Letter to the Editor
Macrophage Activation Syndrome as a Severe Manifestation of Adult's Still's Disease. Hemophagocytic Cells in Ascites
Síndrome de activación macrofágica como complicación severa de la enfermedad de Still del adulto. Células hemofagocíticas en líquido ascítico
César Egües Dubuc
, Miren Uriarte Ecenarro, Nerea Errazquin Aguirre, Joaquín Belzunegui Otano
Corresponding author
Servicio de Reumatología, Hospital Universitario Donostia, San Sebastián, Guipúzcoa, Spain
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Hemophagocytic cells in ascites" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 816 "Ancho" => 1759 "Tamanyo" => 99596 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Líquido ascítico. 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Hemophagocytic Cells in Ascites" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "420" "paginaFinal" => "421" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "César Egües Dubuc, Miren Uriarte Ecenarro, Nerea Errazquin Aguirre, Joaquín Belzunegui Otano" "autores" => array:4 [ 0 => array:4 [ "nombre" => "César" "apellidos" => "Egües Dubuc" "email" => array:1 [ 0 => "tonoeguesdubuc@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Miren" "apellidos" => "Uriarte Ecenarro" ] 2 => array:2 [ "nombre" => "Nerea" "apellidos" => "Errazquin Aguirre" ] 3 => array:2 [ "nombre" => "Joaquín" "apellidos" => "Belzunegui Otano" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Reumatología, Hospital Universitario Donostia, San Sebastián, Guipúzcoa, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de activación macrofágica como complicación severa de la enfermedad de Still del adulto. Células hemofagocíticas en líquido ascítico" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 625 "Ancho" => 1347 "Tamanyo" => 84160 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Ascites. Red cell within macrophages is observed.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Macrophage activation syndrome (MAS) occurs in a minority of patients with adult Still's disease (ASD).<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> It can be the diseases’ first manifestation or be triggered by an infection or change in treatment.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Two cases are presented.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The first case is a 30-year-old woman with ASD, who came to the ER with a fever lasting for one week that did not improve with antibiotics, so she was admitted to the Rheumatology department. During admission, treatment with ceftriaxone and glucocorticoids (GC) mg/kg was started, and multiple complementary tests performed: blood culture positive for <span class="elsevierStyleItalic">pneumococcus</span> and positive serology for <span class="elsevierStyleItalic">cytomegalovirus</span>; analytically: hepatitis, elevated acute phase reactants (APR), hyperferritinemia, hypertriglyceridemia, thrombocytopenia, and anemia, in addition to splenomegaly in the computed tomography (CT).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Despite treatment, evolution was unfavorable, with persistent thrombocytopenia and declining ESR. With these data, we decided to perform a bone marrow biopsy (BMB), observing hemophagocytic cells (HC), diagnosing MAS and initiating treatment with cyclosporine (CSP) 5<span class="elsevierStyleHsp" style=""></span>mg/kg/day and 60<span class="elsevierStyleHsp" style=""></span>GC mg/day, with progressive improvement.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The second case is a 35-year-old woman who presented with fever, a sore throat, musculoskeletal pain, vomiting and rash for a week, diagnosed as the flu. Three days later she came to the ER because of persistent fever and rash, and the onset of lymphadenopathy, and was hospitalized. During admission, treatment was started with ceftriaxone and gentamicin; associated infection and autoimmunity was ruled out. Analytically she presented: anemia, elevated APR, hepatitis, hyperferritinemia, hypertriglyceridemia, impaired renal function and ascites. A CT was performed, which reported a systemic infection with lymphadenopathy and hepatosplenomegaly.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The initial suspicion was ASD, starting GC therapy at a dose of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day and presenting an initial improvement. Subsequently, the fever persisted and skin lesions progressed, added to respiratory and renal failure, which led to her transfer to the Intensive Care Unit (ICU) of our hospital. On admission to the ICU, we performed a BMB, ascites analysis, observing HC in both (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). She was diagnosed as MAS. Evolution was poor despite GC, so we added CSP 5<span class="elsevierStyleHsp" style=""></span>mg/kg/day, with partial improvement and anakinra 100<span class="elsevierStyleHsp" style=""></span>mg/day. The evolution was favorable, so she was transferred to the rheumatology department, where after a few days she was discharged.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Both conditions share several features, such as fever, hepatosplenomegaly, lymphadenopathy, hepatitis, coagulopathy and hyperferritinemia, and often are indistinguishable.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Pleuritis, acute respiratory distress syndrome and pancytopenia are more common in the MAS<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> and cutaneous and articular affection in ASD.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Leukopenia, thrombocytopenia and hypertriglyceridemia are not common in ASD, so these could serve as warning signs<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>; Additionally, hyperferritinemia is generally higher in MAS.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Most of the cases described in the literature have been treated with GC, immunoglobulins, CSP and biological drugs.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Both conditions share certain pathophysiologic characteristics such as the production factor tumor necrosis α and interleukins (IL), IL-1, IL-6, IL-8 and IL-18, with these being potential therapeutic targets.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The first patient probably presented MAS after a triggering infection. We hypothesize that the MAS in ASD can occur after a change in treatment or an infection; the most studied is the Epstein–Barr virus infection, but it has also been described<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3,6</span></a> in cases of cytomegalovirus.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Patient 2 presented HC in ascites; the first case was described in 2007 in a patient with liver cirrhosis and <span class="elsevierStyleItalic">Escherichia coli</span> infection.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In addition, cases described with HC in<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,9</span></a> pleural fluid and in cerebrospinal fluid.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of Interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors have no disclosures to make.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of Interest" ] 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: Egües Dubuc C, Uriarte Ecenarro M, Errazquin Aguirre N, Belzunegui Otano J. Síndrome de activación macrofágica como complicación severa de la enfermedad de Still del adulto. Células hemofagocíticas en líquido ascítico. Reumatol Clin. 2014;10:420–421.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 625 "Ancho" => 1347 "Tamanyo" => 84160 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Ascites. Red cell within macrophages is observed.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Reactive hemophagocytic syndrome in adult systemic disease: report of twenty-six cases and literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R. Dhote" 1 => "J. Simon" 2 => "T. Papo" 3 => "B. Detournay" 4 => "L. Sailler" 5 => "M.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 8 | 6 | 14 |
| 2024 October | 62 | 38 | 100 |
| 2024 September | 86 | 39 | 125 |
| 2024 August | 104 | 72 | 176 |
| 2024 July | 85 | 34 | 119 |
| 2024 June | 86 | 43 | 129 |
| 2024 May | 89 | 38 | 127 |
| 2024 April | 82 | 26 | 108 |
| 2024 March | 62 | 29 | 91 |
| 2024 February | 42 | 17 | 59 |
| 2024 January | 50 | 26 | 76 |
| 2023 December | 44 | 29 | 73 |
| 2023 November | 42 | 24 | 66 |
| 2023 October | 78 | 30 | 108 |
| 2023 September | 56 | 34 | 90 |
| 2023 August | 59 | 12 | 71 |
| 2023 July | 47 | 29 | 76 |
| 2023 June | 59 | 30 | 89 |
| 2023 May | 78 | 23 | 101 |
| 2023 April | 58 | 18 | 76 |
| 2023 March | 76 | 22 | 98 |
| 2023 February | 74 | 23 | 97 |
| 2023 January | 62 | 11 | 73 |
| 2022 December | 68 | 34 | 102 |
| 2022 November | 76 | 28 | 104 |
| 2022 October | 89 | 28 | 117 |
| 2022 September | 92 | 39 | 131 |
| 2022 August | 90 | 37 | 127 |
| 2022 July | 87 | 45 | 132 |
| 2022 June | 83 | 24 | 107 |
| 2022 May | 72 | 35 | 107 |
| 2022 April | 79 | 51 | 130 |
| 2022 March | 55 | 41 | 96 |
| 2022 February | 67 | 37 | 104 |
| 2022 January | 75 | 44 | 119 |
| 2021 December | 55 | 45 | 100 |
| 2021 November | 67 | 50 | 117 |
| 2021 October | 85 | 51 | 136 |
| 2021 September | 68 | 51 | 119 |
| 2021 August | 86 | 50 | 136 |
| 2021 July | 54 | 25 | 79 |
| 2021 June | 83 | 63 | 146 |
| 2021 May | 97 | 49 | 146 |
| 2021 April | 150 | 99 | 249 |
| 2021 March | 124 | 32 | 156 |
| 2021 February | 74 | 22 | 96 |
| 2021 January | 40 | 23 | 63 |
| 2020 December | 66 | 26 | 92 |
| 2020 November | 43 | 23 | 66 |
| 2020 October | 31 | 17 | 48 |
| 2020 September | 46 | 34 | 80 |
| 2020 August | 39 | 14 | 53 |
| 2020 July | 41 | 16 | 57 |
| 2020 June | 48 | 22 | 70 |
| 2020 May | 35 | 13 | 48 |
| 2020 April | 31 | 22 | 53 |
| 2020 March | 23 | 10 | 33 |
| 2020 February | 1 | 0 | 1 |
| 2018 May | 6 | 1 | 7 |
| 2018 April | 82 | 10 | 92 |
| 2018 March | 88 | 5 | 93 |
| 2018 February | 32 | 3 | 35 |
| 2018 January | 36 | 6 | 42 |
| 2017 December | 42 | 6 | 48 |
| 2017 November | 44 | 2 | 46 |
| 2017 October | 33 | 3 | 36 |
| 2017 September | 45 | 3 | 48 |
| 2017 August | 70 | 4 | 74 |
| 2017 July | 67 | 12 | 79 |
| 2017 June | 84 | 10 | 94 |
| 2017 May | 77 | 9 | 86 |
| 2017 April | 78 | 6 | 84 |
| 2017 March | 71 | 34 | 105 |
| 2017 February | 40 | 4 | 44 |
| 2017 January | 66 | 4 | 70 |
| 2016 December | 100 | 12 | 112 |
| 2016 November | 85 | 4 | 89 |
| 2016 October | 121 | 6 | 127 |
| 2016 September | 165 | 15 | 180 |
| 2016 August | 111 | 9 | 120 |
| 2016 July | 48 | 7 | 55 |
| 2015 December | 2 | 0 | 2 |
| 2015 September | 1 | 0 | 1 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 30 | 8 | 38 |
| 2015 June | 55 | 12 | 67 |
| 2015 May | 107 | 17 | 124 |
| 2015 April | 59 | 7 | 66 |
| 2015 March | 45 | 12 | 57 |
| 2015 February | 50 | 6 | 56 |
| 2015 January | 86 | 23 | 109 |
| 2014 December | 68 | 13 | 81 |
| 2014 November | 84 | 17 | 101 |
| 2014 October | 33 | 14 | 47 |
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