Letter to the Editor
Familial Mediterranean Fever Treated With Anakinra: A Case Report
Fiebre mediterránea familiar en tratamiento con anakinra: a propósito de un caso
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"autoresLista" => "Francisca Aguiar, Iva Brito" "autores" => array:2 [ 0 => array:2 [ "nombre" => "Francisca" "apellidos" => "Aguiar" ] 1 => array:2 [ "nombre" => "Iva" "apellidos" => "Brito" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574317300242?idApp=UINPBA00004M" "url" => "/21735743/0000001300000002/v2_201703100919/S2173574317300242/v2_201703100919/en/main.assets" ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Familial Mediterranean Fever Treated With Anakinra: A Case Report" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "120" "paginaFinal" => "121" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Javier Espíldora-Hernández, Manuel Abarca-Costalago" "autores" => array:2 [ 0 => array:4 [ "nombre" => "Javier" "apellidos" => "Espíldora-Hernández" "email" => array:1 [ 0 => "javiviespil@hotmail.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" => "Manuel" "apellidos" => "Abarca-Costalago" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Medicina Interna, Hospital Universitario Virgen de la Victoria, Málaga, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Medicina Interna, Unidad Enfermedades Autoinmunes, Hospital Universitario Virgen de la Victoria, Málaga, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Fiebre mediterránea familiar en tratamiento con anakinra: a propósito de un caso" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The treatment of familial Mediterranean fever (FMF) has several objectives: achieving relief from the symptoms, preventing and managing acute attacks, and avoiding the development of secondary amyloidosis. Classically, the drug of choice is colchicine. Now, however, we also have interleukin 1β (IL-1β) inhibitors.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 37-year-old man, with no familial history or known allergies, who underwent appendectomy and had Ménière's disease in right ear. He began in 1996, at the age of 17 years, with fevers that peaked at up to 40<span class="elsevierStyleHsp" style=""></span>°C, abdominal pain in right iliac fossa and occasional vomiting. These episodes lasted 2 or 3 days. He also customarily complained of pleuritic pain, but pleural effusion was never detected.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Since 2005, he also mentioned pain with swelling in his hands. This responded partially to treatment with ibuprofen, as did the occasional arthralgia in knees and ankles. He underwent genetic testing for FMF, which detected an alteration on exon 10: 2082G>A mutation in a homozygote, leading to a change to Met694Ile. He began therapy with colchicine at a dose of 2<span class="elsevierStyleHsp" style=""></span>mg every 24<span class="elsevierStyleHsp" style=""></span>h, and the number of attacks decreased.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In 2013, the number of episodes increased, requiring maximum doses of colchicine, with no clear improvement. Physical examination revealed a rigid, board-like abdomen, with no other noteworthy finding. A blood test only showed a mean corpuscular volume of 110<span class="elsevierStyleHsp" style=""></span>fL, fibrinogen 366<span class="elsevierStyleHsp" style=""></span>g/L and γ-glutamyl transpeptidase 79<span class="elsevierStyleHsp" style=""></span>IU/L. Systematic urinalysis, serological tests, a chest radiograph and abdominal ultrasound provided no evidence of disease.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In December 2014, the patient began with subcutaneous anakinra at a dose of 100<span class="elsevierStyleHsp" style=""></span>mg daily, and has continued to take colchicine. Since that time, he has remained asymptomatic.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Resistance to colchicine is defined as 2 or more attacks each month while receiving the maximum dose. This occurs in 5%–10% of patients with FMF, although in some cases, it may be due to poor treatment adherence.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> True cases of colchicine resistance are usually found in patients who are homozygous for the M694V.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The mechanism of action of agents that inhibit IL-1β in the etiology and pathogenesis of this disease is well known. The protein, pyrin, is a fundamental part of the so-called inflammasome, the intracellular organelle necessary for the expression of IL-1β<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,4</span></a> which, ultimately, causes the patients’ symptoms. The IL-1β inhibitors most widely used are anakinra, rilonacept and canakinumab.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The biologic agent chosen for our patient was anakinra prescribed for compassionate use, as it has achieved good responses in other inflammatory diseases, such as rheumatoid arthritis and juvenile idiopathic arthritis.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5,6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Our patient continued to receive colchicine associated with the biologic therapy. There is no consensus on that point in this respect: logically, it would be justifiable to discontinue it, but some authors recommend maintaining it as an adjuvant therapy.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In this context, the satisfactory response to the treatment was a sufficiently good reason to decide not to change the initial therapeutic proposal.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The uses of anakinra approved in Spain include the treatment of symptoms and signs of rheumatoid arthritis, in combination with methotrexate, in those patients who have not responded well.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> The secondary effects of this drug, such as pruritus and erythema, are infrequent and appear during the first 4 weeks. There have been no reports of deaths due to severe opportunistic infection.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Since 2006, 22 studies have been published involving 64 patients from 10 different countries. In all, 76.5% of those treated with this drug achieve a complete response. It has been ineffective in only 3 patients, and the remainder had a partial response.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Espíldora-Hernández J, Abarca-Costalago M. Fiebre mediterránea familiar en tratamiento con anakinra: a propósito de un caso. Reumatol Clin. 2017;13:120–121.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "IL-1β biological treatment of familial mediterranean fever" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "A. Soriano" 1 => "E. Verecchia" 2 => "A. Afeltra" 3 => "R. Landolfi" 4 => "R. 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| 2024 October | 48 | 20 | 68 |
| 2024 September | 53 | 12 | 65 |
| 2024 August | 58 | 35 | 93 |
| 2024 July | 65 | 32 | 97 |
| 2024 June | 46 | 35 | 81 |
| 2024 May | 64 | 42 | 106 |
| 2024 April | 55 | 17 | 72 |
| 2024 March | 40 | 45 | 85 |
| 2024 February | 41 | 20 | 61 |
| 2024 January | 36 | 29 | 65 |
| 2023 December | 22 | 26 | 48 |
| 2023 November | 49 | 24 | 73 |
| 2023 October | 35 | 26 | 61 |
| 2023 September | 63 | 41 | 104 |
| 2023 August | 38 | 10 | 48 |
| 2023 July | 31 | 23 | 54 |
| 2023 June | 37 | 28 | 65 |
| 2023 May | 39 | 22 | 61 |
| 2023 April | 27 | 7 | 34 |
| 2023 March | 67 | 24 | 91 |
| 2023 February | 43 | 30 | 73 |
| 2023 January | 31 | 34 | 65 |
| 2022 December | 66 | 29 | 95 |
| 2022 November | 32 | 23 | 55 |
| 2022 October | 75 | 32 | 107 |
| 2022 September | 38 | 33 | 71 |
| 2022 August | 20 | 43 | 63 |
| 2022 July | 26 | 37 | 63 |
| 2022 June | 24 | 26 | 50 |
| 2022 May | 32 | 34 | 66 |
| 2022 April | 39 | 49 | 88 |
| 2022 March | 31 | 36 | 67 |
| 2022 February | 36 | 38 | 74 |
| 2022 January | 41 | 38 | 79 |
| 2021 December | 23 | 34 | 57 |
| 2021 November | 32 | 40 | 72 |
| 2021 October | 54 | 55 | 109 |
| 2021 September | 36 | 40 | 76 |
| 2021 August | 32 | 40 | 72 |
| 2021 July | 17 | 28 | 45 |
| 2021 June | 17 | 34 | 51 |
| 2021 May | 49 | 36 | 85 |
| 2021 April | 48 | 65 | 113 |
| 2021 March | 55 | 33 | 88 |
| 2021 February | 26 | 15 | 41 |
| 2021 January | 23 | 23 | 46 |
| 2020 December | 39 | 23 | 62 |
| 2020 November | 25 | 22 | 47 |
| 2020 October | 18 | 15 | 33 |
| 2020 September | 45 | 22 | 67 |
| 2020 August | 19 | 21 | 40 |
| 2020 July | 13 | 12 | 25 |
| 2020 June | 34 | 23 | 57 |
| 2020 May | 17 | 16 | 33 |
| 2020 April | 18 | 12 | 30 |
| 2020 March | 13 | 2 | 15 |
| 2019 February | 1 | 0 | 1 |
| 2018 September | 0 | 1 | 1 |
| 2018 May | 3 | 0 | 3 |
| 2018 April | 52 | 8 | 60 |
| 2018 March | 73 | 14 | 87 |
| 2018 February | 61 | 7 | 68 |
| 2018 January | 13 | 4 | 17 |
| 2017 December | 60 | 8 | 68 |
| 2017 November | 18 | 9 | 27 |
| 2017 October | 21 | 9 | 30 |
| 2017 September | 22 | 11 | 33 |
| 2017 August | 28 | 7 | 35 |
| 2017 July | 29 | 13 | 42 |
| 2017 June | 35 | 9 | 44 |
| 2017 May | 39 | 13 | 52 |
| 2017 April | 50 | 20 | 70 |
| 2017 March | 27 | 24 | 51 |
| 2017 February | 0 | 2 | 2 |
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