hidden under a cauliflower like growth a case of cutaneous chromoblastomycosis and response to combination therapy

Available online at www.sciencedirect.com www.jdds.org ScienceDirect Journal of Dermatology & Dermatologic Surgery xxx (2016) xxx–xxx Hidden under a cauliflower-like growth: A case of cutaneous chromoblastomycosis and response to combination therapy Mohammad Kamrul Ahsan a, Khalid Mohammed Al Attas a,⇑, Mohammed A Buraik a,b, Ali Mohammed Al-Sheikh a, Sultan M Bajawi a a Department of Dermatology, King Fahad Central Hospital, Jizan, Saudi Arabia b Department of Pathology, King Fahad Central Hospital, Jizan, Saudi Arabia Received 28 September 2016; accepted 12 November 2016 Abstract Chromoblastomycosis is a rare, chronic fungal infection of skin and subcutaneous tissue It is caused by several pigmented fungi commonly seen in tropical and sub-tropical regions Here, we report a case of chronic cutaneous chromoblastomycosis in a middle aged man from the southern part of Jizan, Saudi Arabia, who presented to our derma clinic with verrucous cauliflower like growth on the right hand Later on, histopathological study showed pathognomonic characteristic brown colored spores (copper pennies or medlar bodies) of the fungus within dermal abscess The patient was cured after a month combination use of itraconazole and cryotherapy Review of literature showed this is the first cutaneous chromoblastomycosis reported from Saudi Arabia Ó 2016 The Authors Production and hosting by Elsevier B.V on behalf of King Saud University This is an open access article under the CC BYNC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/) Keywords: Chromoblastomycosis; Copper pennies; Medlar bodies; Itraconazole; Cryotherapy Introduction Chromoblastomycosis is a chronic deep skin mycosis caused by pigmented fungi that are implanted into skin from the environment The most common etiologic agents are Fonsecaea pedrosoi, Phialophora verrucosa and Cladophialophora carrioni (Ameen, 2009) All have low virulence, produce similar clinical manifestations and can be isolated from soil and plant debris The pigmented, socalled, dematiaceous fungus (Fonsecaea pedrosoi) is the etiologic agent in 90–96% of cases (Bonifaz et al., 2001) The infection occurs sporadically in South America, Caribbean region, Madagascar, Australia, and Japan It may also occur as an imported infection outside the usual endemic areas The disease is most frequent in male rural workers The infection usually follows implantation through tissue injury (Rubin et al., 1991) Following inoculation by skin trauma, local infection with slowly growing, raised scaly plaques or warty cauliflower-like lesions develop (Minotto et al., 2001) The presented case displayed features of warty cauliflower like tumor on the right hand Case report ⇑ Corresponding author E-mail address: alattas101khalid@yahoo.com (K.M Al Attas) Peer review under responsibility of King Saud University Production and hosting by Elsevier A 68 year old Saudi man presented to our department with single well defined verrucous firm to hard longitudinal lesion on the right hand that had started 25 years back According to the patient, the lesion had enlarged slowly and he remembered no traumatic skin lesion except minor scratches from gardening many years back It began as a http://dx.doi.org/10.1016/j.jdds.2016.11.001 2352-2410/Ó 2016 The Authors Production and hosting by Elsevier B.V on behalf of King Saud University This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/) Please cite this article in press as: Ahsan, M.K et al., Hidden under a cauliflower-like growth: A case of cutaneous chromoblastomycosis and response to combination therapy, Journal of Dermatology & Dermatologic Surgery (2016), http://dx.doi.org/10.1016/j.jdds.2016.11.001 M.K Ahsan et al / Journal of Dermatology & Dermatologic Surgery xxx (2016) xxx–xxx small, round nodule, whitish-brown in color and hard in consistency Physical examination revealed erythematous, hyperkeratotic, contiguous, indurated, longitudinal plaques with warty, crusted areas with black dots Adjacent to these plaques was a larger whitish area with scar-like appearance along with few scattered nodular lesions (Fig 1) The regional lymph nodes were not palpable Laboratory examination disclosed low hemoglobin (Hb 11 g/dl; normal 12–16) Liver function, renal function, blood sugar and erythrocyte sedimentation rate were normal Serology for sexually transmitted diseases including syphilis, human immunodeficiency virus, hepatitis B, C was negative Microbiological study was inconclusive and chest X-ray was normal Biopsy specimens taken from the lesion showed pseudoepitheliomatous epidermal hyperplasia and suppurative granulomatous inflammation in the dermis There were epithelioid cells and foreign body giant cells, some with engulfed brown color pigmented sclerotic bodies (copper pennies or medlar bodies) Morphologically, round bodies are sized about 7–10 microns; compatible with chromoblastomycosis (Figs and 3) Based on these findings, we established a definitive diagnosis of cutaneous chromoblastomycosis The patient was started on itraconazole 100 mg twice daily along with cryotherapy sessions separated by an interval of month Initially, topical antibiotic (fusidic acid) was also used to treat the secondary bacterial infection Treatment with itraconazole was maintained for months The lesions healed leaving residual scarring and depigmentation in some areas (Fig 4) No recurrence was observed during 12 month follow-up peroid Discussion Figure Showing thick walled brown color, pigmented round fungal spores(medlar bodies or copper pennies) within giant cells of granuloma and mixed suppurative and granulomatous dermal inflammation Figure Showing budding of fungal elements in GMS staining Chromoblastomycosis is endemic in tropical and subtropical regions Cases from Saudi Arabia are rare and Figure Showing cured lesions with some depigmentation and mild residual fibrosis Figure Showing firm, hyperkeratotic, contiguous plaques with black dots and crusted areas on a erythematous base Adjacent to these plaques, larger whitish areas with scar-like appearance along with few scattered nodular lesions are seen often related to work with tropical woods and fields (alHedaithy et al., 1988) Our patient remembered no traumatic skin lesion except minor scratches from gardening many years back He had no travel history outside Saudi Arabia The lesion started 25 years back as a small, round Please cite this article in press as: Ahsan, M.K et al., Hidden under a cauliflower-like growth: A case of cutaneous chromoblastomycosis and response to combination therapy, Journal of Dermatology & Dermatologic Surgery (2016), http://dx.doi.org/10.1016/j.jdds.2016.11.001 M.K Ahsan et al / Journal of Dermatology & Dermatologic Surgery xxx (2016) xxx–xxx nodule, whitish-brown in color and hard in consistency In the literature (Bonifaz et al., 2001; Ezughah et al., 2003) the lower extremities are typically involved, but in our patient the lesions was on the hand Histopathology is pathognomonic for chromoblastomycosis, but the identification of the causative fungal species can only be obtained by culture Delay in diagnosis of chromoblastomycosis up to several years is not unusual, as in our patient Chromoblastomycosis can be confounded with squamous cell carcinoma (SCC), especially if there is clinical absence of pigmentation and histopathology shows pseudoepitheliomatous hyperplasia of the epidermis (Minotto et al., 2001; Bonifaz et al., 2001) In our case, though there was pseudoepitheliomatous hyperplasia and history of long standing infection, no change to SCC was found It is difficult to treat chromoblastomycosis because of differences in antifungal sensitivity patterns and responses among the species isolated Furthermore, the disease itself is refractory in nature particularly in more serious clinical forms The different treatment modalities available have not been compared in clinical settings Recurrence is common, so the recommendation is for long term treatments ranging from to 18 months (Queiroz-Telles et al., 2008) Common complications include secondary bacterial infection with lymphadenitis and less frequently, the development of SCC in lesions that have been present for a long time (Minotto et al., 2001) The only complication in our case was secondary impetiginization, which was resolved with topical antibiotic The best systemic antifungals seem to be itraconazole and terbinafine because of their spectrum of action and safety in long term regimens (Ameen, 2009) However, tissue fibrosis secondary to long term infection can reduce drug tissue level In our patient, itraconazole twice daily was used for months Antifungal combined with cryotherapy has emerged as a possible treatment option in recent years Cryotherapy is associated with the best outcome of all the physical modalities with a cure rate of 40.9% when used as monotherapy (Castro et al., 2003) It appears to be more useful when combined with systemic antifungal to treat long standing cases Surgery may be the best choice in the early stage of disease but not in late and advanced case In our patient, cycles of cryotherapy (one cycle/month) combined with itraconazole (100 mg twice daily) for months achieved cure and caused minimal local effects like hypopigmentation and mild residual fibrosis Recent publications also support the use of combined therapies that is not amenable to surgical treatment Finally, a higher grade of clinical suspicion and a wider differential diagnosis of the pigmented skin tumors would have been needed to initiate proper and adequate treatment Early starting of an appropriate combined treatments are crucial for management of chromoblastomycosis References al-Hedaithy, S.S., Jamjoom, Z.A., Saeed, E.S., 1988 Cerebral phaeohyphomycosis caused by Fonsecaea pedrosoi in Saudi Arabia APMIS Suppl 3, 94–100 Ameen, M., 2009 Chromoblastomycosis: clinical presentation and management Clin Exp Dermatol 849–54 Bonifaz, A., Carrasco-Gerard, E., Saul, A., 2001 Chromoblastomycosis: clinical and mycologic experience of 51 cases Mycoses 44 (1–2), 1–7 [PubMed] Castro, L.G., Pimentel, E.R., Lacaz, C.S., 2003 Treatment of chromomycosis by cryosurgery with liquid nitrogen: 15 years’ experience Int J Dermatol 42, 408–412 Ezughah, F.I., Orpin, S., Finch, T.M., Colloby, P.S., 2003 Chromoblastomycosis imported from Malta Clin Exp Dermatol 28 (5), 486–487 (PubMed) Minotto, D., Varejao Bernarti, C.D., Mallmann, L.F., Albano Edelweiss, M.I., Scrofemeker, M.A., 2001 Chromoblastomycosis: a review of 100 cases in the state of Rio Grande Sul, Brazil J Am Acad Dermatol 44, 585–592 Queiroz-Telles, F., Esterre, P., Perez-Blanco, M., Bonifaz, A., 2008 Chromoblastomycosis: an overview of clinical manifestations, diagnosis and treatment Med Mycol 47, 3–15 Rubin, H.A., Bruce, S., Rosen, T., McBride, M.E., 1991 Evidence for percutaneous inoculation as the mode of transmission for Chromoblastomycosis J Am Acad Dermatol 25, 951–954 Please cite this article in press as: Ahsan, M.K et al., Hidden under a cauliflower-like growth: A case of cutaneous chromoblastomycosis and response to combination therapy, Journal of Dermatology & Dermatologic Surgery (2016), http://dx.doi.org/10.1016/j.jdds.2016.11.001