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It is a severe entity characterised by the triad of rheumatoid arthritis, neutropenia, and splenomegaly. The incidence of Felty's syndrome for a patient diagnosed with rheumatoid arthritis is less than 1%. Characteristically, rheumatoid arthritis first develops, with joint deformities and erosions, and years pass until neutropenia appears. In very few cases neutropenia arises before the arthritis.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a patient with a history of chronic asymptomatic neutropenia for the past 10 years, with regular follow-up in the Haematology outpatients department, who was admitted for febrile neutropenia and was diagnosed with rheumatoid arthritis with associated Felty's syndrome during admission.</p><p id="par0015" class="elsevierStylePara elsevierViewall">A 72-year-old male, an active smoker of 3 packs a day, chronic moderate drinker, who entered our site due to fever. As a notable background, he presented an asymptomatic oscillating chronic neutropenia which had progressed for the past 10 years, with: negative HIV, HBV and HCV serology study; autoimmunity study with positive antinuclear antibodies (ANA) titre of 1:320; bone marrow aspirate with an increase in the erythroblast series (45%), moderate dyserythropoiesis (11%) and a slight decrease in the granulocyte series (48%), morphologically normal; normal cytogenetics and immunophenotype of the lymphocytes without phenotypic abnormalities. During the chronic neutropenia study, a thoracoabdominal CT scan was performed, which showed a homogeneous enlarged spleen of 19<span class="elsevierStyleHsp" style=""></span>cm.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Upon arrival at our site, the patient reported having had a fever for the past 24<span class="elsevierStyleHsp" style=""></span>h, with polyarthralgia and weight loss in the last 2 months (10<span class="elsevierStyleHsp" style=""></span>kg total weight loss). He did not report any infectious focus. The physical examination revealed annular erythematous lesions on the trunk and extremities, with pain on palpation of the metacarpophalanges and multiple oral thrush. In the initial analysis, pancytopenia was highlighted: haemoglobin 11.7<span class="elsevierStyleHsp" style=""></span>g/dL, platelets 111,000, leukocytes 1600/μL (12% neutrophils, 58% lymphocytes), coagulopathy with INR 1.40, hypoalbuminemia 3.3<span class="elsevierStyleHsp" style=""></span>g/dL and PCR 130.2. The chest X-ray showed no infiltrates or condensations. It was targeted as low-risk febrile neutropenia (MASCC index of 21 points), so he was admitted onto the Internal Medicine ward with amoxicillin-clavulanate empirical antibiotic therapy i/v ciprofloxacin. During admission, the patient remained febrile, without a clear infectious focal point and with negative cultures, so the antibiotic coverage was extended to i/v piperacillin-tazobactam. An inter-consultation with Haematology was carried out to perform bone marrow aspiration. Hypercellular bone marrow was observed with the presence of the three hematopoietic series, with granulocytic hyperplasia with a predominance of immature forms, without lymphoid infiltration. Neutropenia persisted during admission, so colony-stimulating factors were initiated, with the disappearance of fever and a slight increase in total neutrophils. Due to the presence of joint pain and weight loss in the last 2 months, a rheumatological study was requested. The following results stood out: rheumatoid factor 103<span class="elsevierStyleHsp" style=""></span>IU/mL, anti-cyclic citrullinated peptide (anti-CCP) 1188<span class="elsevierStyleHsp" style=""></span>U/mL, positive ANA moderate 3–4 homogeneous speckled and nucleolar, positive moderate anti-cytoplasmic antibodies. Negative anti-DNA, anti-Ro, anti-La, anti-Sm, anti-RNP and anti-intrinsic factor. Due to the suspicion of Felty's syndrome of atypical onset, treatment was started with systemic corticosteroids and methotrexate, presenting progressive clinical improvement, cessation of joint pain, improvement of skin lesions and disappearance of the neutropenia.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Few cases are published describing Felty's syndrome in which neutropenia precedes the development of rheumatoid arthritis, and in the literature review, there were no cases describing a neutropenia that had started such a long time prior to the onset of rheumatoid arthritis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In the majority of cases described, neutropenia in Felty's syndrome arises after a long course of rheumatoid arthritis, with the manifestation of joint deformities and erosions, and the subsequent onset of neutropenia. Our case is an example of atypical Felty's syndrome because of the neutropenia appearing first, before any development of deforming rheumatoid arthritis that characterises Felty's syndrome.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Due to this case of neutropenia with associated rheumatoid arthritis, we would also have to consider the diagnostic possibility of T-cell lymphoma, which presents with severe neutropenia associated with recurrent bacterial infections, with some cases with hepatosplenomegaly. In our case, this was ruled out due to the absence of the monoclonal expansion of lymphocytes with a characteristic immunophenotype (CD3+, CD4−, CD8+, CD16+, CD28−, and CD57+) in peripheral blood and bone marrow.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">For this reason, when facing any patient with continued neutropenia without known aetiology and without associated arthritis, Felty's syndrome with atypical presentation should be considered and the rheumatological study with rheumatoid factor and anti-PCC antibodies should be started.</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: Baldà Masmiquel M, Redondo Parejo L, Pla Salas X. Síndrome de Felty atípico. Med Clin (Barc). 2020;154:420–421.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Felty's syndrome as an initial presentation of rheumatoid arthritis: a case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "D. Chavalitdhamrong" 1 => "A. Molovic-Kokovic" 2 => "A. 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Zea" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.reuma.2011.12.014" "Revista" => array:5 [ "tituloSerie" => "Reumatol Clin" "fecha" => "2012" "volumen" => "8" "paginaInicial" => "303" "paginaFinal" => "382" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000015400000010/v1_202005290855/S2387020620301406/v1_202005290855/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000015400000010/v1_202005290855/S2387020620301406/v1_202005290855/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020620301406?idApp=UINPBA00004N" ]
Journal Information
Vol. 154. Issue 10.
Pages 420-421 (May 2020)
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Vol. 154. Issue 10.
Pages 420-421 (May 2020)
Letter to the Editor
Atypical Felty's syndrome
Síndrome de Felty atípico
Visits
6
Maria Baldà Masmiquel
, Laura Redondo Parejo, Xavier Pla Salas
Corresponding author
Servicio de Medicina Interna, Consorci Hospitalari de Vic, Vic, Barcelona, Spain
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