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Letter to the Editor
Schnitzler's syndrome: A case report
Síndrome de Schnitzler: presentación de un caso
Alexandra María Aceituno Caño
Corresponding author
Alexac265@gmail.com

Corresponding author.
, Esteban Alessandro Vogt Sánchez, Laura León Ruiz
Internal Medicine Department, Torrecárdenas University Hospital, Almería, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Schnitzler&#39;s syndrome is a rare disabling disorder characterized by a chronic urticarial rash and monoclonal gammopathy&#46; It was initially described by Liliane Schnitzler&#44; a French dermatologist&#44; who was the first clinician that recognized this particular combination&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> This syndrome is under-diagnosed because many physicians do not know about it&#44; the diagnostic criteria are well established&#44; however&#44; the pathogenesis remains unknown&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Schnitzler&#39;s syndrome is considered a potential autoinflammatory disorder&#44; clinical researchers are focused on the role of proinflammatory cytokines&#44; interleukin-1 &#40;IL-1&#41; in particular&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The main clinical findings are chronic urticaria&#44; periodic fever and musculoskeletal symptoms&#44; the frequency of urticarial eruptions differs between patients&#44; but in most cases&#44; the rash is continuously present&#44; sparing palms&#44; soles&#44; head and neck and it is histamine-independent&#44; so antihistamines are not effective&#46; Recurrent fever is the second most common symptom&#44; the frequency of the episodes also differs between patients&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">We describe a 65-year-old man with arterial hypertension&#44; who has been suffering&#44; for twelve years&#44; daily outbreaks of urticariform non pruritic lesions in chest and both limbs&#46; He was evaluated by dermatologists being cataloged of chronic idiopathic urticaria&#44; having only a partial remission of symptoms with high dose of corticosteroids&#46; In 2014&#44; he associated recurrent febrile episodes&#44; generalized osteoarticular pain&#44; neutrophilic leukocytosis and paraproteinemia in laboratory tests&#46; A skin biopsy and a genetic study were made rulling out autoinflammatory diseases and vasculitis&#46; He also was evaluated and followed by hematologists and rheumatologists&#46; In 2017&#44; he associated weight loss and arthralgias&#44; being referred to Internal Medicine consultation&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The physical examination highlighted anular erythematous and maculopapular non-confluent lesions in chest and upper limbs&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In the blood tests there were leukocytosis with neutrophilia&#44; C-reactive protein &#40;CRP&#41; of 12<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and globular sedimentation rate &#40;ESR&#41; of 79<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46; Autoimmunity&#44; rheumatoid factor and vascular endothelial growth factor &#40;VEGF&#41; were negative&#44; complement was normal&#46; A monoclonal band in the gamma region&#44; IgM 677<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#44; was observed in the proteinogram&#46; Bone sclerosis is seen on the dorsal-lumbar spine and pelvis radiographs and generalized subcentometric adenopathies were found in the thoracoabdominal scanner&#46; The electroneurogram described a polyneuropathy&#46; Bone marrow biopsy was performed with a negative cytogenetic study for lymphoproliferative diseases&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The combination of chronic urticarial rash&#44; monoclonal IgM gammopathy&#44; recurrent fever&#44; bone sclerosis and leukocytosis&#44; allowed us to make the diagnosis of Schnitzler&#39;s syndrome&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">There are several pathologies that can occur with monoclonal gammopathy and chronic urticaria&#44; including autoimmune&#44; hematological&#44; infectious or autoinflammatory diseases&#44; so diagnostic tests are used to exclude these conditions&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> There is no specific test for Schnitzler&#39;s syndrome&#44; the diagnosis is made when patients meet Strasbourg criteria&#44; which include&#58; two obligate criteria and six minor criteria&#46; The diagnosis is considered definite if the two obligate criteria &#40;recurrent&#44; nonpruritic urticaria and monoclonal gammopathy&#44; either IgM or IgG&#41; and&#44; at least&#44; two minor criteria &#40;recurrent fever&#44; abnormal bone remodeling&#44; neutrophilic dermal infiltrate&#44; and elevated protein C reactive &#40;CRP&#41; and&#47;or leukocytosis&#41; are met&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Hematological malignancy is the major complication&#44; lymphoproliferative disorder can develop in about 20&#37; of patients&#44; so&#44; this syndrome needs to be considered in the differential diagnosis of urticarial dermatoses&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">A wide range of drugs has been used in patients with Schnitzler&#39;s syndrome without achieving a complete remission of symptoms&#46; All experts agree that Anakinra&#44; an interleukin-1-neutralizing agent&#44; is the only completely efficient treatment so it has been proposed to be added as a diagnostic criteria&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The patient started treatment with Anakinra at a dose of 100<span class="elsevierStyleHsp" style=""></span>mg&#47;24<span class="elsevierStyleHsp" style=""></span>h&#44; presenting a progressive decrease in the CRP and leukocyte count and an improvement of the skin lesions in 24<span class="elsevierStyleHsp" style=""></span>h&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Delay to diagnosis is still many years&#44; in this case&#44; it took thirteen years being assessed by different specialists&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The aim of this report is to increase the knowledge of this rare and debilitating syndrome&#46; An early treatment can improve the patient&#39;s quality of life and prognosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0065" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest&#46;</p></span></span>"
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Original language: English
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