Abstract
Eumycetomas are chronic suppurative granulomas caused by fungi characterised by invasive tumefactive lesions, sinuses and discharging grains. Herein, we describe a case of pedal eumycetoma due to Fusarium solani sensu stricto in a person with diabetes mellitus. A 45-year-old gentleman presented with an insidious onset swelling over his right foot with nodules and discharging grains. He had received itraconazole and anti-tuberculous therapy elsewhere, without response. Re-evaluation included a biopsy which confirmed eumycetoma and newly diagnosed diabetes. Surgical excision followed by histopathological, microbiological and multigene sequencing analyses [translation elongation factor, calmodulin and internal transcribed spacer region of rDNA] of the mould on culture were performed. Histopathology revealed septate fungal hyphae amidst a dense inflammatory infiltrate (Splendore–Hoeppli) reaction. Oral voriconazole was started and good glycemic control attained. Tissue growth sequences showed > 99% similarity with Fusarium solani sensu stricto. Antifungal susceptibility testing showed lowest MIC to voriconazole (0.5 mg/L). The patient showed excellent response to combined therapeutic modality with a near-complete resolution in size of lesion and obliteration of sinuses following 4 months of therapy and is planned for prolonged voriconazole therapy till complete radiological resolution. Diabetes predisposes to fungal infections of foot but eumycetomas are uncommon. Combined surgery and antifungals can improve morbidity and avoid amputations.
References
Nenoff P, Van de Sande WW, Fahal AH, Reinel D, Schöfer H. Eumycetoma and actinomycetoma–an update on causative agents, epidemiology, pathogenesis, diagnostics and therapy. J Eur Acad Dermatol Venereol. 2015;29(10):1873–83.
Bustamante B, Campos PE. Eumycetoma. In: Essentials of clinical mycology. New York: Springer; 2011, p. 415–25.
Al-Hatmi AM, Bonifaz A, Tirado-Sánchez A, Meis JF, de Hoog GS, Ahmed SA. Fusarium species causing eumycetoma: report of two cases and comprehensive review of the literature. Mycoses. 2017;60(3):204–12.
Tomimori-Yamashita J, Ogawa MM, Hirata SH, Fischman O, Michalany NS, Yamashita HK, Alchorne MM. Mycetoma caused by Fusarium solani with osteolytic lesions on the hand: case report. Mycopathologia. 2002;153(1):11–4.
Sandhu G, Prakash M, Narang T. ‘Dot in circle sign’in thigh: an unusual site of mycetoma. BMJ Case Rep. 2019;12(5):e229499; https://doi.org/10.1136/bcr-2019-229499.
Al-Hatmi AM, Meis JF, de Hoog GS. Fusarium: molecular diversity and intrinsic drug resistance. PLoS Pathog. 2016;12(4):e1005464.
van de Sande WW. Global burden of human mycetoma: a Mycoses review and meta-analysis. PLoS Negl Trop Dis. 2013;7(11):e2550.
Dignani MC, Anaissie E. Human fusariosis. Clin Microbiol Infect. 2004;10:67–75.
Ajello L, Padhye AA, Chandler FW, McGinnis MR, Morganti L, Alberici F. Fusarium moniliforme, a new mycetoma agent restudy of a European case. Eur J Epidemiol. 1985;1(1):5–10.
Sarris I, Berendt AR, Athanasous N, Ostlere SJ. MRI of mycetoma of the foot: two cases demonstrating the dot-in-circle sign. Skeletal Radiol. 2003;32(3):179–83.
Ahmed AO, van Leeuwen W, Fahal A, van de Sande W, Verbrugh H, van Belkum A. Mycetoma caused by Madurellamycetomatis: a neglected infectious burden. Lancet Infect Dis. 2004;4(9):566–74.
Yera H, Bougnoux ME, Jeanrot C, Baixench MT, De Pinieux G, Dupouy-Camet J. Mycetoma of the foot caused by Fusarium solani: identification of the etiologic agent by DNA sequencing. J Clin Microbiol. 2003;41(4):1805–8.
Dutta P, Premkumar A, Chakrabarti A, Shah VN, Behera A, De D, Rudramurthy SM, Bhansali A. Fusarium falciforme infection of foot in a patient with type 2 diabetes mellitus: a case report and review of the literature. Mycopathologia. 2013;176(3–4):225–32.
Queiroz-Telles F, Fahal AH, Falci DR, Caceres DH, Chiller T, Pasqualotto AC. Neglected endemic mycoses. Lancet Infect Dis. 2017;17(11):e367–77.
Velho PE, Drummond MR, Schreiber AZ, Cintra ML. Mycetoma-like phaeohyphomycosis treated with terbinafine. IDCases. 2020;19:e00705. https://doi.org/10.1016/j.idcr.2020.e00705.
Veraldi S, Grancini A, Venegoni L, Merlo V, Guanziroli E, Menicanti C, Nazzaro G, Tortorano A. Mycetoma caused by Aspergillus nidulans. Acta Dermato-Venereol. 2016;96(1):118–9.
Yadav S, Agarwal R, Singh S, Goel S. Pyrenochaeta romeroi causing subcutaneous phaeohyphomycotic cyst in a diabetic female. Med Mycol Case Rep. 2015;8:47–9.
Boyce Z, Collins N. Scedosporium apiospermum: an unreported cause of fungal sporotrichoid-like lymphocutaneous infection in Australia and review of the literature. Australas J Dermatol. 2015;56(2):e39-42.
Ahmed SA, Desbois N, Quist D, Miossec C, Atoche C, Bonifaz A, de Hoog GS. Phaeohyphomycosis caused by a novel species, Pseudochaetosphaeronema martinelli. J Clin Microbiol. 2015;53(9):2927–34.
Ahmed SA, Abbas MA, Jouvion G, Al-Hatmi AM, de Hoog GS, Kolecka A, Mahgoub ES. Seventeen years of subcutaneous infection by Aspergillus flavus; eumycetoma confirmed by immunohistochemistry. Mycoses. 2015;58(12):728–34.
Verma R, Vasudevan B, Sahni AK, Vijendran P, Neema S, Kharayat V. First reported case of Aspergillus nidulans eumycetoma in a sporotrichoid distribution. Int J Dermatol. 2015;54(1):74–7.
Henckaerts L, Naesens R, Vlieghe E, Jansens H, Gielen J, Ieven M, Moorkens G. A tropical diabetic foot. Acta Clin Belg. 2012;67(5):362–4.
Vyawahare C, Gandham N, Roy I, Misra R, Sardar M, Jadhav SV. White grain eumycetoma caused by Aspergillus nidulans, a rare report from district of Maharashtra India. Int J Med Clin Res. 2012;3(7):212.
Kotwal N, Yanamandra U, Badwal S, Nair V. Mycetoma foot caused by Aspergillus in a diabetic patient. Intern Med. 2012;51(5):517–8.
Malone M, Gannass A, Bowling F. A chronic, destructive mycetoma infection in a diabetic foot in Saudi Arabia. Int J Lower Extrem Wounds. 2011;10(1):12–5.
Das S, Saha R, Dar SA, Ramachandran VG. Acremonium species: a review of the etiological agents of emerging hyalohyphomycosis. Mycopathologia. 2010;170(6):361–75.
Wu CY, Chen GS, Lan CC. Onychomycosis caused by Fusarium solani in a woman with diabetes. Clin Exp Dermatol Clin Dermatol. 2009;34(8):e772–4.
Prasanna S, Grover N, Bhatt P, Sahni AK. A case of Aspergillus nidulans causing white granule mycetoma. Med J Armed Forces India. 2016;72(1):88.
Pérez-Pérez L, Pereiro M Jr, Sánchez-Aguilar D, Toribio J. Ulcerous lesions disclosing cutaneous infection with Fusarium solani. Acta Dermato-Venereol. 2007;87(5):422–4.
Geyer AS, Fox LP, Husain S, Della-Latta P, Grossman ME. Acremonium mycetoma in a heart transplant recipient. J Am Acad Dermatol. 2006;55(6):1095–100.
Taj-Aldeen SJ, Gene J, Bozom IA, Buzina W, Cano JF, Guarro J. Gangrenous necrosis of the diabetic foot caused by Fusarium acutatum. Med Mycol. 2006;44(6):547–52.
Lyke KE, Miller NS, Towne L, Merz WG. A case of cutaneous ulcerative alternariosis: rare association with diabetes mellitus and unusual failure of itraconazole treatment. Clin Infect Dis. 2001;32(8):1178–87.
Fahal AH, van De Sande WW. The epidemiology of mycetoma. Curr Fungal Infect Rep. 2012;6(4):320–6.
Relhan V, Mahajan K, Agarwal P, Garg VK. Mycetoma: an update. Indian J Dermatol. 2017;62(4):332.
Ali RS, Newport MJ, Bakhiet SM, Ibrahim ME, Fahal AH. Host genetic susceptibility to mycetoma. PLOS Negl Trop Dis. 2020;14(4):e0008053.
Zijlstra EE, Van De Sande WW, Welsh O, Goodfellow M, Fahal AH. Mycetoma: a unique neglected tropical disease. Lancet Infect Dis. 2016;16(1):100–12.
Welsh O, Vera-Cabrera L, Salinas-Carmona MC. Mycetoma. Clin Dermatol. 2007;25(2):195–202.
El Shamy ME, Fahal AH, Shakir MY, Homeida MM. New MRI grading system for the diagnosis and management of mycetoma. Trans R Soc Trop Med Hyg. 2012;106(12):738–42.
Nazzaro G, Veraldi S. Mycetomas: the experience of the dermatology unit of the University of Milan. Dermatopathology. 2018;5(1):6–9.
Hay R, Denning DW, Bonifaz A, Queiroz-Telles F, Beer K, Bustamante B, Chakrabarti A, Chavez-Lopez MD, Chiller T, Cornet M, Estrada R. The diagnosis of fungal neglected tropical diseases (fungal NTDs) and the role of investigation and laboratory tests: an expert consensus report. Trop Med Infect Dis. 2019;4(4):122.
Hussein MR. Mucocutaneous Splendore–Hoeppli phenomenon. J Cutan Pathol. 2008;35(11):979–88.
Rosa PD, Heidrich D, Corrêa C, Scroferneker ML, Vettorato G, Fuentefria AM, Goldani LZ. Genetic diversity and antifungal susceptibility of Fusarium isolates in onychomycosis. Mycoses. 2017;60(9):616–22.
Bakhiet SM, Fahal AH, Musa AM, Mohamed ES, Omer RF, Ahmed ES, El Nour M, Mustafa ER, Rahman MESA, Suliman SH, El Mamoun MA. A holistic approach to the mycetoma management. PLoS Negl Trop Dis. 2018;12(5):e0006391.
Darré T, Saka B, Mouhari-Toure A, Tchaou M, Dorkenoo AM, Doh K, Walla A, Amégbor K, Pitché VP, Napo-Koura G. Mycetoma in the Togolese: an update from a single-center experience. Mycopathologia. 2018;183(6):961–5.
da Dallé Rosa P, Ramirez-Castrillon M, Borges R, Aquino V, Fuentefria AM, Goldani LZ. Epidemiological aspects and characterization of the resistance profile of Fusarium spp. in patients with invasive fusariosis. J Med Microbiol. 2019;68(10):1489–96.
van de Sande WW. Global burden of human mycetoma: a systematic review and meta-analysis. PLoS Negl Trop Dis. 2013;7(11):e2550.
Schroers HJ, Samuels GJ, Zhang N, Short DP, Juba J, Geiser DM. Epitypification of Fusisporium (Fusarium) solani and its assignment to a common phylogenetic species in the Fusarium solani species complex. Mycologia. 2016;108(4):806–19.
Gupta S, Koirala J, Khardori R, Khardori N. Infections in diabetes mellitus and hyperglycemia. Infect Dis Clin N Am. 2007;21(3):617–38.
Mohanty P, HAmouda W, Garg R, Aljada A, Ghanim H, Dandona P. Glucose challenge stimulates reactive oxygen species (ROS) generation by leucocytes. J Clin Endocrinol Metab. 2000;85(8):2970–3.
Boral H, Van Diepeningen A, Erdem E, Yağmur M, De Hoog GS, Ilkit M, Meis JF, Al-Hatmi AM. Mycotic keratitis caused by Fusarium solani sensu stricto (FSSC5): a case series. Mycopathologia. 2018;183(5):835–40.
Acknowledgements
The authors wish to acknowledge Prof. Arunaloke Chakrabarti from the division of mycology, department of medical microbiology, PGIMER, for his support and guidance.
Funding
There was no grant availed of, for this study.
Author information
Authors and Affiliations
Contributions
All authors contributed to the conception and design, acquisition of data, and/or analysis and interpretation of data and have read before the approval of the final manuscript.
Corresponding authors
Ethics declarations
Conflict of interest
The authors have no conflicts of interest to declare.
Informed Consent
Written, informed consent was obtained from the patient for participation and publication.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Handling Editor: Stephane Ranque.
Electronic supplementary material
Below is the link to the electronic supplementary material.
11046_2020_524_MOESM1_ESM.tif
Supplementary Figure 1: The UPGMA clustering phylogram constructed using the sequences of translation elongation factor, calmodulin gene and internal transcribed spacer region of rDNA in Fusarium MLST database. (Data in the phylogram indicate the isolate of index patient) (TIFF 517 kb)
Rights and permissions
About this article
Cite this article
Das, L., Dahiya, D., Gupta, K. et al. Eumycetoma of the Foot due to Fusarium solani in a Person with Diabetes Mellitus: Report of a Case and Review of Literature. Mycopathologia 186, 277–288 (2021). https://doi.org/10.1007/s11046-020-00524-y
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11046-020-00524-y