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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

Portrait of the patient before, during and after treatment.(A) The patient is shown as a healthy young adult. (B) Before treatment (month 0). (C) After treatment with fluconazole and terbinafine (month 18). (D) After 14 months without therapy (month 32).
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pntd.0003984.g002: Portrait of the patient before, during and after treatment.(A) The patient is shown as a healthy young adult. (B) Before treatment (month 0). (C) After treatment with fluconazole and terbinafine (month 18). (D) After 14 months without therapy (month 32).

Mentions: Humans suffering from rhinoentomophthoromycosis get infected by the attachment of conidia of C. coronatus to nasal/sinusoidal mucosa. Initially, the disease presents like sinusitis [2]. A nodule at the nostrils indicates expansion into the subcutaneous fat (Fig 1) [12,13]. The infection spreads within the subcutaneous fatty layers of the nasal bridge, eyelids, cheek, and upper lip [2,13]. Swellings are firm, indolent, and, initially, often reddened and warm, while later they are often itchy [2,12–15]. Mucosal swellings rarely affect laryngeal structures or cause dyspnoea [2]. Grotesque facial swelling is characteristic of late disease [2]. Ulcerations of skin or mucosa are uncommon; skin-adherent structures, eye motility, and vision usually remain unaffected; and bones, vessels, muscle, and lymph nodes are rarely involved. The course of the disease is usually benign (Figs 1 and 2) [2,14,15].


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)

Portrait of the patient before, during and after treatment.(A) The patient is shown as a healthy young adult. (B) Before treatment (month 0). (C) After treatment with fluconazole and terbinafine (month 18). (D) After 14 months without therapy (month 32).
© Copyright Policy
Related In: Results  -  Collection

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

pntd.0003984.g002: Portrait of the patient before, during and after treatment.(A) The patient is shown as a healthy young adult. (B) Before treatment (month 0). (C) After treatment with fluconazole and terbinafine (month 18). (D) After 14 months without therapy (month 32).
Mentions: Humans suffering from rhinoentomophthoromycosis get infected by the attachment of conidia of C. coronatus to nasal/sinusoidal mucosa. Initially, the disease presents like sinusitis [2]. A nodule at the nostrils indicates expansion into the subcutaneous fat (Fig 1) [12,13]. The infection spreads within the subcutaneous fatty layers of the nasal bridge, eyelids, cheek, and upper lip [2,13]. Swellings are firm, indolent, and, initially, often reddened and warm, while later they are often itchy [2,12–15]. Mucosal swellings rarely affect laryngeal structures or cause dyspnoea [2]. Grotesque facial swelling is characteristic of late disease [2]. Ulcerations of skin or mucosa are uncommon; skin-adherent structures, eye motility, and vision usually remain unaffected; and bones, vessels, muscle, and lymph nodes are rarely involved. The course of the disease is usually benign (Figs 1 and 2) [2,14,15].

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