Primary Cutaneous Zygomycosis Caused by<i>Rhizomucor variabilis:</i>A New Endemic Zygomycosis? A Case Report and Review of 6 Cases Reported from China

Lu, Xinliang; Liu, Ze‐Hu; Shen, Ya; She, Xiaodong; Lu, Guixia; Zhan, Ping; Fu, Meihua; Zhang, Mengli; Ge, Yiping; Liu, Weida

doi:10.1086/600817

Cited by 56 publications

(41 citation statements)

References 5 publications

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“…Rhizomucor species are an infrequent cause of human disease, while Rhizomucor variabilis is rarely reported as an etiologic agent, with few case reports published recently (8,14). In one retrospective review of zygomycosis in children, among 77…”

Section: Case Reportmentioning

confidence: 99%

Rhizomucor variabilis var. regularior and Hormographiella aspergillata Infections in a Leukemic Bone Marrow Transplant Recipient with Refractory Neutropenia

Abuali¹,

Posada²,

Toro³

et al. 2009

J Clin Microbiol

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Rhizomucor variabilis and Hormographiella aspergillata rarely cause human infections. This report details a fatal case of a 14-year-old female with leukemia posthematopoietic cell transplant and relapse with refractory pancytopenia. The patient first developed an R. variabilis var. regularior palate infection and later developed a cutaneous H. aspergillata infection while on posaconazole and caspofungin therapy. CASE REPORTThe patient was a 14-year-old female with a history of acute myelogenous leukemia diagnosed in July 2007. The patient underwent an allogeneic HLA-matched bone marrow transplant in December 2007 and experienced relapse in April 2008, with a bone marrow aspirate showing a preponderance of blasts, and later failed reinduction chemotherapy. In September 2008, a computed tomography (CT) scan of the chest 2 weeks prior to admission revealed patchy ground-glass opacities with tiny peripheral nodular densities in both lung fields (sparing the left upper lobe) and a 1.2-cm nodule in the right upper lobe. The patient's condition was deemed too fragile to tolerate a diagnostic lung biopsy. She was empirically treated with broad antibacterial and antifungal coverage that included voriconazole. Of interest, the patient also started drinking an herbal tea remedy of an unknown variety. Shortly afterwards, the patient presented with a 2-week history of odynophagia and persistent febrile neutropenia. An examination showed that the patient had white plaques involving the soft palate and pharynx. A smear stained with calcofluor white from a throat culture showed hyphal elements and conidiophores. A biopsy of the palate lesion showed submucosa and mucosa infiltrated with hyphal forms with sparse septation, rare branching, and chlamydoconidia. The patient was started empirically on 800-mg oral (p.o.) doses of posaconazole twice a day (BID). A CT scan of the head and sinuses was negative, while a CT scan of the lungs confirmed pulmonary nodules that had been previously visualized. Serial galactomannan assay results were negative. The fungal isolate obtained in culture from palate biopsy was tentatively identified as a Rhizomucor sp. by the Mount Sinai clinical microbiology laboratory. The isolate was sent to the Mycology Laboratory of the New York State Department of Health for further characterization. The option of surgical debridement was declined by the family due to potentially severe morbidity. At week 2 of therapy, caspofungin was added as an adjunct therapy. By week 2 of therapy, the lesion had decreased in size, with improved symptoms. At week 3 of therapy, she began receiving a new regimen of chemotherapy, along with granulocyte infusions. By week 4, the patient's symptoms had resolved. By week 5 of therapy, the palate lesion was no longer visualized. Throughout, the patient had persistent severe refractory pancytopenia. One and onehalf months after initiation of therapy, she developed an altered mental status and had a generalized seizure. A CT scan of the brain showed multiple hypodense lesions of the c...

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Section: Case Reportmentioning

confidence: 99%

Rhizomucor variabilis var. regularior and Hormographiella aspergillata Infections in a Leukemic Bone Marrow Transplant Recipient with Refractory Neutropenia

Abuali¹,

Posada²,

Toro³

et al. 2009

J Clin Microbiol

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“…Chronic progression (3 months to 10 years) of cutaneous infection is characteristic of R. variabilis infection, which was described in eight reported cases in studies in China and Japan (196,328,372). In six of these cases, the infection occurred after trauma (196,328).…”

Section: Rhizomucor Pusillusmentioning

confidence: 96%

Mucormycosis Caused by Unusual Mucormycetes, Non-Rhizopus, -Mucor, and -Lichtheimia Species

2011

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SUMMARY Rhizopus , Mucor , and Lichtheimia (formerly Absidia ) species are the most common members of the order Mucorales that cause mucormycosis, accounting for 70 to 80% of all cases. In contrast, Cunninghamella , Apophysomyces , Saksenaea , Rhizomucor , Cokeromyces , Actinomucor , and Syncephalastrum species individually are responsible for fewer than 1 to 5% of reported cases of mucormycosis. In this review, we provide an overview of the epidemiology, clinical manifestations, diagnosis of, treatment of, and prognosis for unusual Mucormycetes infections (non- Rhizopus , - Mucor , and - Lichtheimia species). The infections caused by these less frequent members of the order Mucorales frequently differ in their epidemiology, geographic distribution, and disease manifestations. Cunninghamella bertholletiae and Rhizomucor pusillus affect primarily immunocompromised hosts, mostly resulting from spore inhalation, causing pulmonary and disseminated infections with high mortality rates. R. pusillus infections are nosocomial or health care related in a large proportion of cases. While Apophysomyces elegans and Saksenaea vasiformis are occasionally responsible for infections in immunocompromised individuals, most cases are encountered in immunocompetent individuals as a result of trauma, leading to soft tissue infections with relatively low mortality rates. Increased knowledge of the epidemiology and clinical presentations of these unusual Mucormycetes infections may improve early diagnosis and treatment.

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“…Rhinocerebral and pulmonary infection are linked to ketoacidotic diabetes and severe neutropenia, respectively, while immunosuppression and prolonged deferoxamine therapy predispose for disseminated infection (11). Chronic disorders observed in individuals without severe immune or metabolic dysfunction are exceptional cases (25). Also, renal mucormycosis tends to occur in immunocompetent individuals (21,27).…”

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confidence: 99%

Antifungal Susceptibility and Phylogeny of Opportunistic Members of the Order Mucorales

et al. 2012

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The in vitro susceptibilities of 66 molecularly identified strains of the Mucorales to eight antifungals (amphotericin B, terbinafine, itraconazole, posaconazole, voriconazole, caspofungin, micafungin, and 5-fluorocytosine) were tested. Molecular phylogeny was reconstructed based on the nuclear ribosomal large subunit to reveal taxon-specific susceptibility profiles. The impressive phylogenetic diversity of the Mucorales was reflected in susceptibilities differing at family, genus, and species levels. Amphotericin B was the most active drug, though somewhat less against Rhizopus and Cunninghamella species. Posaconazole was the second most effective antifungal agent but showed reduced activity in Mucor and Cunninghamella strains, while voriconazole lacked in vitro activity for most strains. Genera attributed to the Mucoraceae exhibited a wide range of MICs for posaconazole, itraconazole, and terbinafine and included resistant strains. Cunninghamella also comprised strains resistant to all azoles tested but was fully susceptible to terbinafine. In contrast, the Lichtheimiaceae completely lacked strains with reduced susceptibility for these antifungals. Syncephalastrum species exhibited susceptibility profiles similar to those of the Lichtheimiaceae. Mucor species were more resistant to azoles than Rhizopus species. Species-specific responses were obtained for terbinafine where only Rhizopus arrhizus and Mucor circinelloides were resistant. Complete or vast resistance was observed for 5-fluorocytosine, caspofungin, and micafungin. Intraspecific variability of in vitro susceptibility was found in all genera tested but was especially high in Mucor and Rhizopus for azoles and terbinafine. Accurate molecular identification of etiologic agents is compulsory to predict therapy outcome. For species of critical genera such as Mucor and Rhizopus, exhibiting high intraspecific variation, susceptibility testing before the onset of therapy is recommended.

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Primary Cutaneous Zygomycosis Caused byRhizomucor variabilis:A New Endemic Zygomycosis? A Case Report and Review of 6 Cases Reported from China

Cited by 56 publications

References 5 publications

Rhizomucor variabilis var. regularior and Hormographiella aspergillata Infections in a Leukemic Bone Marrow Transplant Recipient with Refractory Neutropenia

Rhizomucor variabilis var. regularior and Hormographiella aspergillata Infections in a Leukemic Bone Marrow Transplant Recipient with Refractory Neutropenia

Mucormycosis Caused by Unusual Mucormycetes, Non-Rhizopus, -Mucor, and -Lichtheimia Species

Antifungal Susceptibility and Phylogeny of Opportunistic Members of the Order Mucorales

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