Letter to the Editor
Liver transplantation in a patient with fulminant hepatitis by DRESS syndrome
Trasplante hepático por hepatitis fulminante en un paciente con síndrome DRESS
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Both the anteroposterior (A) and posteroanterior (B) scintigraphy shows an increased right paramediastinal density. In the SPECT-CT images, a mass is observed that coincides with said uptake in the coronal section (<span class="elsevierStyleSmallCaps">C</span>) and the sagittal section (D). Increased uptake in the thyroid bed, compatible with multinodular goitre, and physiological uptake in the salivary glands can also be observed.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Álvaro Vidal-Suárez, María del Mar Roca-Rodríguez, Isabel Torres-Barea" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Álvaro" "apellidos" => "Vidal-Suárez" ] 1 => array:2 [ "nombre" => "María del Mar" "apellidos" => "Roca-Rodríguez" ] 2 => array:2 [ "nombre" => "Isabel" "apellidos" => "Torres-Barea" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S002577532030110X" "doi" => "10.1016/j.medcli.2020.01.015" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S002577532030110X?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020621000929?idApp=UINPBA00004N" "url" => "/23870206/0000015600000007/v1_202104020834/S2387020621000929/v1_202104020834/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S2387020621000942" "issn" => "23870206" "doi" => "10.1016/j.medcle.2021.03.001" "estado" => "S300" "fechaPublicacion" => "2021-04-09" "aid" => "5162" "copyright" => "Elsevier España, S.L.U." 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A perivascular and periadnexal lymphocytic infiltrate (arrows) can be seen in the underlying dermis, compatible with a cutaneous reaction to drugs. B) Liver ×100. Extensive necrosis, predominantly centrilobular, is identified, showing significant venular involvement (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Drug Rash with Eosinophilia and Systemic Symptoms (DRESS) syndrome is a rare entity secondary to an adverse drug reaction. Although its pathophysiology is not well known, some causes are proposed, such as a deficiency of detoxification enzymes of the inducing drug's active metabolites, the inflammatory cascade of interleukin-5 released by drug-specific T cells, or interaction with the herpes-6 virus<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>. It is associated with the use of antibiotics, anticonvulsants and antihypertensives, and symptoms appear more than 2 weeks after exposure.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The diagnosis is based on Bocquet’s criteria<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a>: 1) skin rash, 2) eosinophilia greater than 1500/mm<span class="elsevierStyleSup">3</span> and/or atypical lymphocytes, 3) systemic involvement with lymphadenopathy (more than 2<span class="elsevierStyleHsp" style=""></span>cm in diameter) and/or hepatitis with elevated aminotransferases at least twice the normal values and/or interstitial nephritis, and/or pneumonitis, and/or carditis and 4) fever<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a>. Skin manifestations initially appear on the face, trunk and limbs, and periorbital oedema is characteristic. It usually improves after discontinuation of the drug, although a 10% mortality rate has been reported. Treatment requires initiation with prednisone 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day and immunoglobulins in severe cases<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>.</p><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of a 15-year-old young man with severe acne as the only history of interest. The patient had fever of 39<span class="elsevierStyleHsp" style=""></span>°C and low back pain, for which empirical antibiotic therapy with amoxicillin-clavulanate was started. Beta-lactam resistant <span class="elsevierStyleItalic">Streptococcus hominis</span> was isolated in blood cultures, so it is replaced by vancomycin 1<span class="elsevierStyleHsp" style=""></span>g/12<span class="elsevierStyleHsp" style=""></span>h. Following an MRI showing increased signal in L3 and T12 vertebrae, the possibility of lumbar osteomyelitis is considered, combining rifampicin and ceftriaxone, until it is ruled out by bone scintigraphy. The course was unfavourable, with persistent fever and elevated acute phase reactants, for which he received multiple empirical antibiotic regimens (cefotaxime, cloxacillin, trimethoprim-sulfamethoxazole and piperacillin-tazobactam).</p><p id="par0020" class="elsevierStylePara elsevierViewall">Given the absence of a conclusive infectious diagnosis, the possibility of a SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) was considered, prescribing non-steroidal anti-inflammatory drugs. After 2 weeks of progressive improvement, the patient was discharged. However, he was readmitted after 2 days with a generalized scaly macular rash, eosinophilia of 13.4% (3500/mm<span class="elsevierStyleSup">3</span>) and alteration of aminotransferases. Treatment with prednisone 20<span class="elsevierStyleHsp" style=""></span>mg/day, dexchlorpheniramine, metoclopramide, paracetamol and metamizole was started. The patient showed poor progression with worsening of toxicoderma, fever, renal failure, and liver failure with a Model for End-stage Liver Disease (MELD) of 39 points and was therefore admitted to the intensive care unit (ICU). An echocardiogram was performed that showed no abnormalities and an abdominal CT that revealed significant hepatosplenomegaly and generalized lymphadenopathy. In order to rule out a lymphoproliferative syndrome, a fine needle aspiration of an axillary lymphadenopathy is performed, which rules out a tumour process. Viral, vascular, and autoimmune aetiology is also ruled out. After a skin biopsy showing a histopathology consistent with a "reaction to drugs" (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), it was regarded as DRESS syndrome and the dose of prednisone was increased to 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">In less than 12<span class="elsevierStyleHsp" style=""></span>h the patient suffered a metabolic coma and worsening of laboratory results, with bilirubin of 17<span class="elsevierStyleHsp" style=""></span>mg/dl, AST 2186<span class="elsevierStyleHsp" style=""></span>U/l, ALT 3348<span class="elsevierStyleHsp" style=""></span>U/l and prothrombin rate of 10%; therefore, it is included in "Zero-grade Emergency" (imminent emergency) for liver transplantation, with good post-transplant progression and without complications in the outpatient follow-up.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Our patient had a "definitive" DRESS syndrome according to the RegiSCAR-group Diagnosis Score for DRESS<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a>, with a total of 6 points: lymphadenopathy (+1), eosinophilia >10% (+2), skin <span class="elsevierStyleItalic">rash</span> (+1), 2 organs involved (+2). The patient received drugs such as vancomycin, piperacillin-tazobactam, rifampicin, or ibuprofen, described as causing DRESS syndrome in the literature reviewed<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>.</p><p id="par0035" class="elsevierStylePara elsevierViewall">While it is true that, given the multitude of drugs administered, it was impossible to determine the agent responsible, this clinical case alerts us to the serious adverse reactions of drugs and the need to thoroughly investigate previous drug intake. Adverse drug reactions pose a diagnostic challenge, especially in DRESS syndrome since its symptoms are delayed and mimic other diseases<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>. Early recognition of the syndrome and discontinuation of the causative agent is essential in this life-threatening reaction, so in the event of skin lesions and liver failure associated with drug intake, we must consider the diagnostic possibility of DRESS syndrome.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare that they have not received any type of funding.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Álvarez-García L, López Amor L, Escudero Augusto D. Trasplante hepático por hepatitis fulminante en un paciente con síndrome DRESS. Med Clin (Barc). 2021;156:363–364.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1023 "Ancho" => 2500 "Tamanyo" => 227299 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Skin <span class="elsevierStyleItalic">punch</span> (×200): epidermis with presence of spongiosis and minimal lymphocyte exocytosis. A perivascular and periadnexal lymphocytic infiltrate (arrows) can be seen in the underlying dermis, compatible with a cutaneous reaction to drugs. B) Liver ×100. Extensive necrosis, predominantly centrilobular, is identified, showing significant venular involvement (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The DRESS syndrome: a literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "P. Cacoub" 1 => "P. Musette" 2 => "V. Descamps" 3 => "O. Meyer" 4 => "C. Speirs" 5 => "L. Finzi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.amjmed.2011.01.017" "Revista" => array:6 [ "tituloSerie" => "Am J Med." 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