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Leishmaniasis: Clinical characteristics of 22 cases
G Model
ARTICLE IN PRESS Med Clin (Barc). 2017;xxx(xx):xxx–xxx
www.elsevier.es/medicinaclinica
Scientific letter Leishmaniasis: Clinical characteristics of 22 cases夽 Leishmaniasis: características clínicas de 22 casos Dear Editor, Leishmaniasis is a parasitosis with a complex immunomodulatory response to infection. This response depends on the Leishmania species, the transmission vector and the host. The result is a wide clinical spectrum of polymorphic intermediate syndromic forms.1 Currently, atypical cases of multifocal cutaneous leishmaniasis (MCL) have been described in the literature,2–4 which encompass cases of localized CL, with a form of presentation that includes multiple lesions, smaller than the well-known “Middle East button”. Some authors have suggested that these atypical cases are associated with immunosuppression or malnutrition.4 In order to analyze their characteristics, we performed a caseseries retrospective observational study of leishmaniasis diagnosed in our department. Patients diagnosed in our service from 1st January 2008 to 31st October 2016, with at least one confirmatory method (histological, microbiological or molecular biology) were selected. Twenty-two cases of leishmaniasis were collected. Three cases were excluded, as they only had a clinical diagnosis. Of the 19 patients, 12 (63%) were males and 7 (37%) were females. The median age was 40 years, range: 1–83 years; 18 cases were autochthonous and one case was imported Leishmania (Equatorial Guinea). Analyzing the initial presentation, there were 9 cases of papular lesions, 5 cases of nodular lesions, 4 cases with ulcers and one plaque-shaped case. The single-element lesion was found in 13 cases, while 6 patients had multiple lesions. Topographically, there were 11 lesions in the region of the upper limbs, 10 in the cephalic region, 3 in the lower limbs and one lesion in the thorax, abdomen and back, respectively. Thirteen of our cases were immunocompetent patients and 6 immunocompromised patients (2 cases of diabetes mellitus, one in combination with golimumab for psoriatic arthropathy, one case of Crohn’s disease treated with adalimumab, one case of Takayasu arteritis treated with methotrexate, one case of human immunodeficiency virus infection [HIV], and one case of hepatitis C virus). Excluding the HIV case, 60% of cases involved multiple lesions, compared to 40% in patients classified as immunocompromised; and 23% of multiple lesions, compared to 77% in immunocompetent patients (p: 0.26). There was only one case of visceral leishmaniasis (VL), our HIV patient from Equatorial Guinea, who was treated with
intravenous liposomal amphotericin B (3 mg/kg for 10 days). VL was clinically ruled out with abdominal ultrasound and lab tests in the remaining immunosuppressed patients, and in selected cases, through polymerase chain reaction (PCR) detection in peripheral blood. The diagnosis was reached in 16 cases by histological techniques (including Giemsa), and in 3 cases by PCR of the lesion. Fifteen patients were treated with intralesional meglumine antimoniate (MA), with an average dose of 0.5–1 cm/lesion, and a median of 2 infiltrations. A complete response rate of 80% was obtained, with the remaining patients pending completion of treatment, with partial response. Of the remaining cases, 2 cases underwent local surgery, achieving recovery. One case was lost to follow-up. The single papular lesion was the most frequent presentation, predominantly affecting upper limbs and cephalic region, findings similar to those previously described.5 MA was the treatment of choice in our series, both in single and multiple forms. This treatment is the most widely used in our country, especially in the single forms. It is also used in the multiple forms, with results similar to those obtained.5 The proportion of multiple lesions in immunocompromised patients was higher than that of single lesions compared to immunocompetent patients, although this tendency did not reach statistical significance. These immunosuppressed patients (excluding HIV) were treated with MA, with complete response. This finding could demonstrate that, despite finding patients with MCL and some degree of immunosuppression, a systemic treatment would not be necessary, provided VL is ruled out, in order to avoid systemic drug toxicity. A less aggressive therapeutic option, such as the MA used, could control the disease even with multiple lesions. Larger studies are needed in order to test this hypothesis. References 1. Lupi O, Bartlett BL, Haugen RN, Dy LC, Sethi A, Klaus SN, et al. Tropical dermatology: tropical diseases caused by protozoa. J Am Acad Dermatol. 2009;60:897–925, quiz 926–28. 2. Maniscalco M, Noto G, Zichichi L, Veraldi S. Multifocal cutaneous leishmaniasis: a new clinical presentation of the disease. Acta Derm Venereol. 2007;87:275–6. 3. Madke B, Kharkar V, Chikhalkar S, Mahajan S, Khopkar U. Successful treatment of multifocal cutaneous leishmaniasis with miltefosine. Indian J Dermatol. 2011;56:587–90. 4. Ceyhan AM, Yildirim M, Basak PY, Akkaya VB. Unusual multifocal cutaneous leishmaniasis in a diabetic patient. Eur J Dermatol. 2009;19:514–5. 5. Aguado M, Espinosa P, Romero-Maté A, Tardío JC, Córdoba S, Borbujo J. Outbreak of cutaneous leishmaniasis in Fuenlabrada, Madrid. Actas Dermosifiliogr. 2013;104:334–42.
Sergio Santos Alarcón ∗ , María Isabel García-Briz, Almudena Mateu-Puchades Servicio de Dermatología, Hospital Universitario Doctor Peset, Valencia, Spain
夽 Please cite this article as: Santos Alarcón S, García-Briz MI, Mateu-Puchades A. Leishmaniasis: características clínicas de 22 casos. Med Clin (Barc) 2017. http://dx.doi.org/10.1016/j.medcli.2017.01.018
∗ Corresponding
author. E-mail address: [email protected] (S. Santos Alarcón).
˜ S.L.U. All rights reserved. 2387-0206/© 2017 Elsevier Espana,
MEDCLE-3955; No. of Pages 1
ARTICLE IN PRESS Med Clin (Barc). 2017;xxx(xx):xxx–xxx
www.elsevier.es/medicinaclinica
Scientific letter Leishmaniasis: Clinical characteristics of 22 cases夽 Leishmaniasis: características clínicas de 22 casos Dear Editor, Leishmaniasis is a parasitosis with a complex immunomodulatory response to infection. This response depends on the Leishmania species, the transmission vector and the host. The result is a wide clinical spectrum of polymorphic intermediate syndromic forms.1 Currently, atypical cases of multifocal cutaneous leishmaniasis (MCL) have been described in the literature,2–4 which encompass cases of localized CL, with a form of presentation that includes multiple lesions, smaller than the well-known “Middle East button”. Some authors have suggested that these atypical cases are associated with immunosuppression or malnutrition.4 In order to analyze their characteristics, we performed a caseseries retrospective observational study of leishmaniasis diagnosed in our department. Patients diagnosed in our service from 1st January 2008 to 31st October 2016, with at least one confirmatory method (histological, microbiological or molecular biology) were selected. Twenty-two cases of leishmaniasis were collected. Three cases were excluded, as they only had a clinical diagnosis. Of the 19 patients, 12 (63%) were males and 7 (37%) were females. The median age was 40 years, range: 1–83 years; 18 cases were autochthonous and one case was imported Leishmania (Equatorial Guinea). Analyzing the initial presentation, there were 9 cases of papular lesions, 5 cases of nodular lesions, 4 cases with ulcers and one plaque-shaped case. The single-element lesion was found in 13 cases, while 6 patients had multiple lesions. Topographically, there were 11 lesions in the region of the upper limbs, 10 in the cephalic region, 3 in the lower limbs and one lesion in the thorax, abdomen and back, respectively. Thirteen of our cases were immunocompetent patients and 6 immunocompromised patients (2 cases of diabetes mellitus, one in combination with golimumab for psoriatic arthropathy, one case of Crohn’s disease treated with adalimumab, one case of Takayasu arteritis treated with methotrexate, one case of human immunodeficiency virus infection [HIV], and one case of hepatitis C virus). Excluding the HIV case, 60% of cases involved multiple lesions, compared to 40% in patients classified as immunocompromised; and 23% of multiple lesions, compared to 77% in immunocompetent patients (p: 0.26). There was only one case of visceral leishmaniasis (VL), our HIV patient from Equatorial Guinea, who was treated with
intravenous liposomal amphotericin B (3 mg/kg for 10 days). VL was clinically ruled out with abdominal ultrasound and lab tests in the remaining immunosuppressed patients, and in selected cases, through polymerase chain reaction (PCR) detection in peripheral blood. The diagnosis was reached in 16 cases by histological techniques (including Giemsa), and in 3 cases by PCR of the lesion. Fifteen patients were treated with intralesional meglumine antimoniate (MA), with an average dose of 0.5–1 cm/lesion, and a median of 2 infiltrations. A complete response rate of 80% was obtained, with the remaining patients pending completion of treatment, with partial response. Of the remaining cases, 2 cases underwent local surgery, achieving recovery. One case was lost to follow-up. The single papular lesion was the most frequent presentation, predominantly affecting upper limbs and cephalic region, findings similar to those previously described.5 MA was the treatment of choice in our series, both in single and multiple forms. This treatment is the most widely used in our country, especially in the single forms. It is also used in the multiple forms, with results similar to those obtained.5 The proportion of multiple lesions in immunocompromised patients was higher than that of single lesions compared to immunocompetent patients, although this tendency did not reach statistical significance. These immunosuppressed patients (excluding HIV) were treated with MA, with complete response. This finding could demonstrate that, despite finding patients with MCL and some degree of immunosuppression, a systemic treatment would not be necessary, provided VL is ruled out, in order to avoid systemic drug toxicity. A less aggressive therapeutic option, such as the MA used, could control the disease even with multiple lesions. Larger studies are needed in order to test this hypothesis. References 1. Lupi O, Bartlett BL, Haugen RN, Dy LC, Sethi A, Klaus SN, et al. Tropical dermatology: tropical diseases caused by protozoa. J Am Acad Dermatol. 2009;60:897–925, quiz 926–28. 2. Maniscalco M, Noto G, Zichichi L, Veraldi S. Multifocal cutaneous leishmaniasis: a new clinical presentation of the disease. Acta Derm Venereol. 2007;87:275–6. 3. Madke B, Kharkar V, Chikhalkar S, Mahajan S, Khopkar U. Successful treatment of multifocal cutaneous leishmaniasis with miltefosine. Indian J Dermatol. 2011;56:587–90. 4. Ceyhan AM, Yildirim M, Basak PY, Akkaya VB. Unusual multifocal cutaneous leishmaniasis in a diabetic patient. Eur J Dermatol. 2009;19:514–5. 5. Aguado M, Espinosa P, Romero-Maté A, Tardío JC, Córdoba S, Borbujo J. Outbreak of cutaneous leishmaniasis in Fuenlabrada, Madrid. Actas Dermosifiliogr. 2013;104:334–42.
Sergio Santos Alarcón ∗ , María Isabel García-Briz, Almudena Mateu-Puchades Servicio de Dermatología, Hospital Universitario Doctor Peset, Valencia, Spain
夽 Please cite this article as: Santos Alarcón S, García-Briz MI, Mateu-Puchades A. Leishmaniasis: características clínicas de 22 casos. Med Clin (Barc) 2017. http://dx.doi.org/10.1016/j.medcli.2017.01.018
∗ Corresponding
author. E-mail address: [email protected] (S. Santos Alarcón).
˜ S.L.U. All rights reserved. 2387-0206/© 2017 Elsevier Espana,
MEDCLE-3955; No. of Pages 1