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boy aged 11 years with no relevant history that presented with fever of 12 days&#8217; duration&#44; mild lymph node enlargement in the submandibular&#44; axillary and inguinal regions&#44; and hepatosplenomegaly detectable on palpation through the iliac fossa&#46; The salient findings of blood tests were leukopenia &#40;1570&#47;mm<span class="elsevierStyleSup">3</span>&#41;&#44; neutropenia &#40;610&#47;mm<span class="elsevierStyleSup">3</span>&#41; and thrombocytopenia &#40;77<span class="elsevierStyleHsp" style=""></span>000&#47;mm<span class="elsevierStyleSup">3</span>&#41;&#44; confirmed by examination of a peripheral blood smear&#44; a creatinine level of 0&#46;93<span class="elsevierStyleHsp" style=""></span>mg&#47;dL and hypertransaminasaemia &#40;aspartame aminotransferase&#44; 73<span class="elsevierStyleHsp" style=""></span>U&#47;L&#59; alanine aminotransferase&#44; 85<span class="elsevierStyleHsp" style=""></span>U&#47;L&#41;&#46; There was no elevation of C-reactive protein or the erythrocyte sedimentation rate&#46; The findings of the chest radiograph were unremarkable&#44; and the abdominal ultrasound scan confirmed the presence of hepatosplenomegaly&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Serological tests&#44; cultures and viral polymerase chain reaction tests for viral detection &#40;including cytomegalovirus and Epstein&#8211;Barr virus &#91;EBV&#93;&#41;&#44; as were the Mantoux test and QuantiFERON assay&#46; Immunoglobulin levels were normal&#46; The patient had an elevated level of angiotensin converting enzyme &#40;ACE&#44; 248&#46;9<span class="elsevierStyleHsp" style=""></span>U&#47;L&#59; normal range&#44; 8&#8211;55<span class="elsevierStyleHsp" style=""></span>U&#47;L&#41; and hypercalcaemia &#40;10&#46;86<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient underwent a magnetic resonance scan of the thorax and abdomen and a positron emission tomography &#40;PET&#41; scan &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The bone marrow biopsy revealed an absence of cancerous cells&#44; and the lymph node biopsy the presence of Schaumann bodies&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">These findings&#44; having ruled out an infectious or cancerous aetiology&#44; led to diagnosis of sarcoidosis&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">During the diagnostic process&#44; the fever and blood test abnormalities resolved&#44; and the hepatosplenomegaly improved&#44; so the episode was considered to be self-limited&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">At 18 months&#44; the patient experienced a recurrence of leukopenia&#44; hypertransaminasaemia and elevation of ACE&#46; The ultrasound scan revealed nephrocalcinosis&#46; This led to initiation of treatment with oral corticosteroids at a dose of 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#44; with a positive response&#44; although the patient also required methotrexate delivered subcutaneously at a dose of 15<span class="elsevierStyleHsp" style=""></span>mg&#47;m<span class="elsevierStyleSup">2</span>&#47;week that was eventually switched to adalimumab due to the presence of hypertransaminasaemia&#46; During the follow-up&#44; the patient developed a new episode triggered by infection that resolved with corticosteroids&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Case 2&#58; boy aged 4 years with persisting hepatosplenomegaly 8 months after diagnosis of primary infection by EBV with serological confirmation&#46; The abdominal ultrasound scan revealed massive splenomegaly and mesenteric lymphadenopathy&#44; and the EBV load in blood remained detectable at 100<span class="elsevierStyleHsp" style=""></span>000<span class="elsevierStyleHsp" style=""></span>copies&#47;mL&#44; while all other infection screening tests were negative&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">A month later&#44; the patient developed fever and the number and size of involved lymph nodes in the abdominal region increased&#46; The blood EBV load exhibited a decreasing trend&#46; The patient underwent screening for X-linked autoimmune lymphoproliferative syndrome &#40;ALPS&#41; and bone marrow and lymph node biopsies&#44; with detection of necrotising epithelioid granulomas&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Three months later&#44; when the EBV load in blood was undetectable&#44; the patient experienced a recurrence of fever accompanied by anemia and hypertransaminasaemia&#46; The computed tomography scan of the thorax and abdomen revealed mediastinal and abdominal lymphadenopathy and 24 nodules in the lungs &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>a&#41;&#44; which led to performance of additional biopsies &#40;lymph node and spleen&#41; that revealed sarcoid-like nonspecific granulomatous lymphadenitis&#46; The Mantoux test and QuantiFERON assay were both negative&#46; Examination of a bronchoalveolar lavage sample revealed a CD4&#47;CD8 ratio of 3&#46;92 &#40;normal range&#44; 0&#46;9&#8211;1&#46;9&#41;&#46; The serum level of ACE was 239<span class="elsevierStyleHsp" style=""></span>U&#47;L&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0065" class="elsevierStylePara elsevierViewall">Following diagnosis of sarcoidosis&#44; the patient started treatment with systemic corticosteroids &#40;1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41; and later on with methotrexate&#44; which achieved a reduction in the number of pulmonary nodules and in the size of the spleen&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Although in the initial evaluation the level of immunoglobulins had been within the normal range&#44; during the follow-up the patient developed hypogammaglobulinemia&#46; After intensification of immunosuppression with adalimumab and administration of several doses of intravenous immunoglobulin&#44; the levels recovered and remained normal over time without immunoglobulin replacement therapy&#44; which ruled out common variable immune deficiency&#46; The patient became asymptomatic and the number of nodules had decreased significantly at 10 years of follow-up &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>b&#41;&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">In conclusion&#44; sarcoidosis is a disease that is rare and exhibits a heterogeneous clinical presentation in the pediatric age group&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Splenomegaly&#44; described in fewer than 10&#37; of affected adults&#44; may be present in up to 50&#37; of pediatric cases&#44; and be severe in some&#44; as was the case in our sample&#46; This entails an extensive differential diagnosis<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> including neoplasias and ALPS&#44; especially in the presence of cytopenia&#44; and infection &#40;especially tuberculosis&#41; and immune deficiencies in case of pulmonary involvement&#46; Common variable immune deficiency has a similar presentation&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> as it may have onset with splenomegaly and one of its most frequent complications is granulomatous-lymphocytic interstitial lung disease&#44; so it is essential that immunoglobulin levels are measured before initiating immunosuppressive therapy&#46; On the other hand&#44; X-linked lymphoproliferative disease should be considered in male patients if the trigger is EBV infection&#44; as was the case of the second patient presented here&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Nephrocalcinosis is a complication described in up to 10&#37; of cases&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> highlighting the need of monitoring to guide treatment planning and prevent sequelae&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">As for treatment&#44; there is no standardized approach to the management of systemic sarcoidosis in the pediatric population&#46; Steroid therapy is the first-line treatment&#44; as is the case in adult patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;6</span></a> In refractory or steroid-dependent cases&#44; there is the option of adding an immunosuppressive agent such as methotrexate or anti-TNF-&#945;&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Cobreros-P&#233;rez &#193;&#44; Galindo-Zavala R&#44; Carazo-Gallego B&#44; Mart&#237;n-Pedraz L&#44; N&#250;&#241;ez-Cuadros E&#46; Sarcoidosis sist&#233;mica&#58; cuando la esplenomegalia no es lo que parece&#46; An Pediatr &#40;Barc&#41;&#46; 2021&#59;94&#58;48&#8211;50&#46;</p>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Previous presentations&#58; this study was presented at the 46th Annual Meeting of the Sociedad de Pediatr&#237;a de Andaluc&#237;a Oriental&#44; March 2019&#44; Jaen&#44; Spain&#59; and the 67th Congress of the Asociaci&#243;n Espa&#241;ola de Pediatr&#237;a&#44; June 2019&#44; Burgos&#44; Spain&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;a&#41; Late-onset hepatosplenomegaly&#46; No signs of pulmonary involvement&#46; &#40;b&#41; Hypermetabolic lymphadenopathy &#40;maximum standardized uptake value &#91;SUVmax&#93;&#44; 3&#46;89&#41;&#46; Hypermetabolic splenomegaly &#40;SUVmax&#44; 3&#46;15&#41;&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">&#40;a&#41; Before treatment with anti-TNF-&#945;&#58; multiple well-delimited parenchymal nodules with a subpleural&#44; peripheral location and a maximum diameter of 11<span class="elsevierStyleHsp" style=""></span>mm&#46; No evidence of pulmonary fibrosis&#46; Findings compatible with stage 2 pulmonary sarcoidosis&#46; &#40;b&#41; After treatment with anti-TNF-&#945;&#58; significant decrease in the number and size of pulmonary nodules&#46;</p>"
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