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Oral Entomophthoramycosis: a Rare Fungal Infection

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Oral Entomophthoramycosis: a Rare Fungal Infection Oral Entomophthoramycosis: A Rare Fungal Infection KD Shah*, RA Bradoo**, UV Warwantkar***, AA Joshi**** Abstract Entomophthoramycosis is a rare fungal infection which primarily affects the nose and can later spread to involve the paranasal sinuses, nasopharnyx, oropharynx, palate and the cervical region. It usually has a nodular appearance with infiltration of the underlying tissues and no clear demarcation from the surrounding tissues. We present the case of a 28 year old man, who presented with entomophthoramycosis affecting the palate, oropharynx and nasopharnyx with- out involvement of the nose. The diagnosis was confirmed by histopathology, which showed invasive mycotic inflammation, and the fungal culture revealed entomophthoramycosis. The patient was treated with oral Itraconazole (400 mg / day) for a period of one year. He responded very well to this single-drug therapy with clinical resolution of the lesion within 6 months. Further, the patient was kept under a regular follow-up for a period of one year, without any recurrence. This case is reported due to the absence of a nasal lesion of a rare fungal infection which usually originates in the nose. Introduction The disease is essentially chronic, evolving ntomophthoramycosis is a fungal over the course of years. No incubation period E infection which affects individuals with or cases of spontaneous involution are known. an apparently intact immunological status Diagnosis is made by clinical, and occurs primarily in tropical areas. Two histopathological, and mycological 4 zygomycetes belonging to the order examination. Entomophthorales are the aetiologic agents A review of medical literature indicates of subcutaneous entomophthoramycosis, viz., that the condition can be treated with Basodiobolus ranarum (= B. haptosporus) and potassium iodide, amphotericin B, and Conidiobolus coronatus.1,2 ketoconazole. However, the literature Infection with C. Coronatus usually begins provides little information on details of long- in the inferior turbinate, and spreads in the term follow-up and occurrence of relapses. submucosa through the natural ostia to the We present a case of oral paranasal sinus, and to the subcutaneous entomophthoramycosis where a single-drug, tissue of the face (forehead, periorbital region i.e., Itraconazole, has been used for a period and upper lip).3 Infection by B. ranarum of 1 year to achieve complete remission. manifests itself as a subcutaneous Case Report tumefaction located on the trunk, buttocks A 28 year old male, residing in Mumbai and a or proximal portion of the limbs or as an labourer by occupation, presented with chief invasion of the thoracic or abdominal cavities. complaints of intra-oral growth since three months. The growth was insidious in onset and had gradually increased in size over the last three months. He had *Senior Registrar; **Professor and Head; history of odynophagia and dysphagia. On enquiry, ***Registrar; ****Associate Professor, Department he also had a history of change in voice and nasal of ENT, LTMG Hospital, Sion, Mumbai. blockage. 118 Bombay Hospital Journal, Vol. 52, No. 1, 2010 Examination revealed a diffuse inflammatory sub- chronic inflammatory tissue. mucosal lesion involving the soft palate, uvula, both The patient was treated empirically with antibiotics the anterior pillars and extending upto the posterior but he showed no response. A short course of intra- pharyngeal wall [Fig. 1]. On diagnostic nasal venous steroids (dexamethasone) was also given. The endoscopy, the lesion was seen to be involving the lesion showed partial regression; however, it nasopharnyx also. Examination of the larynx, neck increased in size on discontinuing steroid therapy. and the ears was unremarkable. An incisional biopsy was repeated again from the lesion A CT scan of the neck showed a diffuse, sub- on the soft palate. The tissue was sent for KOH mount mucosal, non-enhancing mass involving the and fungal culture in addition to histopathology. This nasopharnyx, the oropharynx and soft palate biopsy was positive for an invasive mycotic bilaterally [Fig. 2]. inflammation and the fungal culture was suggestive A punch biopsy of the lesion was taken from the of entomophthoramycosis. The patient was started soft palate which was suggestive of vascular on oral Itraconazole 200 mg twice a day. proliferative tissue. A nasal endoscopy was performed The patient responded well to this treatment and a biopsy was taken from the nasopharyngeal protocol. All lesions on the soft palate, in the lesion. This tissue was reported to be non-specific oropharynx and in the nasopharnyx had resolved after Fig. 3: Post-treatment clinical photograph of lesion. Fig. 1 : Pre-treatment clinical photograph of lesion. Fig. 4 : Post-treatment CT scan of neck - Axial and Fig. 2 : Pre-treatment CT scan of neck - Axial and Sagittal view (White circle encircling area Sagittal view (White circle encircling lesion). of resolved lesion). Bombay Hospital Journal, Vol. 52, No. 1, 2010 119 6 months of treatment [Fig. 3]. irregular branching hyphae, frequently A CT scan of the neck done at the end of 10 months approaching right angles. These hyphae do showed complete resolution of all the lesions [Fig. 4]. not usually display septa and are surrounded Itraconazole was continued for a period of one by a peculiar eosinophilic mass (the year. The patient is asymptomatic without any clinical Splendore-Hoeppli phenomenon). signs of recurrence, one year after complete resolution of the lesions. The condition is slowly progressive, but seldom life-threatening. The clinical Discussion manifestations result from the expansion of Entomophthoramycosis (conidiobolo- the tumour mass. It initially affects the nose mycosis) is a rare and chronically indolent first and so the disease is also referred to as fungal infection. This localized, subcutaneous rhinoentomophthoramycosis. More advanced zygomycosis is characterized by a painless, cases show involvement of the nasopharynx, woody swelling of the rhinofacial region.5 It oropharynx, palate, cervical region, and even occurs mainly in the tropical rain forests of the mediastinum.1,2 The first mycologically Africa, South and Central America, and proven cases in humans were described South-East Asia. A few cases have been independently and simultaneously in 1965 in reported from India. The fungus lives as a Jamaica7 and Africa.8 Adult males are affected saprophyte in soil humus and on decomposing more than females. Our case differed in the plant matter in moist, warm climates.6 It can fact that though the lesions involved the also parasitize certain insects and frogs. nasopharynx, oropharynx and soft palate, Infection is acquired through inhalation of there were no lesions in the nose. spores, or their introduction into the nasal Clinical and histopathological findings may cavities by soiled hands. Most cases affect men be quite suggestive, but definitive diagnosis with agricultural or outdoor occupations. is only possible by isolating and identifying Lesions by C. coronatus begin as the fungus in appropriate culture media. The infiltration of the nasal mucosa and differential diagnosis should consider submucosa, extending to the adjacent tissues scleroma and tumours of the minor salivary of the paranasal sinuses, nasal dorsum, upper gland. In our case, the diagnosis was based lip, and face. The lesion shows a nodular on clinical and histopathological examination appearance with infiltration of the underlying and confirmed by mycological identification tissues. It is covered by erythematous and of the fungus. fibrotic skin or mucosa, with no clear Treatment of entomophthoramycosis is demarcation from the surrounding tissues. difficult because the diagnosis is usually As a rule, the lesions are firmly attached to established late, but patients often respond the underlying tissue, although the bone is to oral itraconazole (200 to 400 mg/day), spared. The overlying skin and mucosa ketoconazole (200 to 400 mg/day), fluconazole remain intact. Histopathological examination (100-200 mg/day), amphotericin-B, and shows an inflammatory granulomatous cotrimoxazole.9 Itraconazole and fluconazole reaction, with a predominantly mononuclear are both effective and relatively safe.10 infiltrate consisting of lymphocytes, Treatment should be continued for at least 1 histiocytes, and some multinucleated giant month after the lesions have resolved. cells, along with plasma cells and eosinophils. Combination therapy with oral potassium The central portion shows broad, thin-walled, iodide and oral azoles gives rapid and lasting 120 Bombay Hospital Journal, Vol. 52, No. 1, 2010 results. Surgical resection is seldom helpful References and it may hasten the spread of infection. 1. Bandeira V, Lascet IG. Zigomicose, in TALHARI Cryotherapy has been tried with little success. S. & NEVES R.G., ed. Dermatologia tropical. São Relapse is common, even after successful Paulo, Medsi, 1995: 191–202. treatment. 2. Ellis DH. The zygomycetes, in AJELLO L and HAY RJ, Medical Mycology. 9. ed. London, Arnold; We used a single drug, i.e., itraconazole, New York, Oxford University Press, 1998 : in a dose of 400 mg / day for one year to 247-76. achieve complete cure. As our patient 3. Ochoa LF, Duque CS, Velez A. Rhinoento- responded very well to Itraconazole alone, we mophthoramycosis - Report of two cases. J did not need to give potassium iodide. We Laryngol Otol 1996; 110 : 1154-6. continued the drug for 6 months after clinical 4. Andrade ZA, Andrade SG. A entomophthorose resolution
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