Letter to the Editor
Löfgren syndrome: Report of two cases
Síndrome de Löfgren: presentación de dos casos
Mariuxi Burgos, Jonathan Franco
, Angel Charte
Corresponding author
Departamento de Medicina Interna, Hospital Universitario Quirón Dexeus, Barcelona, Spain
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Chest X-ray with evidence of bilateral hilar lymphadenopathies (yellow arrows).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Löfgren's syndrome is a rare presentation of sarcoidosis, which is usually acute with a typical triad of arthritis, erythema nodosum, and hilar lymphadenopathies. Treatment is usually symptomatic, with non-steroidal anti-inflammatory drugs (NSAIDs), as it usually has spontaneous remission. However, sometimes it requires immunosuppressant drugs.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Special care must be taken to recognize the clinical manifestations of this entity, since they can take atypical forms.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report two cases of Löfgren's syndrome, with different clinical manifestations. The first case is a 50 year-old male patient without medical or surgical history of interest, who went to the emergency department with a 3 week history of migratory arthritis of feet and hands, predominantly bilateral malleolar, associated with fever. Given the suspicion of Löfgren's syndrome, lab tests were requested, which showed elevated acute phase reactants (CRP and ESR), as well as increased angiotensin converting enzyme (ACE) values. The chest X-ray showed bilateral hilar lymphadenopathies (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), and the chest computed tomography (CT) detected the presence of hilar, mediastinal and paratracheal lymphadenopathies. Ibuprofen treatment was started for 3 weeks, with resolution of the symptoms.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second case is a 45-year-old male with a history of smoking. The patient came to the emergency department for a 2 week history of predominantly postprandial fever, associated with cough, arthritis in the malleoli, asthenia, adynamia and general malaise. A few days before, the patient went to another hospital, where they performed laboratory tests that showed elevated inflammatory response markers (CRP), and where he was administered NSAIDs and antibiotic therapy. In the absence of improvement, he came to our hospital. Upon arrival, the physical examination showed increased respiratory rate without changes in the vesicular breath sounds. The chest X-ray showed an image compatible with bilateral hilar lymphadenopathy. Ancillary tests were completed: Elevated levels of ACE, at 116 (2 times the normal limit) and chest CT with findings of bilateral micronodular pattern and bilateral and symmetric hilar and mediastinal lymphadenopathy. Respiratory function tests showed an obstructive pattern. An endoscopic ultrasound-guided Trucut biopsy of the mediastinal lymphadenopathy and pulmonary micronodules was performed, which confirmed the presence of non-caseating granulomas and excluded differential diagnoses, including tuberculosis. Treatment with prednisone was started at 0.5<span class="elsevierStyleHsp" style=""></span>mg/kg/day with a good symptomatic response in the lungs. Corticosteroids were gradually discontinued, with a good response after withdrawal.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Löfgren's syndrome is considered a self-limited condition, characterized by the triad of bilateral pulmonary hilar lymphadenopathy, erythema nodosum, and arthritis, mainly in large joints. Sarcoidosis occurs as Löfgren in 9–35% of cases; epidemiologically it is more common in young white women, and rarer in black people.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Acute arthritis in this syndrome usually affects the tibiofibular and tibiotarsal joints bilaterally; is usually self-limited, but approximately 30% of patients show a persistent course with symptoms exceeding a 2 year course. Chest lymph nodes typically occur at bilateral hilar and right paratracheal level; they show a tendency to resolve within weeks (90% of cases) and there is rarely persistence and progression.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> With respect to atypical presentations, they can differ between men and women, with the presence of erythema nodosum predominantly in women and significant ankle arthritis without erythema nodosum in men.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The indications for treatment are determined by the symptoms and the level of systemic involvement. Löfgren's syndrome is generally self-limiting, and treatment usually includes NSAIDs for symptomatic control of arthritis.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Glucocorticoids improve symptoms and lung function; however, there is no evidence regarding having an impact on its long-term progression. Methotrexate has been shown to be useful and well tolerated in patients with corticosteroid-resistant pulmonary, muscular, and cutaneous sarcoidosis. For the control of cytokine production, the usefulness of tumour necrosis factor antagonists has been assessed; apparently, infliximab could join the therapeutic arsenal for extrathoracic sarcoidosis.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion, Löfgren's syndrome shows typical and atypical clinical characteristics, and clinicians must identify them in time to avoid delays in its diagnosis and treatment, which could compromise the patient's quality of life. Likewise, there are currently various treatments, such as the use of non-steroidal anti-inflammatory drugs in cases of joint involvement without respiratory involvement, and the use of systemic corticosteroids when there is evidence of severe symptoms or target organ involvement.</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: Burgos M, Franco J, Charte A. Síndrome de Löfgren: presentación de dos casos. Med Clin (Barc). 2020;155:321–322.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 826 "Ancho" => 900 "Tamanyo" => 62963 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical case 1. Chest X-ray with evidence of bilateral hilar lymphadenopathies (yellow arrows).</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" => "Current and emerging pharmacological treatments for sarcoidosis: a review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "S.H. Beegle" 1 => "K. Barba" 2 => "R. 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