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Entomophthoramycosis: An unusual cause of facial disfigurement
Abstract
Entomophthoramycosis is a rare fungal infection of nose, paranasal sinuses and subcutaneous tissues found in tropical and subtropical region. From India very few cases have been reported. Here we report a case of Entomophthoramycosis due to Conidiobolus coronatus from the eastern India who presented with slowly growing rhinofacial swelling and right sided nasal obstruction due to intranasal mass. The case was diagnosed by typical histopathological findings of broad aseptate hyphae with surrounding eosinophilic granular material (Splendore Hoeppli phenomenon) on microscopy of nasal biopsy material and confirmed by PCR assay of DNA and sequencing from biopsy tissue. Treatment with saturated solution of potassium iodide and itraconazole was successful and clinical cure was attained in 8 months.
... Infections in immunocompromised patients are described in the lungs with hemoptysis and fever. Invasions of liver and lymph nodes are described [71][72][73][74]. ...
Purpose of Review
To provide an updated description of the epidemiology of the main dermatological fungal neglected diseases, the molecular taxonomy of their main etiological agents, in addition to the clinical aspects in different hosts, differential diagnoses for clinicians and the best current treatments.
Recent Findings
Prevalence maps of chromblastomycosis and mycetoma were constructed. The new molecular nomenclature of the main pathogens is available, as well as the results of physical therapeutic modalities and new triazoles (voriconazole, posaconazole).
Summary
Although there are great literature reviews to describe the burden of mycetoma and chromoblastomycosis over the last few years, no country has routine surveillance for either of these diseases, and the burden is likely substantially underestimated. An exponential expansion of feline sporotrichosis has been observed in Latin America, representing an important concern for other continents. Jorge Lôbo’s disease (Lacaziosis) and entomophthoromycosis remain marginalized by researchers and public entities. New triazoles must have a democratic access policy in poor nations. Finally, clinical trials are needed to define the best therapeutic strategies, including physical methods and immunomodulatory drugs.
Purpose of review:
to review recent advances in the epidemiology, diagnosis, and treatment of deep fungal infections.
Recent findings:
Mycetoma and chromoblastomycosis are the only deep fungal infections incorporated in the list of neglected tropical diseases. These infections start in the skin but progress to deep tissues if not recognized early. A wide array of fungal pathogens are the causative agents. Molecular methods allow for early and accurate identification of the pathogens, but are unfortunately not available in endemic areas. Treatment options are mostly based upon clinical experience rather than on well-designed clinical trials.
Summary:
Deep fungal infections of the skin and soft tissues are rare conditions of wide world distribution but mostly reported from tropical countries. Urgent need for affordable and easily accessible molecular methods and well-conducted studies to allow for accurate diagnosis and to provide evidence to guide proper therapy are urgently needed.
A case of midfacial swelling in a middle aged woman proved to be caused by a fungus Conidiobolus coronatus has been reported. It is a very rare condition and the knowledge of this fungus affecting the rhinofacial region in human started since 1965 only. These cases pose diagnostic problem. The treatment with the combination of ketoconazole and the saturated solution of potassium iodide was effective for this fungal infection.
Rhinofacial conidiobolomycosis (RFC) is an uncommon subcutaneous fungal infection producing painless swelling with grotesque deformity of the face. Although there are case reports and small case series; there are very few prospective studies evaluating treatment outcome and long-term follow-up.
To evaluate the safety and efficacy of combination of itraconazole (200 mg twice daily) and saturated solution of potassium iodide (SSKI) in patients with RFC.
Ten patients of RFC were studied over a period of 5 years. Diagnosis was confirmed by clinical, histopathological, and microbiological evaluation. Conidiobolus was cultured in four cases and in the rest of the cases, the histopathology was suggestive of RFC. They were treated with itraconazole (200 mg twice daily) and SSKI and followed up for a minimum of 1 year after stopping treatment.
The mean age was 38.7 years and the mean duration of symptoms was 22.4 months. Males were predominantly involved (9 : 1). Seven patients responded to the combination treatment, five had complete resolution and two had good improvement (50-75%); however, in two patients the response was minimal (<25% regression of the swelling) and one patient did not show any improvement after 6 months of treatment.
Combination of itraconazole and SSKI is an effective treatment modality for RFC with relatively faster onset of action, low relapse rates, and minimal adverse effects. It can be considered as first-line treatment in patients with RFC.
© 2015 The International Society of Dermatology.
Entomophthoromycosis due to Conidiobolus coronatus is a granulomatous infection characterized by lesions that originate in the inferior turbinate, spread through ostia and foramina to involve the facial and subcutaneous tissues and paranasal sinuses. The majority of the cases have been described from areas of tropical rainforest in West Africa, agricultural and outdoor workers (aged 20-60 years) being the ones most frequently affected. The fungus is common in soil and decaying vegetation. Infection probably occurs by implantation of the spores of the fungus in nasal mucosa. C. incongruus is a rare agent of the disease, so far known only from two cases with lesions involving the pericardium, mediastinum, lungs, liver, oesophagus and jejunum. C. coronatus is known to cause a clinically similar disease in horses, mules, a dolphin and a chimpanzee. A characteristic histological feature is the presence of thin-walled, broad, often septate hyphae or hyphal fragments with a thick eosinophilic sheath, frequently phagocytosed within giant cells. The fungus is known to produce in vitro several enzymes, e.g., elastase, esterase, collagenase and lipase, which have a possible role in pathogenicity. A concentrated brain heart infusion culture filtrate antigen is useful for immunodiagnosis. Several drugs e.g., potassium iodide, cotrimoxazole, amphotericin B, ketoconazole and itraconazole have been tried with varying success. Investigations on the immunology of disease and the role of proteases and lipases in the pathogenesis of infection is an important area of further research.
In our increasingly immunocompromised populations, zygomycosis is becoming more common. It occurs in two forms. Mucormycosis is an acute, necrotic, rapidly progressive disease of the immunocompromised patient that may lead quickly to death. Entomophthoromycosis is a chronic, slowly progressive subcutaneous disease seen mostly in immunocompetent patients living in tropical climates.
A case of entomophthoromycosis of the nose and paranasal sinus is presented. To our knowledge, this is the youngest patient reported with this infection. Though the clinical picture mimicked a malignancy, histopathology clinched the diagnosis of entomophthoromycosis. The lesions resolved completely with oral potassium iodide.
A nasal granuloma caused by Entomophthora coronata is reported from an 11-year old boy residing on Grand Cayman, an island near Jamaica. This is the first case of phycomycosis reported from Jamaica. It is also the first report of a human infection caused by E. coronata (Summary)
Address correspondence to: Ayan Basu, Department of Infectious Diseases & Advanced Microbiology
Accepted December 27, 2021.
*Address correspondence to:
Ayan Basu, Department of Infectious Diseases & Advanced
Microbiology, School of Tropical Medicine, 108, Chittaranjan
Avenue, Kolkata-700073, West Bengal, India.
E-mail: ayanbasustm@gmail.com
and Entomophthoramycosis. In: Principles and Practice of
Infectious Diseases (Mandell, Douglas, Bennett, eds.). 9th