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Classification of Rhinoentomophthoromycosis into Atypical, Early, Intermediate, and Late Disease: A Proposal.

Blumentrath CG, Grobusch MP, Matsiégui PB, Pahlke F, Zoleko-Manego R, Nzenze-Aféne S, Mabicka B, Sanguinetti M, Kremsner PG, Schaumburg F - PLoS Negl Trop Dis (2015)

Bottom Line: The search yielded 8,333 results including 198 cases from 117 papers; of these, 145 met our inclusion criteria and were included in the final analysis.Cure rates were clearly associated with stage of disease and were 57%, 100%, 82%, and 43% in atypical, early, intermediate, and late disease, respectively.We suggest a clinical staging system that underlines the benefit of early case detection and may guide the duration of antifungal treatment.

View Article: PubMed Central - PubMed

Affiliation: Institut für Tropenmedizin, Eberhard Karls Universität Tübingen and Deutsches Zentrum für Infektionsforschung, Tübingen, Germany; Centre de Recherches Médicales de Lambaréné (CERMEL), Albert Schweitzer Hospital Lambaréné, Lambaréné, Gabon; Centre de Recherche Médicale de la Ngounié, Fougamou, Gabon.

ABSTRACT

Background: Rhinoentomophthoromycosis, or rhino-facial conidiobolomycosis, is a rare, grossly disfiguring disease due to an infection with entomophthoralean fungi. We report a case of rhinoentomophthoromycosis from Gabon and suggest a staging system, which provides information on the prognosis and duration of antifungal therapy.

Methods: We present a case of rhinoentomophthoromycosis including the histopathology, mycology, and course of disease. For the suggested staging system, all cases on confirmed rhinoentomophthoromycosis published in the literature without language restriction were eligible. Exclusion criteria were missing data on (i) duration of disease before correct diagnosis, (ii) outcome, and (iii) confirmation of entomophthoralean fungus infection by histopathology and/or mycology. We classified cases into atypical (orbital cellulitis, severe pain, fever, dissemination), early, intermediate, and late disease based on the duration of symptoms before diagnosis. The outcome was evaluated for each stage of disease.

Findings: The literature search of the Medpilot database was conducted on January 13, 2014, (updated on January 18, 2015). The search yielded 8,333 results including 198 cases from 117 papers; of these, 145 met our inclusion criteria and were included in the final analysis. Median duration of treatment was 4, 3, 4, and 5 months in atypical, early, intermediate, and late disease, respectively. Cure rates were clearly associated with stage of disease and were 57%, 100%, 82%, and 43% in atypical, early, intermediate, and late disease, respectively.

Conclusion: We suggest a clinical staging system that underlines the benefit of early case detection and may guide the duration of antifungal treatment. The scientific value of this classification is its capacity to structure and harmonize the clinical and research approach towards rhinoentomophthoromycosis.

No MeSH data available.


Related in: MedlinePlus

Flow chart of the selection of published cases on rhinoentomophthoromycosis.All cases assigned to staging analysis (n = 145) were included in the statistical analysis.
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pntd.0003984.g005: Flow chart of the selection of published cases on rhinoentomophthoromycosis.All cases assigned to staging analysis (n = 145) were included in the statistical analysis.

Mentions: Search terms yielded 8,333 matches. By screening the reference lists of these manuscripts, we identified an additional 38 articles (Fig 5). In total, 198 cases reported in 117 articles met our case definition. Out of these cases, 53 cases were excluded due to missing essential information (Fig 5). A total of 145 cases provided data on duration of disease before diagnosis, outcome, and mycological or histopathological proof (Fig 5). In total, 145 cases were classified into early (n = 9), intermediate (n = 62), late (n = 60), and atypical (n = 14) disease (Table 1 and Fig 5). Of the 131 patients with atypical rhinoentomophthoromycosis, 95% (n = 124) were previously healthy. The concomitant diseases of the other seven patients were: helminthic infection (n = 3), HIV/tuberculosis coinfection (n = 1), tuberculosis (n = 1), diabetes mellitus (n = 1), and hypertension (n = 1). Atypical rhinoentomophthoromycosis presented as orbital cellulitis (100%, n = 14) associated with fever (71%, n = 10), severe sinus/orbital pain or headache (79%, n = 11), loss of vision (21% n = 3), and eye-movement restrictions (36% n = 5). Atypical disease patients had highly elevated infectious parameters (e.g., leukocytes, C-reactive protein, and erythrocyte sedimentation rate), which are uncommon in other stages. Systemic dissemination (i.e., lung and CNS) and comorbidities were more frequently found in atypical disease compared to other stages (86% vs. 2%, p<0.001 and 50% vs. 5%, p<0.001, respectively).


Classification of Rhinoentomophthoromycosis into Atypical, Early, Intermediate, and Late Disease: A Proposal.

Blumentrath CG, Grobusch MP, Matsiégui PB, Pahlke F, Zoleko-Manego R, Nzenze-Aféne S, Mabicka B, Sanguinetti M, Kremsner PG, Schaumburg F - PLoS Negl Trop Dis (2015)

Flow chart of the selection of published cases on rhinoentomophthoromycosis.All cases assigned to staging analysis (n = 145) were included in the statistical analysis.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4591341&req=5

pntd.0003984.g005: Flow chart of the selection of published cases on rhinoentomophthoromycosis.All cases assigned to staging analysis (n = 145) were included in the statistical analysis.
Mentions: Search terms yielded 8,333 matches. By screening the reference lists of these manuscripts, we identified an additional 38 articles (Fig 5). In total, 198 cases reported in 117 articles met our case definition. Out of these cases, 53 cases were excluded due to missing essential information (Fig 5). A total of 145 cases provided data on duration of disease before diagnosis, outcome, and mycological or histopathological proof (Fig 5). In total, 145 cases were classified into early (n = 9), intermediate (n = 62), late (n = 60), and atypical (n = 14) disease (Table 1 and Fig 5). Of the 131 patients with atypical rhinoentomophthoromycosis, 95% (n = 124) were previously healthy. The concomitant diseases of the other seven patients were: helminthic infection (n = 3), HIV/tuberculosis coinfection (n = 1), tuberculosis (n = 1), diabetes mellitus (n = 1), and hypertension (n = 1). Atypical rhinoentomophthoromycosis presented as orbital cellulitis (100%, n = 14) associated with fever (71%, n = 10), severe sinus/orbital pain or headache (79%, n = 11), loss of vision (21% n = 3), and eye-movement restrictions (36% n = 5). Atypical disease patients had highly elevated infectious parameters (e.g., leukocytes, C-reactive protein, and erythrocyte sedimentation rate), which are uncommon in other stages. Systemic dissemination (i.e., lung and CNS) and comorbidities were more frequently found in atypical disease compared to other stages (86% vs. 2%, p<0.001 and 50% vs. 5%, p<0.001, respectively).

Bottom Line: The search yielded 8,333 results including 198 cases from 117 papers; of these, 145 met our inclusion criteria and were included in the final analysis.Cure rates were clearly associated with stage of disease and were 57%, 100%, 82%, and 43% in atypical, early, intermediate, and late disease, respectively.We suggest a clinical staging system that underlines the benefit of early case detection and may guide the duration of antifungal treatment.

View Article: PubMed Central - PubMed

Affiliation: Institut für Tropenmedizin, Eberhard Karls Universität Tübingen and Deutsches Zentrum für Infektionsforschung, Tübingen, Germany; Centre de Recherches Médicales de Lambaréné (CERMEL), Albert Schweitzer Hospital Lambaréné, Lambaréné, Gabon; Centre de Recherche Médicale de la Ngounié, Fougamou, Gabon.

ABSTRACT

Background: Rhinoentomophthoromycosis, or rhino-facial conidiobolomycosis, is a rare, grossly disfiguring disease due to an infection with entomophthoralean fungi. We report a case of rhinoentomophthoromycosis from Gabon and suggest a staging system, which provides information on the prognosis and duration of antifungal therapy.

Methods: We present a case of rhinoentomophthoromycosis including the histopathology, mycology, and course of disease. For the suggested staging system, all cases on confirmed rhinoentomophthoromycosis published in the literature without language restriction were eligible. Exclusion criteria were missing data on (i) duration of disease before correct diagnosis, (ii) outcome, and (iii) confirmation of entomophthoralean fungus infection by histopathology and/or mycology. We classified cases into atypical (orbital cellulitis, severe pain, fever, dissemination), early, intermediate, and late disease based on the duration of symptoms before diagnosis. The outcome was evaluated for each stage of disease.

Findings: The literature search of the Medpilot database was conducted on January 13, 2014, (updated on January 18, 2015). The search yielded 8,333 results including 198 cases from 117 papers; of these, 145 met our inclusion criteria and were included in the final analysis. Median duration of treatment was 4, 3, 4, and 5 months in atypical, early, intermediate, and late disease, respectively. Cure rates were clearly associated with stage of disease and were 57%, 100%, 82%, and 43% in atypical, early, intermediate, and late disease, respectively.

Conclusion: We suggest a clinical staging system that underlines the benefit of early case detection and may guide the duration of antifungal treatment. The scientific value of this classification is its capacity to structure and harmonize the clinical and research approach towards rhinoentomophthoromycosis.

No MeSH data available.


Related in: MedlinePlus