SUBMITTED 30 MAR 22 1 REVISION REQ. 14 JUN 22; REVISION RECD. 16 JUN 22 2 ACCEPTED 21 JUL 22 3 ONLINE-FIRST: AUGUST 2022 4 DOI: https://doi.org/ 10.18295/squmj.8.2022.048 5 6 Long-standing Lymphocutaneous Sporotrichosis 7 Israel Perez-López and *Ricardo Ruiz-Villaverde 8 9 Department of Dermatology, Hospital Universitario San Cecilio, Inst Invest 10 Biosanitaria IBS, Granada, Spain 11 *Corresponding Author’s e-mail: ismenios@hotmail.com 12 13 Introduction 14 A 36-year-old male, farmer, with no relevant personal or family medical history , 15 attended our Dermatology outpatient clinic, complaining of a warty plaque on the back 16 of his right hand that involved the entire dorsal aspect of the third finger and that 17 presented two satellites lesions on the arm with a linear arrangement (Fig.1.). 18 Locoregional adenopathies were not noted. He recalled a previous traumatic history 19 performing his work 10 months earlier. Two weeks later the lesions began to develop 20 until they reached their current state. 21 22 Laboratory examinations included blood sample test (haemogram and basical 23 coagulation), general biochemistry (lipid profile, hepatic and renal function), 24 autoimmunity (autoantibodies, immunoglobulins, complements), thyrotropin and 25 thoracoabdominal CT was requested to evaluate systemic dissemination with normality 26 of all the tests mentioned. No skin biopsy was performed. Regarding the initial clinical 27 suspicion of sporotrichosis, we started treatment with Itraconazole 200mg/12h, which 28 had to be discontinued due to gastrointestinal discomfort 10 days later. The study by 29 PCR assay of M. tuberculosis complex and Non tuberculous Mycobacterium spp was 30 negative. However, culture showed positive results for Sporothrix schenckii in five days 31 (being reported two days later). One week later the patient began treatment with 32 liposomal Amphotericin B at a dose of 5 mg/kg/day for 3 days as an induction regimen 33 and then once monthly as maintenance treatment until resolution of the clinical course, 34 four months later with no skin lesions. There have been no recurrences of the lesion six 35 months after the end of treatment. Written informed consent of the patient for 36 publication purposes has been obtained. 37 38 Comment 39 Sporotrichosis is a subcutaneous mycosis caused by a dimorphic fungus of the genus 40 Sporothrix. Sporotrichosis can be observed in any part of the world, with some areas of 41 "hyperendemicity", being particularly frequent in tropical and subtropical countries (in 42 Peru the reported incidence is 1/1,000 cases/year, while in the United States it is 1- 2 43 cases per million) due to more occupational exposure.1 44 45 Without predilection for age, sex or race, the occurence of the disease depends on the 46 fungus being in the environment and traumatic inoculation into the skin . The male 47 predominance is believed to be due to greater exposure rather than greater 48 predisposition. Traumatic inoculation is the reason why the extremities (particularly 49 upper extremities) and bare parts are affected most often. Single or multiloculated 50 cutaneous forms, lymphocutaneous, as in the case at hand, and systemic (may 51 compromise lung, breasts, liver, kidney, eyes, heart, and genitalia) have been 52 described.2 Culture in Sabouraud Dextrose Agar continues to be the gold standard, since 53 histological study may be less profitable and require histochemical techniques such as 54 PAS, Groccot or Gomori to identify fungal structures. In some reference centers, PCR 55 assay is used for its diagnosis, although the kits are not commercially available. The 56 differential diagnosis following a sporotrichoid eruption pattern is wide, encompassing 57 cutaneous tuberculosis, leishmaniasis, nocardiosis, chromoblastomycosis, 58 blastomycosis, paracoccidioidomycosis, and atypical mycobacteriosis. 59 60 The first line of treatment in the lymphocutaneous variety is oral itraconazole 61 100mg/day.3 Oral terbinafine has been used with moderate success and in more 62 resistant cases the use of oral saturated solution of potassium iodide (SSKI) has been 63 reported. This last modality lacks a standardized commercial formulation and its 64 metallic taste and uncertain mechanism of action have relegated it to a second or third 65 line choice. Liposomal amphotericin B may be a more effective option in 66 immunosuppressed patients or those with low therapeutic adherence.4 Among its side 67 effects are fever, headache, malaise, hypokaliemia, hypomagnesemia, cardio and 68 nephrotoxicity. The use of other less conventional treatments such as photodynamic 69 therapy has only been carried out in case series and with uneven results.5 70 71 Author Contributions 72 IPL and RRV were both involved in conceptualization, collection of data, writing and 73 final approval of the manuscript. 74 75 References 76 1. Sharma B, Sharma AK, Sharma U. Sporotrichosis: a Comprehensive Review on 77 Recent Drug-Based Therapeutics and Management. Curr Dermatol Rep. 2022 78 Mar 17:1-10. doi: 10.1007/s13671-022-00358-5. 79 2. Martínez-Herrera E, Arenas R, Hernández-Castro R, Frías-De-León MG, 80 Rodríguez-Cerdeira C. Uncommon Clinical Presentations of Sporotrichosis: A 81 Two-Case Report. Pathogens. 2021 Sep 27;10(10):1249. doi: 82 10.3390/pathogens10101249. 83 3. Poester VR, Basso RP, Stevens DA, Munhoz LS, de Souza Rabello VB, 84 Almeida-Paes R, Zancopé-Oliveira RM, Zanchi M, Benelli JL, Xavier MO. 85 Treatment of Human Sporotrichosis Caused by Sporothrix brasiliensis. J Fungi 86 (Basel). 2022 Jan 10;8(1):70. doi: 10.3390/jof8010070. 87 4. Belda W Jr, Domingues Passero LF, Stradioto Casolato AT. Lymphocutaneous 88 Sporotrichosis Refractory to First-Line Treatment. Case Rep Dermatol Med. 89 2021 Oct 6;2021:9453701. doi: 10.1155/2021/9453701. 90 5. Legabão BC, Fernandes JA, de Oliveira Barbosa GF, Bonfim-Mendonça PS, 91 Svidzinski TIE. The zoonosis sporotrichosis can be successfully treated by 92 photodynamic therapy: A scoping review. Acta Trop. 2022 Apr;228:106341. 93 doi: 10.1016/j.actatropica.2022.106341. 94 95 96 Fig. 1: Verrucous plaque on the back of the right hand that involved the entire dorsal 97 aspect of the third finger with two satellites lesions on the arm with a linear 98 arrangement. 99