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She was being monitored by the rheumatology department because of generalised arthromyalgia, positive lupus anticoagulant and Raynaud's phenomenon. She had abdominal pain (mainly in the right iliac fossa (RIF)) for one week that required admission to internal medicine. Abdominal MRI showed mesenteric fibrosis/panniculitis and PET/CT showed a slight increase of metabolism in RIF without neoplastic involvement. Her autoimmunity study was negative and IgG4<span class="elsevierStyleHsp" style=""></span>>194<span class="elsevierStyleHsp" style=""></span>mg/dl. In her mesenteric fat biopsy, a perivascular inflammatory infiltrate and occasional plasma cells with positivity for IgG4 are observed. Corticoid treatment was initiated with 40<span class="elsevierStyleHsp" style=""></span>mg/day prednisone for one month with clinical improvement and a subsequent gradual reduction.</p><p id="par0020" class="elsevierStylePara elsevierViewall">When she reached a dose of 10<span class="elsevierStyleHsp" style=""></span>mg/day she relapsed, so we decided to start azathioprine at doses of 100<span class="elsevierStyleHsp" style=""></span>mg daily. An abdominal MRI showed radiological improvement of mesenteric panniculitis in addition to the disappearance of pleural effusion and free abdominal fluid.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Two years later she was treated with 10<span class="elsevierStyleHsp" style=""></span>mg prednisone daily and methotrexate 15<span class="elsevierStyleHsp" style=""></span>mg weekly (azathioprine was changed because of side effects); she maintained mild arthromyalgia but no abdominal pain and IgG4 was 89<span class="elsevierStyleHsp" style=""></span>mg/dl.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Case 2: 46-year-old female patient with a history of long-term arthromyalgia. She was presented with subacute pain in lower abdomen of two months’ evolution, with change in depositional rate and loss of 3<span class="elsevierStyleHsp" style=""></span>kg. Abdominal CT and MRI scan, colonoscopy and exploratory laparotomy were performed, which showed stenosing neoformation in the transverse colon. A wide right hemicolectomy was performed with a block resection of part of the anterior abdominal wall. The pathological anatomy of the sample was compatible with sclerosing disease (inflammatory pseudotumour) associated with IgG4 disease. Analytical results showed normal IgG4. After the intervention the patient was asymptomatic and the subsequent PET/CT control found nothing significant. She has not suffered relapses and has not needed treatment in four years.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Case 3: A 28-year-old female patient from Ecuador with a history of vitiligo and autoimmune hypothyroidism (positive anti-TPO). Being monitored by the rheumatology department due to Sjogren's syndrome, positive ANA (1/160), with abnormal sialometry. She visited the emergency room because of pleuritic chest pain and progressive dyspnoea of one month of evolution. Chest X-ray showed a left pleural effusion. In pleural fluid, there was a predominance of leukocytes with 43% of eosinophils. Analytical results showed eosinophilia, polyclonal hypergammaglobulinaemia and ANA 1/160, with IgG4<span class="elsevierStyleHsp" style=""></span>>194<span class="elsevierStyleHsp" style=""></span>mg/dl. Serology and parasitic cultures were negative. The pleural biopsy showed a predominantly plasmocitary infiltrate with more than 60% positive IgG4 plasmocytes. Pleural effusion is thus associated with IgG4 disease, as is peripheral eosinophilia. Treatment was started with prednisone at 0.5<span class="elsevierStyleHsp" style=""></span>mg/kg daily with good response.</p><p id="par0040" class="elsevierStylePara elsevierViewall">When a reduction of corticoids is initiated, she experienced relapse, so azathioprine 50<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h was introduced with good results.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Two years later she was asymptomatic with IgG4 at 100<span class="elsevierStyleHsp" style=""></span>mg/dl, she was maintained with the same dose of azathioprine in addition to 1.25<span class="elsevierStyleHsp" style=""></span>mg prednisone daily.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The three clinical cases outlined involve different manifestations of the disease: mesenteric fibrosis related to IgG4 disease, sclerosing disease related to IgG4 disease and pleural effusion secondary to IgG4 disease. This shows how IgG4 disease can affect any body organ. Clinical, histopathological and serological criteria are required for its diagnosis, which our patients met (cases 1 and 3 complied with all three and case 2 with the clinical and histopathological criteria). Differential diagnosis is necessary with other diseases such as neoplasms, infectious and autoimmune processes.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Its treatment is based on the use of glucocorticoids; although immunomodulators and biological drugs are also used in case they are insufficient to control the disease (as in cases 1 and 3).<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</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: Lorente Ó, Rueda A, Campos C. Enfermedad relacionada con la inmunoglobulina G4: variabilidad del espectro clínico. Med Clin (Barc). 2019;153:221–222.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Current approach to the diagnosis of IgG4-related disease – combination of comprehensive diagnostic and organ-specific criteria" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "H. Umehara" 1 => "K. Okazaki" 2 => "T. Nakamura" 3 => "T. Satoh-Nakamura" 4 => "A. Nakajima" 5 => "M. 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Journal Information
Vol. 153. Issue 5.
Pages 221-222 (September 2019)
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Vol. 153. Issue 5.
Pages 221-222 (September 2019)
Letter to the Editor
Disease related to immunoglobulin G4: Clinical variability
Enfermedad relacionada con la inmunoglobulina G4: variabilidad del espectro clínico
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