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LETTER TO EDITOR  
Year : 2014  |  Volume : 57  |  Issue : 3  |  Page : 519-521
Isolated nasal chromoblastomycosis


1 Department of ENT, AIIMS, New Delhi, India
2 Department of Pathology, AIIMS, New Delhi, India

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Date of Web Publication14-Aug-2014
 

How to cite this article:
Shresta D, Kumar R, Durgapal P, Singh CA. Isolated nasal chromoblastomycosis. Indian J Pathol Microbiol 2014;57:519-21

How to cite this URL:
Shresta D, Kumar R, Durgapal P, Singh CA. Isolated nasal chromoblastomycosis. Indian J Pathol Microbiol [serial online] 2014 [cited 2022 May 23];57:519-21. Available from: https://www.ijpmonline.org/text.asp?2014/57/3/519/138819


Editor,

Chromoblastomycosis is a slow growing fungal infection of skin and subcutaneous tissues often resulting in disfigurement of the affected body parts. [1] It is mainly a disease of tropical and subtropical regions such as India, Brazil, Mexico, Madagascar, and Venezuela. [2] Several species are accepted to cause chromoblastomycosis such as Fonsacea pedrosoi, Phialophora verrucosa, Cladophialophora carrionii, Fonsacea compacta, Rhinocladiella aquaspersa, and Exophiala species. These are saprophytes normally found in soil and plants. F. pedrosoi is the most common organism, which has been isolated from the skin lesions. [3] Chromoblastomycosis is commonly seen in population working outdoors such as farmers and forest workers. The common sources are wood splinters, wood pulp, cactus spines, plant debris, fence posts, tree stumps, and soil and animal feces. The mode of transmission suggested is either by traumatic inoculation by wood splinters or inoculation of soil or vegetable matter contaminated with fungus into the exposed body parts mainly extremities. However, unusual body parts such as genitalia and nose has been described in the literature as sites of inoculation. [4],[5]

A 26-year-old female hailing from a village from Mathura district of Uttar Pradesh in Northern India presented with left sided nasal obstruction and mucopurulent discharge for last 2 years. The patient was initially seen at the local hospital (ESI dispensary) and diagnosed to have left sided nasal mass. However, no biopsy was done, and the patient was directly referred to our center for further management. The nasal obstruction was associated with postnasal drip and pain over nasal dorsum. There was no swelling of the nasal dorsum or upper lip suggestive of subcutaneous tissue involvement. There was no history of nasal trauma or epistaxis. The patient is a housewife and used to collect wood from the nearby forest for daily cooking purposes. Anterior rhinoscopy revealed blackish necrotic mass in the left nasal cavity attached to inferior turbinate. It was nontender and bony hard on probing. Nasal mucosa, nasal septum, and middle turbinate were normal. Rest of the ENT examination was also normal. Noncontrast computed tomography of the nose and paranasal sinuses revealed well-defined lobulated hyperdense calcified mass measuring approximately 2.7 cm × 1.9 cm in left nasal cavity, arising from concha of inferior turbinate [Figure 1]a and b. Based on the radiological findings, patient was taken up for endoscopic excision of nasal mass under local anesthesia. The mass was completely excised along with part of the inferior turbinate. Histopathology (GMS) showed inflammatory exudate and numerous golden brown oval septate "copper penny" bodies suggestive of chromoblastomycosis. These features were consistent with chromoblastomycosis [Figure 2]. No fungal elements were appreciable in the 10% KOH preparation. Sabouraud's dextrose agar media was used to culture the species (incubation period 3 days to 3 weeks; at temperature 25°C and 37°C; 2 incubation plates), but no growth was obtained after 2-3 weeks. Therefore, based on the histopathology, patient was started on oral tablet itraconazole 200 mg twice a day for 3 months. Patient is on regular follow-up for last 10 months without any evidence of disease recurrence or appearance of any skin lesions.
Figure 1: (a and b) Noncontrast computed tomography of paranasal sinuses (coronal cuts) shows lobulated hyperdense calcified mass arising from the inferior turbinate of the left nasal cavity

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Figure 2: H and E stain shows "copper pennies" pathognomonic for chromoblastomycosis (×20)

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Isolated nasal chromoblastomycosis is a rare condition. Clinical presentation is in the form of skin lesions, which can be nodular, plaque-like, verrucous, cicatricial, or tumorous. The most common sites involved by chromoblastomycosis are legs, arms, and buttocks. [6] Sporadic reports mentioned in the literature include lesions on face, ear, and breast. Other unusual sites reported are penile shaft, vulva, and ala of nose. [2] In our case, the disease was confined only to inferior turbinate of the nasal cavity. Nasal form of chromoblastomycosis may present as nasal obstruction, nasal discharge, or epistaxis. It is difficult to diagnose clinically because of its rarity in the nasal cavity. Grossly, lesions are blackish, nodular or warty. The differential diagnosis of chromoblastomycosis includes verrucous variants of tuberculosis, sporotrichosis and leishmaniasis. Symmers in 1960 had reported a case of nasal chromoblastomycosis, which was initially diagnosed as nasal rhinosporiodosis. [4],[7]

Examination by direct microscopy is a rapid and easy to perform and confirms the mycological diagnosis. The scrapings should be taken from "black dots," which represents the transepidermal elimination of fungai. Direct examination of the scrapings in 10% KOH demonstrates thick-walled, multiseptate, brown sclerotic cells, in smear or tissue sections, are pathognomonic for chromoblastomycosis. [6] These sclerotic bodies are also known as "copper-pennies," "medlar bodies" or muriform cells. [8] Although, KOH mount of the scrapings in our case was negative for copper pennies, they were well seen in H and E stained tissue sections. Histologically, typical lichenoid dermal granulomatous infiltrate can be seen. Inflammatory infiltrate is characterized by multinucleated cells, fibrosis, acanthosis, papillomatosis, hyperkeratosis, pseudoepitheliomatous hyperplasia and typical fumagoid cells. [6] Cutaneous lesions shows hyperkeratosis, keratolytic microabscesses and pseudoepitheliomatous hyperplasia. Culture is done for species identification, but is a slow process and can be inconclusive at times. Culture should be observed for a period of 3 weeks to 30 days as fungal agents are typically slow growing and culture may be inconclusive due to poor morphological differentiation. Polymerase chain reaction is also useful for species identification and can supplement culture when culture is inconclusive. [9],[10] It is a rapid and sensitive investigation and is widely available in the majority of centers. ELISA is another investigative tool, which is less commonly used but useful in monitoring response to long-term treatment. [8]

Treatment options for chromoblastomycosis include antifungal therapy, surgery and physical destructive procedures. Antifungals are recommended in high doses. First-line of antifungal treatment includes itraconazole (200-400 mg daily) and terbinafine (500-1000 mg daily). Itraconazole and terbinafine has shown synergistic activity and is recommended. Furthermore, the pulse therapy of itraconazole (200 mg twice daily for 1 week every month) has been shown to be equally efficacious as daily itraconazole. Pulse therapy helps to bring down the cumulative dose, long-term side-effects and total cost of the therapy. For skin lesions, antifungal are recommended for the duration of 6-12 months. Older drugs like ketoconazole, flucytosine has been out of favor owing to potential side-effects on long-term treatment. Ketoconazole has reported to cause hepatotoxicity on the long-term use. New drugs like posaconazole and voriconazole are available as an alternative option. These drugs are proven to efficacious in deep cutaneous mycosis and in cases where standard antifungal treatment has failed. [8] Although, drug therapy is the modality of choice for treating chromoblastomycosis long duration of treatment, cost and long-term compliance is a problem.

Surgery for localized lesions is very effective, as was done in our case. However, postoperative anti-fungal therapy should be continued to prevent recurrence. Physical destructive procedures including cryotherapy and thermotherapy have been described for extensive skin lesions in combination with drug therapy. These procedures were reported to be successful. The advantages are low cost, easy to administer and a good alternative for those patients who are unable to take systemic medication. Chromoblastomycosis is known for its chronicity and frequent recurrences after the initial treatment; therefore, long duration of treatment and close follow-up has been recommended. [8]

Isolated nasal chromoblastomycosis is very rare, but should be considered in the differential diagnosis in a case of isolated nasal mass. Complete surgical excision either endoscopically or with an open approach is recommended. Postsurgical long-term antifungal therapy should be prescribed to prevent recurrence.

 
   References Top

1.
Queiroz-Telles F, Esterre P, Perez-Blanco M, Vitale RG, Salgado CG, Bonifaz A. Chromoblastomycosis: An overview of clinical manifestations, diagnosis and treatment. Med Mycol 2009;47:3-15.  Back to cited text no. 1
    
2.
Rajendran C, Ramesh V, Misra RS, Kandhari S, Upreti HB, Datta KK. Chromoblastomycosis in India. Int J Dermatol 1997;36:29-33.  Back to cited text no. 2
    
3.
Lupi O, Tyring SK, McGinnis MR. Tropical dermatology: Fungal tropical diseases. J Am Acad Dermatol 2005;53:931-51.  Back to cited text no. 3
    
4.
Zaror L, Fischman O, Pereira CA, Felipe RG, Gregório LC, Castelo A. A case of primary nasal chromoblastomycosis. Mykosen 1987;30:468-71.  Back to cited text no. 4
    
5.
Nakamura T, Grant JA, Threlkeld R, Wible L. Primary chromoblastomycosis of the nasal septum. Am J Clin Pathol 1972;58:365-70.  Back to cited text no. 5
    
6.
López Martínez R, Méndez Tovar LJ. Chromoblastomycosis. Clin Dermatol 2007;25:188-94.  Back to cited text no. 6
    
7.
Symmers WS. Chromoblastomycosis simulating rhinosporidiosis in a patient from Ceylon. J Clin Pathol 1960;13:287-90.  Back to cited text no. 7
    
8.
Ameen M. Managing chromoblastomycosis. Trop Doct 2010;40:65-7.  Back to cited text no. 8
    
9.
de Andrade TS, Cury AE, de Castro LG, Hirata MH, Hirata RD. Rapid identification of Fonsecaea by duplex polymerase chain reaction in isolates from patients with chromoblastomycosis. Diagn Microbiol Infect Dis 2007;57:267-72.  Back to cited text no. 9
    
10.
Abliz P, Fukushima K, Takizawa K, Nishimura K. Specific oligonucleotide primers for identification of Cladophialophora carrionii, a causative agent of chromoblastomycosis. J Clin Microbiol 2004;42:404-7.  Back to cited text no. 10
    

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Correspondence Address:
Rajeev Kumar
Department of ENT, AIIMS, New Delhi - 110029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.138819

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