement are essential for the treatment of CNS fungal infections.
Aspergillus ; Mucormycosis

Humans ; Adult ; Mucormycosis ; Leukemia, Myeloid, Acute/complications* ; Acute Disease ; Antifungal Agents

Mucormycosis is an important invasive fungal disease that is difficult to diagnose and treat, and has a high mortality rate. To improve the diagnosis and treatment of mucormycosis by clinicians, the Medical Mycology Society of Chinese Medicine and Education Association engaged multidisciplinary experts to compile this expert consensus. This consensus refers to the latest international guidelines for diagnosis and treatment of mucormycosis, combined with the characteristics and treatment needs of mucormycosis in China and covers the following eight aspects to provide reference for Chinese clinicians: pathogenic agents, high-risk factors, clinical types, imaging manifestations, etiological diagnosis, clinical diagnosis, treatment, and prevention.
Humans ; Mucormycosis/drug therapy* ; Consensus ; China

Bacterial Infections ; Humans ; Lymphoma, Large B-Cell, Diffuse/diagnosis* ; Mucormycosis/diagnosis* ; Respiratory Sounds ; Thyroid Gland

Aspergillosis ; Humans ; Mucor ; Mucormycosis/drug therapy*

Objective: To investigate the clinical features, diagnosis,treatment and prognosis of children with acute lymphoblastic leukemia complicated with mucormycosis, and to improve the understanding of the disease. Methods: The clinical data of 3 children with acute lymphoblastic leukemia (ALL) complicated with mucormycosis treated at the First Affiliated Hospital of Zhengzhou University between October 2020 and January 2021 were analyzed retrospectively. Literature search and review covered the China national knowledge infrastructure, Wanfang database and Pubmed using the keywords of "acute lymphoblastic leukemia" and "mucormycosis" up to June 2021. Results: Case 1, a 12-year-old boy, was diagnosed with ALL, developed fever and chest pain during induction therapy. The Metagenomic next-generation sequencing (mNGS) testing of alveolar perfusion fluid suggested infection with Rhizopus oryzae. Amphotericin B combined with posaconazole was applied and amphotericin B was removed after improvement. Bone destruction was indicated by CT. Amphotericin B was applied again. Case 2, a 4-year-old boy, with a history of pallor and tetter, was diagnosed with ALL. He developed cough and fever during induction therapy. mNGS of blood suggested infection with Rhizomucor pusillus. Amphotericin B combined with voriconazole was applied, but the situation was not significantly improved. The disseminated infection occurred. Amphotericin B combined with posaconazole was applied and vacuum sealing drainage was performed. Case 3, a 2-year-old girl, was diagnosed with ALL, developed fever and cough during induction therapy. Rhizomucor pusillus was indicated by mNGS. Amphotericin B combined with posaconazole was used, and posaconazole was stopped after improvement. Follow-up until June 2021, the condition of the 3 children improved. There was no recurrent Mucor infection, and the primary hematopathy was in complete remission. According to the literature, 7 reports were found in Chinese journals, while 17 reports were found in English literature, 25 cases have been reported. Among a total of 28 children, 11 cases rhino-orbito-cerebral mucormycosis, four pulmonary mucormycosis, 2 cutaneous mucormycosis, 2 gastrointestinal mucormycosis and 9 disseminated mucormycosis. There were 17 cases developed infection during induction chemotherapy, 8 cases during maintenance therapy, 3 cases after hematopoietic stem cell transplantation. Voriconazole was used in 15 cases; 19 cases were treated with combined surgery, 7 cases were treated with drugs only, 2 cases were untreated; 21 cases showed improvement after treatment. Death occurred in seven cases. Conclusions: ALL complicated with mucormycosis often occurs in the stage of induction therapy. The clinical features lacked specificity, mNGS can help find the pathogen and provide evidence for diagnosis. Surgical treatment also could be combined when necessary, which is helpful to improve the prognosis.
Amphotericin B/therapeutic use* ; Antifungal Agents/therapeutic use* ; Child ; Child, Preschool ; Female ; Humans ; Male ; Mucormycosis/drug therapy* ; Precursor Cell Lymphoblastic Leukemia-Lymphoma/drug therapy* ; Retrospective Studies

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*

OBJECTIVE: To investigate the clinical features, treatment and prognosis of patients with hematological diseases complicated with mucor infection. METHODS: The risk factors, clinical features, treatment regimen and prognosis of 18 hematological disease patients with mucor infection diagnosed by histopathology in our center from April 2014 to June 2020 were retrospectively analyzed. RESULTS: Thirteen males and five females, with an average age of 30 (13－54) years old, were diagnosed as mucor infection by histopathological examination at the site of infection, including 16 cases of mucor infection alone and 2 cases of mucor + aspergillus mixed infection. There were 12 cases with malignant hematological disease and 6 cases with severe aplastic anemia, all of whom with long-term agranulocytosis, and their clinical manifestations and imaging findings were not specific. The common sites of infection were sinuses and lungs, and some patients showed multiple systemic manifestations. The remission status of hematological diseases and recovery of immune function showed an impact on the prognosis. All the patients were treated with amphotericin B liposome combined with posaconazole, and 15 patients were treated with surgery combined with antifungal drugs, 9 of whom were effective and 6 were ineffective, while intravenous administration in 3 cases was ineffective. CONCLUSION It is difficult to diagnose hematological disease complicated with mucor infection. After early diagnosis, prognosis can be improved by amelioration of primary state and combination of drugs and surgery.
Adolescent ; Adult ; Antifungal Agents/therapeutic use* ; Female ; Hematologic Diseases/complications* ; Humans ; Male ; Middle Aged ; Mucormycosis/drug therapy* ; Prognosis ; Retrospective Studies ; Young Adult

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

Introduction: Subcutaneous zygomycosis or basidiobolomycosis is caused by a saprophytic fungi Basidiobolus ranarum (B. ranarum). It presents clinically as a slowly growing painless subcutaneous nodule more commonly on the extremity, trunk and rarely on other parts of the body. Mode of infection has not been established but most likely follows traumatic implantation. We report a gradually evolving case of basidiobolomycosis in a 12-year-old immunocompetent Filipino male with no history of trauma who responded dramatically to itraconazole. Case summary: Subcutaneous zygomycosis is caused by Basidiobolus ranarum (B. ranarum) which is endemic in parts of Africa, India and other parts of Asia. We report an evolving case of a Filipino male child who presented with chronic and persistent subcutaneous nodules and plaques on the left extremity. Dense cell infiltrates consisting of lymphocytes, histiocytes and eosinophils were seen on histology with non-septated hyphal structures on Gomori’s methenamine silver stain. Fungal culture of the lesion yielded B. ranarum. Complete resolution of the lesions were observed after 6 months of itraconazole therapy alone. Conclusion: This case demonstrated the gradual progression of the disease, and that the lack of a history of trauma does not exclude its diagnosis. Subcutaneous zygomycosis should be highly suspected in chronic painless subcutaneous nodules with swelling, and its early recognition is crucial to prevent progression of the disease. In patients whom an infectious cause is suspected, appropriate cultures, special staining, and sometimes repeat biopsies of new cutaneous lesions may be helpful in determining or ruling out the associated disease. Furthermore, performing PAS stain alone to identify the presence or absence of a fungi is insufficient, rather, additional staining such as GMS should be done to heighten detection of fungal elements.
Child ; Dermatology ; Itraconazole ; Basidiobolus ranarum ; entomophthoromycosis ; Zygomycosis