Research letter
Possible DRESS syndrome in a child with borreliosis
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Lane P<span class="elsevierStyleInf">1</span>: Patient 1 serum; lane P<span class="elsevierStyleInf">2</span>: Patient 2 serum; lane P<span class="elsevierStyleInf">3</span>: Patient 3 serum; lane C: Control serum (pool of serum from non-atopic subjects); Lane M: Molecular mass marker.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "V. Marenco-Arellano, B. Bartolome, M. Reaño-Martos, C. Marrero, M. Rodríguez-Cabreros, A. Iglesias-Cadarso" "autores" => array:6 [ 0 => array:2 [ "nombre" => "V." "apellidos" => "Marenco-Arellano" ] 1 => array:2 [ "nombre" => "B." "apellidos" => "Bartolome" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Reaño-Martos" ] 3 => array:2 [ "nombre" => "C." "apellidos" => "Marrero" ] 4 => array:2 [ "nombre" => "M." "apellidos" => "Rodríguez-Cabreros" ] 5 => array:2 [ "nombre" => "A." "apellidos" => "Iglesias-Cadarso" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0301054611001583?idApp=UINPBA00004N" "url" => "/03010546/0000004000000002/v1_201304101104/S0301054611001583/v1_201304101104/en/main.assets" ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Research letter</span>" "titulo" => "Possible DRESS syndrome in a child with borreliosis" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">To the Editor,</span>" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "129" "paginaFinal" => "131" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "R. Silva, C. Botelho, S. Cadinha, C. Lisboa, I. Azevedo, J.R. Cernadas" "autores" => array:6 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Silva" "email" => array:1 [ 0 => "ruisilva@inbox.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "C." "apellidos" => "Botelho" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "S." "apellidos" => "Cadinha" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "C." "apellidos" => "Lisboa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 4 => array:3 [ "nombre" => "I." "apellidos" => "Azevedo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 5 => array:3 [ "nombre" => "J.R." "apellidos" => "Cernadas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Allergy Division, Hospital S. João EPE, Porto, Portugal" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Dermatology Division, Hospital S. João EPE, Porto, Portugal" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Paediatrics Division, Hospital S. João EPE, Porto, Portugal" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 469 "Ancho" => 1250 "Tamanyo" => 72916 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Lesions at sites of penicillin administration.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Antibiotic hypersensitivity reactions are a major health concern as they can be a significant cause of morbidity and mortality, limit therapeutic options, and increase socio-economic costs. Diagnosis can be challenging, as great number of drugs can elicit different immune-mediated reactions. A combination of clinical history and different tests is generally necessary to confirm the diagnosis, since none has sufficient sensitivity to be used alone.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In some non-immediate hypersensitivity reactions, the lymphocyte transformation test (LTT), which measures the <span class="elsevierStyleItalic">in vitro</span> proliferation of T lymphocytes in the presence of a suspected drug, can be the only tool to confirm the diagnosis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a previously healthy 34-month-old girl who due to fever and odynophagia (interpreted as tonsillitis), was treated with amoxicillin without improvement. Three days later she began treatment with cefaclor, without result. She was then given two shots of penicillin, one in each buttock, with apyrexia after 48<span class="elsevierStyleHsp" style=""></span>h. Two weeks later, in the same location where penicillin was administered, two nodular inflammatory lesions appeared, with progressive worsening (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Due to the severity of these lesions, she was admitted to local hospital. Shortly after, she developed fever and was submitted to a surgical intervention at her buttocks for drainage of suspected abscess (not confirmed). Due to deterioration of her general status and suspected infectious panniculitis, she was transferred to our Central Hospital, and began flucloxacillin and clindamycin. Three days later, a diffuse exanthema (including both soles) appeared and an allergist was called. Flucloxacillin was stopped and the exanthema faded away, but a cutaneous <span class="elsevierStyleItalic">erythema migrans</span>, oedema of the left hand and both feet developed. After a careful and complete clinical history, a non-immediate hypersensitivity reaction to penicillin was suspected. The child began treatment with prednisolone (1.5<span class="elsevierStyleHsp" style=""></span>mg/kg/day). Buttock lesions began to disappear with objective improvement after third day of corticotherapy. Because of the <span class="elsevierStyleItalic">erythema migrans</span> lesion, a dermatologist was called.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Haemoglobin, platelets and creatinine results were normal. The white blood cells count showed leukocytosis with immature cells and eosinophilia (3000/μL), elevated AST (231<span class="elsevierStyleHsp" style=""></span>UI/L) and an aPTT time <20<span class="elsevierStyleHsp" style=""></span>s. IgG and IgA levels were normal, IgM was slightly elevated (239<span class="elsevierStyleHsp" style=""></span>mg/dL), as well as total IgE (314<span class="elsevierStyleHsp" style=""></span>mg/dL). Parvovirus B19, Epstein Barr virus (EBV) and Borrelia IgM antibodies were positive, as well as Herpesvirus 6 (HHV-6) DNA on peripheral blood. The immunophenotyping and lymphocyte function <span class="elsevierStyleItalic">in vitro</span> study were normal. Auto-antibody screening revealed negative antinuclear and anticardiolipin antibodies. A skin biopsy at the site of one <span class="elsevierStyleItalic">erythema migrans</span> lesion showed dermis with inflammatory infiltrate, mainly lymphocytes, some neutrophils and eosinophils, with perivascular localisation and transendothelial migration, and the presence of DNA of <span class="elsevierStyleItalic">Borrelia burgdorferi,</span> Parvovirus B19 and EBV. At the same time, the result from the buttock lesion biopsy done a few days before became available, showing an unspecific inflammatory infiltrate with abundant eosinophils and scarce plasma cells. Prednisolone was maintained, with slow dose tapering.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Because allergy to penicillin was one of the most probable diagnosis, second line therapy with azithromycin for Borreliosis (14 days) was started, with clinical improvement and disappearance of the <span class="elsevierStyleItalic">erythema migrans</span>. The inflammatory lesions on both buttocks slowly disappeared. Specific IgE for penicillolyl G and V were both negative. A LTT was performed with penicillin, amoxicillin and cefuroxime. The stimulation index (SI) was calculated as proliferation with drug/proliferation without drug. The result was clearly positive to penicillin with a SI of 60.2 (considered positive when >3) at 100<span class="elsevierStyleHsp" style=""></span>μg/mL, and indeterminate for amoxicillin and cefuroxime. DNA for all agents on peripheral blood was negative after five months.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Lyme borreliosis is the most commonly reported vector-borne disease in Europe and North America, and represents a major public health challenge. It is transmitted by ticks harbouring the infectious agent <span class="elsevierStyleItalic">B. burgdorferi</span>.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The age distribution of lyme disease is bimodal, with the highest number of cases occurring in children with ages ranging from 5 to 14 years and adults from 55 to 74 years.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">This child developed a delayed hypersensitivity reaction to penicillin, with severe local manifestation, misinterpreted as panniculitis. She was successfully treated for Borreliosis (her basic illness) with a non-beta-lactam antibiotic. The diagnosis of beta-lactam allergy was confirmed by the LTT, which prove to be a useful tool to confirm the diagnosis of non-immediate drug hypersensitivity reaction. Several authors have suggested an association between severe drug hypersensitivity reactions, as is the Drug Rash with Eosinophilia and Systemic Symptoms (DRESS) syndrome, and reactivation of herpesvirus, including HHV-6 and EBV,<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5–7</span></a> such as seen in this patient, and which might explain the leukocytosis with immature cells and eosinophilia, and the hepatic involvement.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 469 "Ancho" => 1250 "Tamanyo" => 72916 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Lesions at sites of penicillin administration.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Work up of patients with history of beta-lactam hypersensitivity" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R. 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| 2017 August | 9 | 5 | 14 |
| 2017 July | 10 | 15 | 25 |
| 2017 June | 17 | 9 | 26 |
| 2017 May | 37 | 9 | 46 |
| 2017 April | 22 | 4 | 26 |
| 2017 March | 24 | 36 | 60 |
| 2017 February | 11 | 4 | 15 |
| 2017 January | 9 | 3 | 12 |
| 2016 December | 29 | 4 | 33 |
| 2016 November | 20 | 6 | 26 |
| 2016 October | 29 | 4 | 33 |
| 2016 September | 33 | 5 | 38 |
| 2016 August | 12 | 8 | 20 |
| 2016 July | 13 | 2 | 15 |
| 2016 June | 17 | 7 | 24 |
| 2016 May | 19 | 10 | 29 |
| 2016 April | 32 | 31 | 63 |
| 2016 March | 30 | 7 | 37 |
| 2016 February | 24 | 8 | 32 |
| 2016 January | 12 | 7 | 19 |
| 2015 December | 19 | 8 | 27 |
| 2015 November | 20 | 3 | 23 |
| 2015 October | 21 | 4 | 25 |
| 2015 September | 11 | 4 | 15 |
| 2015 August | 39 | 6 | 45 |
| 2015 July | 41 | 3 | 44 |
| 2015 June | 25 | 1 | 26 |
| 2015 May | 24 | 1 | 25 |
| 2015 April | 13 | 7 | 20 |
| 2015 March | 4 | 2 | 6 |
| 2015 February | 6 | 1 | 7 |
| 2015 January | 18 | 3 | 21 |
| 2014 December | 14 | 4 | 18 |
| 2014 November | 6 | 2 | 8 |
| 2014 October | 16 | 9 | 25 |
| 2014 September | 19 | 2 | 21 |
| 2014 August | 5 | 3 | 8 |
| 2014 July | 13 | 1 | 14 |
| 2014 June | 3 | 1 | 4 |
| 2014 May | 14 | 3 | 17 |
| 2014 April | 5 | 3 | 8 |
| 2014 March | 57 | 13 | 70 |
| 2014 February | 49 | 19 | 68 |
| 2014 January | 37 | 10 | 47 |
| 2013 December | 45 | 6 | 51 |
| 2013 November | 39 | 9 | 48 |
| 2013 October | 49 | 10 | 59 |
| 2013 September | 44 | 10 | 54 |
| 2013 August | 70 | 11 | 81 |
| 2013 July | 39 | 11 | 50 |
| 2013 June | 44 | 4 | 48 |
| 2013 May | 44 | 7 | 51 |
| 2013 April | 24 | 6 | 30 |
| 2013 March | 14 | 4 | 18 |
| 2013 February | 12 | 8 | 20 |
| 2013 January | 5 | 2 | 7 |
| 2012 December | 5 | 5 | 10 |
| 2012 November | 1 | 3 | 4 |
| 2012 October | 1 | 2 | 3 |
| 2012 September | 1 | 1 | 2 |
| 2012 March | 338 | 0 | 338 |
Show all
