Letter to the Editor
Symmetric polyarthritis in a patient with multicentric reticulohistiocytosis
Poliartritis simétrica en un paciente con reticulohistiocitosis multicéntrica
M. Ángeles Contreras Blasco
, Patricia López Viejo
Corresponding author
Servicio de Reumatología, Hospital Universitario Rey Juan Carlos, Móstoles , Madrid, Spain
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Ángeles Contreras Blasco, Patricia López Viejo" "autores" => array:2 [ 0 => array:4 [ "nombre" => "M. Ángeles" "apellidos" => "Contreras Blasco" "email" => array:1 [ 0 => "macblasco@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Patricia" "apellidos" => "López Viejo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Reumatología, Hospital Universitario Rey Juan Carlos, Móstoles , Madrid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Poliartritis simétrica en un paciente con reticulohistiocitosis multicéntrica" ] ] "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" => 732 "Ancho" => 976 "Tamanyo" => 94443 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Arthritis in wrists, tenosynovitis of both flexor carpi, most evident in the left hand, and fleshy papulonodular lesions on the back of several fingers.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 44-year-old woman with a longstanding autoimmune hypothyroidism, multicentric reticulohistiocytosis (MRH) diagnosed through skin biopsy in 2003 and erosive rheumatoid arthritis (RA) with positive rheumatoid factor (RF) and anti-cyclic citrullinated peptide antibodies (anti-CCP Ab).</p><p id="par0010" class="elsevierStylePara elsevierViewall">The MRH is a rare systemic granulomatous disease characterized by skin involvement in the form of papulonodular lesions, mainly in the hands and face, and a symmetric, erosive and progressive polyarthritis, which mainly affects the distal interphalangeal joints (DIPJ) in the hands, followed by knees, shoulders, wrists and hips. It is associated with other autoimmune diseases, such as diabetes mellitus, hypothyroidism, Sjogren's syndrome, systemic sclerosis, dermatomyositis, systemic lupus erythematosus, celiac disease and primary biliary cirrhosis. However, its RA association is exceptional.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient consulted due to a 9-year progression symmetric polyarthritis in the fingers, carpi, wrists and knees, with negative antinuclear antibodies (ANA) and HLA-B27, RF 30<span class="elsevierStyleHsp" style=""></span>IU/ml and anti-CCP Ab of 86<span class="elsevierStyleHsp" style=""></span>IU/ml, which was diagnosed as RA or MRH arthritis in 2005. Throughout these years, the patient has received several treatments (NSAIDs, oral corticosteroids, methotrexate orally and subcutaneously in a maximum dose of 20<span class="elsevierStyleHsp" style=""></span>mg weekly, with poor gastrointestinal tolerability by both routes, hydroxychloroquine at a maximum dose of 400<span class="elsevierStyleHsp" style=""></span>mg daily, sulfasalazine at a maximum dose of 2<span class="elsevierStyleHsp" style=""></span>g daily, leflunomide in a dose of 20<span class="elsevierStyleHsp" style=""></span>mg daily and weekly alendronate), with no arthritis or skin lesions response. Arthritis in wrists, several DIPJ in hands and the right knee, flexor carpi tenosynovitis and fleshy papulonodular lesions on the back of several fingers were observed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Normal complete blood count, ESR 42<span class="elsevierStyleHsp" style=""></span>ml/first hour, complete biochemistry with normal muscle enzymes, CRP 1.8<span class="elsevierStyleHsp" style=""></span>mg/dl, normal thyroid function tests, normal concentrations of angiotensin converting enzyme and tumour markers (CEA, CA 125, CA 19-9, CA 15-3, AFP), RF negative, positive anti-CCP Ab 80<span class="elsevierStyleHsp" style=""></span>IU/ml and ANA titre 1/80, with no specific pattern, negative for anticentromere antibodies, anti-native DNA, anti-Sm, anti-RNP, anti-Scl-70, anti-Jo-1, anti-Ro, anti-La, antimitochondrial, anti-SMA, ANCA and antithyroglobulin and positive antimicrosomal antibodies (1100<span class="elsevierStyleHsp" style=""></span>IU/ml). Negative for cryoglobulins. Titres of G, A and M immunoglobulins were normal as well as the complement (C3 and C4) and beta-2-microglobulin. Serologies for hepatitis B and C and HIV were negative, as well as the QuantiFERON<span class="elsevierStyleSup">®</span> test. The contrast CT scan of thorax, abdomen and pelvis, gynaecological examination and mammography were normal. Although no erosions were found on hand and foot radiographs, the right wrist and hand gadolinium MRI showed active bone erosion in the distal and palmar edge of the radius, besides an important synovitis, which captured contrast in the carpus and the distal radioulnar joint, and tenosynovitis in the second, third and fourth dorsal compartment of the wrist and around the flexor tendons of the second, third and fifth finger. Treatment was initiated with etanercept at a dose of 50<span class="elsevierStyleHsp" style=""></span>mg weekly, subcutaneously. After 12 weeks, polyarthritis had significantly improved both clinically and analytically, while skin lesions remained unchanged.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Our patient met the classification criteria for rheumatoid arthritis ACR 1987,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> as well as the classification and diagnosis criteria for rheumatoid arthritis ACR/EULAR 2010.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">MRH's arthritis has its maximum aggressiveness in the early years and, in up to half of patients, there is a rapid progression to severe or mutilating erosive arthropathy, characterized by a lesion called “telescoping or accordion fingers”. It differs from RA in that the destructive changes in the DIP joints of the hands are frequent, the RF is often negative and it is not accompanied by joint space narrowing or juxtaarticular osteoporosis on radiographs.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Although there is no curative treatment for MRH, the start of it should be early in order to prevent skin and joint sequelae, which can lead to the need of orthopaedic surgery. NSAIDs have been used in mild types. Systemic corticosteroids alone have been used to control joint symptoms, although they do not induce disease remission. They have also been used in combination regimens with antimalarial agents, methotrexate or cyclophosphamide, with satisfactory results. Treatment with cyclosporine, chlorambucil, azathioprine and leflunomide has been applied with good results, both articular and cutaneous. There are contrasting cases of response to bisphosphonates, mainly with alendronate and zoledronate. In difficult cases, TNF-α antagonists such as etanercept,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> infliximab<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> and adalimumab<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> have been used, as well as IL-6 inhibitors, such as tocilizumab.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> In our case, RA responded favourably to treatment with a TNF-α antagonist, but there was no response in MRH skin lesions.</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: Contreras Blasco MÁ, López Viejo P. Poliartritis simétrica en un paciente con reticulohistiocitosis multicéntrica. Med Clin (Barc). 2015;145:508–509.</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" => 732 "Ancho" => 976 "Tamanyo" => 94443 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Arthritis in wrists, tenosynovitis of both flexor carpi, most evident in the left hand, and fleshy papulonodular lesions on the back of several fingers.</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" => "Multicentric reticulohistiocytosis associated with rheumatoid arthritis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "D. 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