Am. J. Trop. Med. Hyg., 75(5), 2006, pp. 936-938
Copyright © 2006 by The American Society of Tropical Medicine and Hygiene
MUCOCUTANEOUS ENTOMOPHTHORAMYCOSIS ACQUIRED BY CONJUNCTIVAL INOCULATION OF THE FUNGUS
ACHILÉA L. BITTENCOURT*,
ROBERTO MARBACK, AND
LIVIA M. B. NOSSA
Department of Pathology, and Department of Ophthalmology, School of Medicine, Federal University of Bahia, Salvador, Bahia, Brazil
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ABSTRACT
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Entomophthoramycoses are classified into subcutaneous, mucocutaneous, and primary visceral forms. The mucocutaneous form, also known as rhinoentomophthoramycosis, involves the mucosa and subcutaneous tissues of the nose and is caused by Conidiobolus coronatus (Entomophthora coronata). In this report, we describe the first case of mucocutaneous entomophthoramycosis acquired by introduction of the fungus through the conjunctival mucosa as a consequence of trauma involving contamination with soil. The patient was a 37-year-old man with no other complaints. The lesion was tumoral and extended into the ethmoidal and maxillary sinuses. The histopathologic appearance of the lesion was characteristic of this infection with a granulomatous process rich in eosinophils and with hyphae surrounded by an eosinophilic, periodic acid–Schiff stain–positive halo (Splendore-Hoeppli phenomenon). To the best of our knowledge, this case constitutes the first report of mucocutaneous entomophthoramycosis acquired by ocular contamination.
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INTRODUCTION
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Entomophthoramycoses are classified into subcutaneous, mucocutaneous, and primary visceral forms. Subcutaneous entomophthoramycosis is an indolent chronic infection of the subcutaneous tissues caused by Basidiobolus haptosporus, whereas the mucocutaneous form is caused by Conidiobolus coronatus (Entomophthora coronata) and involves the mucosa and subcutaneous tissues of the nose.1
The mucocutaneous form is also referred to as rhinoento-mophthoramycosis, conidiobolomycosis, or rhino-facial ento-mophthoramycosis, and is more frequently observed in adult males. Entomophthoramycosis occurs in tropical and subtropical areas of Africa, India and Latin America.1 Approximately 35 cases of rhinoentomophthoramycosis have been reported in Brazil.2–12
The disease begins as a tumefaction of the mucosa of the inferior turbinate that spreads to other parts of the nasal cavity, paranasal sinuses, and subcutaneous tissue of the nose and surrounding areas. The infection initially appears as a nasal obstruction followed by diffuse erythema and thickening of the skin of the nose. The skin of the cheek and lips may be infiltrated, which causes monstrous deformations in cases of longer evolution. The transmission mode of C. coronatus is probably by contamination with dust or soil containing spores or by trauma to the nasal mucosa, such as that caused by an insect bite or by the introduction of a fragment of vegetation.13,14 In this report, we present the first case of ocular inoculation of mucocutaneous entomophthoramycosis.
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CASE REPORT
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A 37-year-old man who worked in a gas station in Salvador, Bahia, Brazil came to the Hospital Universitário Professor Edgard Santos with conjunctival irritation and lacrimation in his left eye 12 days after a fall that resulted in his left eye being contaminated with soil. Soon afterwards, a tumoral mass appeared in the region of the left lacrimal sac. The patient had no other complaints and had never used immu-nosuppressive therapy. Ophthalmologic examination showed a solid tumor 2.0 x 1.0 cm in the left lacrimal sac region. The clinical suspicion was a tumor of the lacrimal sac. The nose was normal (Figure 1
). Rhinoscopy showed no abnormalities. Laboratory test results, including blood counts, erythrocyte sedimentation rate, and fasting blood glucose, were all normal. Serologic analysis for human immunodeficiency virus showed negative results. A computed tomographic (CT) scan of the face showed a mass occupying the left ethmoidal and maxillary sinuses. Lesion debulking was performed and during this procedure the lesion was observed to be diffuse, with an extensive erosion of the bone in the orbital floor. The histopathologic diagnosis was mucocutaneous entomoph-thoramycosis. After diagnosis, a biopsy was performed for culture but no fungal growth was observed.
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FIGURE 1. Patient after surgery. The vertical scar corresponds to the area from which the tumor was excised. Note the normal aspect of the nose.
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Histopathologic analysis.
Abscesses of eosinophils surrounded by epithelioid and giant cells associated with diffuse and intense infiltation of eosinophils were seen intermingled with extensive areas of fibrosis. Transverse and cross sections of hyphae surrounded by an eosinophilic, periodic acid–Schiff –positive halo (Splendore-Hoeppli phenomenon) were found throughout the inflammatory process (Figure 2). The Grocott method provided good visualization of the walls of the hyphae, which were thin-walled, broad, and rarely septated (Figure 3). Treatment with potassium iodide was instituted (40 mg/kg of body weight/day). After three months of treatment, the patient improved and stopped taking the drug. Twenty-six months later, a rhinoscopy and a CT scan failed to demonstrate any evidence of the disease. The patient has been followed-up for an additional four years and is free of the disease.
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FIGURE 2. Patient biopsy specimen showing transverse and cross sections of hyphae surrounded by an eosinophilic and periodic acid–Schiff–positive halo (Splendore-Hoeppli phenomenon). (hematoxy-lin and eosin stained, original magnification x 160). This figure appears in color at www.ajtmh.org.
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FIGURE 3. Patient biopsy specimen showing oblique and cross sections of broad and thin-walled hyphae, occasionally septated (Grocott stained, original magnification x 200). Inset, A septated hypha. (Grocott stained, original magnification x 640). This figure appears in color at www.ajtmh.org.
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DISCUSSION
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In addition to rhinoentomophthoramycosis, another tropical infection that begins in the nasal mucosa is rhinosporidi-osis, a condition that was previously considered a deep mycosis. Rhinosporidiosis is inoculated through the nasal or ocular mucosae, whereas rhinoentomophthoramycosis is considered to be the result of nasal inoculation of the infective agent.1 However, the two infections have different histo-pathologic patterns. The ocular form of rhinosporidiosis occurs predominantly in arid regions and dust is considered the vehicle of infection.13 In the present case, the history and the clinical presentation of the disease point to ocular penetration of the fungus during accidental introduction of soil into the conjunctival mucosa. Conidiobolus coronatus has been isolated from soil and decaying vegetation in different latitudes; however, cases are more common in tropical and subtropical areas.13 Since no cutaneous lesion or infiltration of the nasal mucosa was observed, the infection probably occurred through the conjunctival mucosa.
As occurred in the present case, the diagnosis of infection by Conidiobolus is generally based on histologic examination because cultures for the causative organism are negative in more than 85% of cases.15 If one considers the predominance of eosinophils in the infiltrate and the exuberant Hoeppli-Splendore phenomenon around the hyphae, the histopathology of this case is characteristic of entomophthoramycosis and different from that of other subcutaneous mycoses.1 Although the inflammatory reaction is similar in all forms of entomophthoramycosis, fibrosis is much more accentuated in the mucocutaneous than in the subcutaneous form of the disease,1 as seen in the present case.
Many antifungal drugs, such as potassium iodide, ampho-tericin B, imidazole derivatives (fluconazole, ketoconazole, and itraconazole), or a combination of two of these drugs,2,4,7–12,16 have been used for the treatment of rinoento-mophthoramycoses. In the present case, a good response was obtained after surgery and subsequent treatment with potassium iodide. To the best of our knowledge, this case constitutes the first report of mucocutaneous entomophthoramyco-sis acquired by conjunctival inoculation.
Received May 16, 2006.
Accepted for publication June 21, 2006.
Acknowledgment: The American Committee on Clinical Tropical Medicine and Travellers’ Health (ACCTMTH) assisted with publication expenses.
* Address correspondence to Achiléa L. Bittencourt, Serviço de Anatomia Patológica, Hospital Universitário Professor Edgard San-tos, Rua Dr. Augusto, Viana s/n° Canela, CEP 40.110-060, Salvador, Bahia, Brazil. E-mail: achilea{at}uol.com.br 
Authors’ addresses: Achiléa L. Bittencourt and Livia M. B. Nossa, Department of Pathology, School of Medicine, Federal University of Bahia, Salvador, Bahia, Brazil. Roberto Marback, Department of Ophthalmology, School of Medicine, Federal University of Bahia, Salvador, Bahia, Brazil.
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ABSTRACT
INTRODUCTION
