{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T16:14:10Z","timestamp":1753892050167,"version":"3.41.2"},"reference-count":36,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,1,16]],"date-time":"2024-01-16T00:00:00Z","timestamp":1705363200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Pediatr."],"abstract":"<jats:p>Lymphocytic interstitial pneumonia (LIP) in pediatric patients without human immunodeficiency virus (HIV) infection remains a poorly characterized and enigmatic disease. Immunological dysregulation, mutations in the COPA gene, and increased morbidity and mortality have been reported in these patients. We present a case of LIP in a pediatric patient without HIV infection. This patient was infected with human T-lymphotropic virus type 1 (HTLV-1) and required right lower lobectomy with pathological findings compatible with lymphocytic interstitial pneumonia. In addition, bronchiectasis, dermatological involvement, and malnutrition were documented. However, no autoimmune disease, polymyositis, myelopathy, or opportunistic infections were found. There were no abnormalities in cellular and humoral immunity. A genetic study identified heterozygous mutations in the SCNN1B, FCHO1, and IL7R genes using single exome sequencing of coding and splicing regions. Although these heterozygous variants are not reported to be aetiological for LIP or diagnostic for the patient's congenital immunodeficiency, we believe they are associated with the severe lung damage seen in the patient's case.<\/jats:p>","DOI":"10.3389\/fped.2023.1307607","type":"journal-article","created":{"date-parts":[[2024,1,16]],"date-time":"2024-01-16T04:29:39Z","timestamp":1705379379000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Lymphocytic interstitial non-HIV-related pneumonia in pediatrics: a case report"],"prefix":"10.3389","volume":"11","author":[{"given":"Andrea Dionelly","family":"Murillo Casas","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Diana Mar\u00eda","family":"Duarte Dorado","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Manuela","family":"Olaya Hern\u00e1ndez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2024,1,16]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"463","DOI":"10.1016\/j.ccm.2016.04.009","article-title":"Lymphocytic interstitial pneumonia","volume":"37","author":"Panchabhai","year":"2016","journal-title":"Clin Chest Med"},{"key":"B2","doi-asserted-by":"publisher","first-page":"406","DOI":"10.1055\/s-0036-1580691","article-title":"Lymphoid interstitial pneumonia and other benign lymphoid disorders","volume":"37","author":"Arcadu","year":"2016","journal-title":"Semin Respir Crit Care Med"},{"key":"B3","doi-asserted-by":"publisher","first-page":"2150","DOI":"10.1378\/chest.122.6.2150","article-title":"Lymphoid interstitial pneumonia: a narrative review","volume":"122","author":"Swigris","year":"2002","journal-title":"Chest"},{"key":"B4","doi-asserted-by":"publisher","first-page":"450","DOI":"10.1055\/s-0032-1325156","article-title":"Lymphocytic interstitial pneumonia and other benign lymphoid disorders","volume":"33","author":"Tian","year":"2012","journal-title":"Semin Respir Crit Care Med"},{"key":"B5","doi-asserted-by":"publisher","first-page":"364","DOI":"10.1183\/09031936.06.00076705","article-title":"Lymphoid interstitial pneumonia: clinical features, associations and prognosis","volume":"28","author":"Cha","year":"2006","journal-title":"Eur Respir J"},{"key":"B6","doi-asserted-by":"publisher","first-page":"745","DOI":"10.1097\/00004728-199409000-00012","article-title":"Lung cyst formation in lymphocytic interstitial pneumonia: CT features","volume":"18","author":"Ichikawa","year":"1994","journal-title":"J Comput Assist Tomogr"},{"key":"B7","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1016\/j.ejro.2021.100336","article-title":"Interstitial lung abnormality (ILA) and nonspecific interstitial pneumonia (NSIP)","volume":"8","author":"Hino","year":"2021","journal-title":"Eur J Radiol Open"},{"key":"B8","doi-asserted-by":"publisher","first-page":"691","DOI":"10.5858\/134.5.691","article-title":"Update on nonneoplastic pulmonary lymphoproliferative disorders and related entities","volume":"134","author":"Guinee","year":"2010","journal-title":"Arch Pathol Lab Med"},{"key":"B9","doi-asserted-by":"publisher","first-page":"123","DOI":"10.4137\/CCRPM.S23320","article-title":"Pathology of idiopathic interstitial pneumonias","volume":"9","author":"Hashisako","year":"2016","journal-title":"Clin Med Insights Circ Respir Pulm Med"},{"key":"B10","doi-asserted-by":"publisher","first-page":"231","DOI":"10.1136\/thoraxjnl-2017-210519","article-title":"International management platform for children\u2019s interstitial lung disease (chILD-EU)","volume":"73","author":"Griese","year":"2018","journal-title":"Thorax"},{"key":"B11","doi-asserted-by":"publisher","first-page":"899","DOI":"10.25259\/jpats_33_2020","article-title":"Pediatric interstitial lung disease","volume":"43","author":"Vijayasekaran","year":"2020","journal-title":"Indian Pediatr"},{"key":"B12","doi-asserted-by":"publisher","first-page":"7","DOI":"10.3390\/jcm11061747","article-title":"Etiologic classification of diffuse parenchymal (interstitial) lung diseases","volume":"11","author":"Griese","year":"2022","journal-title":"J Clin Med"},{"key":"B13","doi-asserted-by":"publisher","first-page":"1078","DOI":"10.1136\/thoraxjnl-2015-207349","article-title":"European Protocols for the diagnosis and initial treatment of interstitial lung disease in children","volume":"70","author":"Bush","year":"2015","journal-title":"Thorax"},{"key":"B14","doi-asserted-by":"publisher","first-page":"1451","DOI":"10.1513\/AnnalsATS.201508-558PS","article-title":"Children\u2019s interstitial and diffuse lung disease: progress and future horizons","volume":"12","author":"Deterding","year":"2015","journal-title":"Ann Am Thorac Soc"},{"key":"B15","doi-asserted-by":"publisher","first-page":"568","DOI":"10.1016\/S2352-4642(19)30117-8","article-title":"Children\u2019s interstitial and diffuse lung disease","volume":"3","author":"Cunningham","year":"2019","journal-title":"Lancet Child Adolesc Health"},{"key":"B16","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1183\/16000617.0188-2022","article-title":"Diagnostic workup of childhood interstitial lung disease","volume":"32","author":"Nathan","year":"2023","journal-title":"Eur Respir Rev"},{"key":"B17","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1186\/s13019-022-01826-6","article-title":"Transformation of lymphoid interstitial pneumonia (LIP) into malignant lymphoma in patients with sjogren\u2019s syndrome: a case report and literature review","volume":"17","author":"Zhu","year":"2022","journal-title":"J Cardiothorac Surg"},{"key":"B18","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1007\/s10875-016-0271-8","article-title":"Copa syndrome: a novel autosomal dominant immune dysregulatory disease","volume":"36","author":"Vece","year":"2016","journal-title":"J Clin Immunol"},{"key":"B19","doi-asserted-by":"publisher","first-page":"2232","DOI":"10.1002\/ppul.25373","article-title":"Pulmonary manifestations of immune dysregulationin CTLA-4 haploinsufficiency and LRBA deficiency","volume":"56","author":"Krone","year":"2021","journal-title":"Pediatr Pulmonol"},{"key":"B20","doi-asserted-by":"publisher","first-page":"591","DOI":"10.1182\/blood-2014-09-602763","article-title":"Early-onset lymphoproliferation and autoimmunitycaused by germline STAT3 gain-of-function mutations","volume":"125","author":"Milner","year":"2015","journal-title":"Blood"},{"key":"B21","doi-asserted-by":"publisher","first-page":"3303","DOI":"10.1002\/ppul.26660","article-title":"Unclassifiable interstitial lung disease and autoimmunity: towards IPAF in children?","volume":"58","author":"Aoust","year":"2023","journal-title":"Pediatr Pulmonol"},{"key":"B22","doi-asserted-by":"publisher","first-page":"436","DOI":"10.1126\/science.aaa1663","article-title":"Autoimmune disease. Patients with LRBA deficiency showCTLA4 loss and immune dysregulation responsive to Abatacept therapy","volume":"349","author":"Lo","year":"2015","journal-title":"Science"},{"key":"B23","doi-asserted-by":"publisher","first-page":"909","DOI":"10.1002\/ppul.24680","article-title":"Lymphocytic interstitial pneumonia and follicular bronchiolitis in children: a registry-based case series","volume":"55","author":"Prenzel","year":"2020","journal-title":"Pediatr Pulmonol"},{"year":"","key":"B24"},{"year":"","key":"B25"},{"year":"","key":"B26"},{"key":"B27","doi-asserted-by":"publisher","first-page":"564","DOI":"10.1002\/humu.22981","article-title":"HGVS recommendations for the description of sequence variants: 2016 update","volume":"37","author":"den Dunnen","year":"2016","journal-title":"Hum Mutat"},{"year":"","key":"B28"},{"year":"","key":"B29"},{"year":"","key":"B30"},{"key":"B31","doi-asserted-by":"crossref","DOI":"10.1007\/978-3-030-63365-3","article-title":"Diffuse cystic lung diseases","volume-title":"Respiratory medicine series","author":"Gupta","year":"2021"},{"key":"B32","doi-asserted-by":"publisher","first-page":"1361","DOI":"10.1164\/ajrccm\/144.6.1361","article-title":"Detection of human T-cell lymphotropic virustype I-related antibodies in patients with lymphocytic interstitial pneumonia","volume":"144","author":"Setoguchi","year":"1991","journal-title":"Am Rev Respir Dis"},{"key":"B33","doi-asserted-by":"publisher","first-page":"474","DOI":"10.1007\/s00277-002-0465-4","article-title":"KL-6 as an indicator for lymphocytic interstitial pneumonia (LIP) in a human T-lymphotrophic virus type 1 (HTLV-1) carrier","volume":"81","author":"Ishikawa","year":"2002","journal-title":"Ann Hematol"},{"key":"B34","doi-asserted-by":"publisher","first-page":"664","DOI":"10.1053\/crad.2001.0677","article-title":"HTLV-1-associated bronchopneumonopathy (HAB): cT-pathological correlation","volume":"56","author":"Tateishi","year":"2001","journal-title":"Clin Radiol"},{"key":"B35","doi-asserted-by":"publisher","first-page":"1","DOI":"10.3389\/fitd.2021.824067","article-title":"Case series: pediatric human T-lymphotropic virus type 1 and its clinical expression","volume":"2","author":"James","year":"2022","journal-title":"Front Trop Dis"},{"key":"B36","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1183\/16000617.0100-2017","article-title":"Chronic interstitial lung disease in children","volume":"27","author":"Griese","year":"2018","journal-title":"Eur Respir Rev"}],"container-title":["Frontiers in Pediatrics"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2023.1307607\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,1,16]],"date-time":"2024-01-16T04:29:42Z","timestamp":1705379382000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2023.1307607\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,16]]},"references-count":36,"alternative-id":["10.3389\/fped.2023.1307607"],"URL":"https:\/\/doi.org\/10.3389\/fped.2023.1307607","relation":{},"ISSN":["2296-2360"],"issn-type":[{"type":"electronic","value":"2296-2360"}],"subject":[],"published":{"date-parts":[[2024,1,16]]},"article-number":"1307607"}}