{"id":"https://openalex.org/W2989757258","doi":"https://doi.org/10.1097/mph.0000000000001662","title":"A Case of Langerhans Cell Histiocytosis With Multifocal, Single-System GI Tract Involvement and Literature Review","display_name":"A Case of Langerhans Cell Histiocytosis With Multifocal, Single-System GI Tract Involvement and Literature Review","publication_year":2019,"publication_date":"2019-11-22","ids":{"openalex":"https://openalex.org/W2989757258","doi":"https://doi.org/10.1097/mph.0000000000001662","mag":"2989757258","pmid":"https://pubmed.ncbi.nlm.nih.gov/31764515"},"language":"en","primary_location":{"id":"doi:10.1097/mph.0000000000001662","doi":"https://doi.org/10.1097/mph.0000000000001662","is_oa":false,"landing_page_url":"https://doi.org/10.1097/mph.0000000000001662","pdf_url":null,"source":{"id":"https://openalex.org/S15952725","display_name":"Journal of Pediatric Hematology/Oncology","issn_l":"1077-4114","issn":["1077-4114","1536-3678"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core","dongbi-d","doyens","jufo-1","ki-jl-1","medline","norway-1","poland-40","russia-white-list-2"],"host_organization":"https://openalex.org/P4310315671","host_organization_name":"Lippincott Williams & Wilkins","host_organization_lineage":["https://openalex.org/P4310315671","https://openalex.org/P4310318547"],"host_organization_lineage_names":["Lippincott Williams & Wilkins","Wolters Kluwer"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Pediatric Hematology/Oncology","raw_type":"journal-article"},"volume":null,"type":"article","indexed_in":["crossref","pubmed"],"open_access":{"is_oa":false,"oa_status":"closed","oa_url":null,"any_repository_has_fulltext":false},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5030455946","display_name":"Moran Gotesman","orcid":"https://orcid.org/0000-0002-4695-8439","observed_orcids":["https://orcid.org/0000-0002-4695-8439"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Moran Gotesman","raw_affiliation_strings":["Pediatrics, Pediatric Hematology and Oncology"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics, Pediatric Hematology and Oncology","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5089714018","display_name":"Ruth Getachew","orcid":"https://orcid.org/0009-0003-6986-1386","observed_orcids":["https://orcid.org/0009-0003-6986-1386"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Ruth Getachew","raw_affiliation_strings":["Family Medicine"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Family Medicine","institution_ids":[]}]},{"author_position":"middle","author":{"id":null,"display_name":"Sonia Morales","orcid":null,"observed_orcids":[]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Sonia Morales","raw_affiliation_strings":["Pediatrics"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5075273637","display_name":"Kenneth M. Zangwill","orcid":"https://orcid.org/0000-0002-5026-4368","observed_orcids":["https://orcid.org/0000-0002-5026-4368"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Kenneth M. Zangwill","raw_affiliation_strings":["Pediatrics"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5084262665","display_name":"George B. Gershman","orcid":"https://orcid.org/0009-0001-2610-9229","observed_orcids":["https://orcid.org/0009-0001-2610-9229"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"George Gershman","raw_affiliation_strings":["Pediatrics, Pediatric Gastroenterology"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics, Pediatric Gastroenterology","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5100666688","display_name":"Steven Lee","orcid":"https://orcid.org/0000-0002-6853-1460","observed_orcids":["https://orcid.org/0000-0002-6853-1460"]},"institutions":[{"id":"https://openalex.org/I161318765","display_name":"University of California, Los Angeles","ror":"https://ror.org/046rm7j60","country_code":"US","type":"education","lineage":["https://openalex.org/I161318765"]},{"id":"https://openalex.org/I4210165985","display_name":"Harbor\u2013UCLA Medical Center","ror":"https://ror.org/05h4zj272","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I1328359107","https://openalex.org/I4210165985"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Steven Lee","raw_affiliation_strings":["Surgery, Pediatric Surgery, Harbor-UCLA Medical Center, Torrance, CA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Surgery, Pediatric Surgery, Harbor-UCLA Medical Center, Torrance, CA","institution_ids":["https://openalex.org/I161318765","https://openalex.org/I4210165985"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5048721663","display_name":"Joseph L. Lasky","orcid":"https://orcid.org/0000-0002-2565-1921","observed_orcids":["https://orcid.org/0000-0002-2565-1921"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Joseph L. Lasky","raw_affiliation_strings":["Pediatrics, Pediatric Hematology and Oncology"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics, Pediatric Hematology and Oncology","institution_ids":[]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5044220421","display_name":"Eduard H. Panosyan","orcid":"https://orcid.org/0000-0003-1701-1251","observed_orcids":["https://orcid.org/0000-0003-1701-1251"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Eduard H. Panosyan","raw_affiliation_strings":["Pediatrics, Pediatric Hematology and Oncology"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Pediatrics, Pediatric Hematology and Oncology","institution_ids":[]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":2,"corresponding_author_ids":["https://openalex.org/A5030455946","https://openalex.org/A5044220421","https://openalex.org/A5048721663"],"corresponding_institution_ids":[],"apc_list":null,"apc_paid":null,"fwci":0.5697,"has_fulltext":false,"cited_by_count":9,"citation_normalized_percentile":{"value":0.68861202,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":{"min":88,"max":96},"biblio":{"volume":"42","issue":"6","first_page":"e491","last_page":"e493"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T11909","display_name":"Histiocytic Disorders and Treatments","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2737","display_name":"Physiology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T11909","display_name":"Histiocytic Disorders and Treatments","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2737","display_name":"Physiology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10980","display_name":"Vascular Malformations and Hemangiomas","score":0.0,"subfield":{"id":"https://openalex.org/subfields/2746","display_name":"Surgery"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T11616","display_name":"Autoimmune and Inflammatory Disorders Research","score":0.0,"subfield":{"id":"https://openalex.org/subfields/2720","display_name":"Hematology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/langerhans-cell-histiocytosis","display_name":"Langerhans cell histiocytosis","score":1.0},{"id":"https://openalex.org/keywords/gastrointestinal-involvement","display_name":"gastrointestinal involvement","score":0.8610000014305115},{"id":"https://openalex.org/keywords/multifocality","display_name":"multifocality","score":0.8320000171661377},{"id":"https://openalex.org/keywords/appendectomy","display_name":"appendectomy","score":0.6660000085830688},{"id":"https://openalex.org/keywords/cd1a","display_name":"CD1a","score":0.699999988079071},{"id":"https://openalex.org/keywords/s100-protein","display_name":"S-100 protein","score":0.9330000281333923},{"id":"https://openalex.org/keywords/histiocytic-disorders","display_name":"histiocytic disorders","score":0.6790000200271606},{"id":"https://openalex.org/keywords/young-adults","display_name":"young adults","score":0.5860000252723694}],"concepts":[{"id":"https://openalex.org/C2777484599","wikidata":"https://www.wikidata.org/wiki/Q374036","display_name":"Langerhans cell histiocytosis","level":3,"score":0.9487557411193848},{"id":"https://openalex.org/C119765067","wikidata":"https://www.wikidata.org/wiki/Q6485957","display_name":"Langerin","level":4,"score":0.9162111878395081},{"id":"https://openalex.org/C43100952","wikidata":"https://www.wikidata.org/wiki/Q654612","display_name":"Histiocyte","level":2,"score":0.7198281288146973},{"id":"https://openalex.org/C2780174970","wikidata":"https://www.wikidata.org/wiki/Q1411740","display_name":"Histiocytosis","level":3,"score":0.6674754023551941},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.5463999509811401},{"id":"https://openalex.org/C142724271","wikidata":"https://www.wikidata.org/wiki/Q7208","display_name":"Pathology","level":1,"score":0.525807797908783},{"id":"https://openalex.org/C2778379841","wikidata":"https://www.wikidata.org/wiki/Q1623344","display_name":"Langerhans cell","level":3,"score":0.5013668537139893},{"id":"https://openalex.org/C2780512811","wikidata":"https://www.wikidata.org/wiki/Q11829360","display_name":"Gastrointestinal tract","level":2,"score":0.49698594212532043},{"id":"https://openalex.org/C2779701055","wikidata":"https://www.wikidata.org/wiki/Q929833","display_name":"Rare disease","level":3,"score":0.47829610109329224},{"id":"https://openalex.org/C2779134260","wikidata":"https://www.wikidata.org/wiki/Q12136","display_name":"Disease","level":2,"score":0.4553409814834595},{"id":"https://openalex.org/C203014093","wikidata":"https://www.wikidata.org/wiki/Q101929","display_name":"Immunology","level":1,"score":0.27538180351257324},{"id":"https://openalex.org/C2778170410","wikidata":"https://www.wikidata.org/wiki/Q506253","display_name":"Dendritic cell","level":3,"score":0.19598057866096497},{"id":"https://openalex.org/C8891405","wikidata":"https://www.wikidata.org/wiki/Q1059","display_name":"Immune system","level":2,"score":0.1627596914768219},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.14778226613998413}],"mesh":[{"descriptor_ui":"D000328","descriptor_name":"Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D000328","descriptor_name":"Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D000328","descriptor_name":"Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D000964","descriptor_name":"Antimetabolites, Antineoplastic","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D000964","descriptor_name":"Antimetabolites, Antineoplastic","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D000964","descriptor_name":"Antimetabolites, Antineoplastic","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D003561","descriptor_name":"Cytarabine","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D003561","descriptor_name":"Cytarabine","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D003561","descriptor_name":"Cytarabine","qualifier_ui":"Q000627","qualifier_name":"therapeutic use","is_major_topic":false},{"descriptor_ui":"D005260","descriptor_name":"Female","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D005260","descriptor_name":"Female","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D005260","descriptor_name":"Female","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D005767","descriptor_name":"Gastrointestinal Diseases","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000150","qualifier_name":"complications","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D006646","descriptor_name":"Histiocytosis, Langerhans-Cell","qualifier_ui":"Q000473","qualifier_name":"pathology","is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D011379","descriptor_name":"Prognosis","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D011379","descriptor_name":"Prognosis","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D011379","descriptor_name":"Prognosis","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D055815","descriptor_name":"Young Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D055815","descriptor_name":"Young Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D055815","descriptor_name":"Young Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false}],"locations_count":2,"locations":[{"id":"doi:10.1097/mph.0000000000001662","doi":"https://doi.org/10.1097/mph.0000000000001662","is_oa":false,"landing_page_url":"https://doi.org/10.1097/mph.0000000000001662","pdf_url":null,"source":{"id":"https://openalex.org/S15952725","display_name":"Journal of Pediatric Hematology/Oncology","issn_l":"1077-4114","issn":["1077-4114","1536-3678"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core","dongbi-d","doyens","jufo-1","ki-jl-1","medline","norway-1","poland-40","russia-white-list-2"],"host_organization":"https://openalex.org/P4310315671","host_organization_name":"Lippincott Williams & Wilkins","host_organization_lineage":["https://openalex.org/P4310315671","https://openalex.org/P4310318547"],"host_organization_lineage_names":["Lippincott Williams & Wilkins","Wolters Kluwer"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Pediatric Hematology/Oncology","raw_type":"journal-article"},{"id":"pmid:31764515","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/31764515","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of pediatric hematology/oncology","raw_type":"Case Reports"}],"best_oa_location":null,"sustainable_development_goals":[{"id":"https://metadata.un.org/sdg/3","display_name":"Good health and well-being","score":0.9922999739646912}],"sustainable_development_goals_aurora":[{"id":"https://metadata.un.org/sdg/3","display_name":"Good health and well-being","score":0.5199999809265137}],"study_designs":[{"id":"https://openalex.org/study-designs/case-report","display_name":"Case Report"}],"awards":[],"funders":[],"has_content":{"pdf":false,"grobid_xml":false},"content_urls":null,"referenced_works_count":11,"referenced_works":["https://openalex.org/W1989156909","https://openalex.org/W2020435262","https://openalex.org/W2021520947","https://openalex.org/W2041650858","https://openalex.org/W2062892036","https://openalex.org/W2069035104","https://openalex.org/W2126196693","https://openalex.org/W2134279621","https://openalex.org/W2318756797","https://openalex.org/W2775456530","https://openalex.org/W2793774897"],"related_works":["https://openalex.org/W1577975980","https://openalex.org/W1965717142","https://openalex.org/W2153773747","https://openalex.org/W2160493984","https://openalex.org/W3202606992","https://openalex.org/W2333959279","https://openalex.org/W4245270170","https://openalex.org/W2901178386","https://openalex.org/W2739739521","https://openalex.org/W2895196065"],"abstract_inverted_index":{"Langerhans":[0,20],"cell":[1],"histiocytosis":[2],"(LCH)":[3],"is":[4,42],"the":[5,12],"most":[6],"common":[7],"histiocytic":[8],"disorder,":[9],"characterized":[10],"by":[11],"pathologic":[13],"clonal":[14],"proliferation":[15],"and":[16,44,53,75,84,95],"accumulation":[17],"of":[18,34,98],"immature":[19],"cells":[21],"within":[22],"organs.":[23],"Multiple":[24],"organ":[25],"systems":[26],"can":[27],"be":[28],"affected,":[29],"resulting":[30],"in":[31,40,47],"a":[32,50,60,72,92],"spectrum":[33],"clinical":[35,94],"manifestations.":[36],"Isolated":[37],"gastrointestinal":[38,67],"involvement":[39],"LCH":[41],"rare":[43],"usually":[45,54],"presents":[46],"childhood":[48],"as":[49],"multisystem":[51],"disease":[52,99],"has":[55],"poor":[56],"outcomes.":[57],"We":[58],"describe":[59],"20-year-old":[61],"Hispanic":[62],"female":[63],"with":[64,87,100],"multifocal,":[65],"single-system":[66],"LCH.":[68],"Initially":[69],"diagnosed":[70],"from":[71],"CD1a,":[73],"S100,":[74],"CD207":[76],"(Langerin)":[77],"positive":[78],"appendix":[79],"tissue":[80],"after":[81],"an":[82,88],"appendectomy":[83],"confirmed":[85],"multifocal":[86],"endoscopy.":[89],"She":[90],"had":[91],"full":[93],"endoscopic":[96],"resolution":[97],"cytarabine":[101],"therapy.":[102]},"counts_by_year":[{"year":2026,"cited_by_count":1},{"year":2025,"cited_by_count":2},{"year":2024,"cited_by_count":1},{"year":2023,"cited_by_count":1},{"year":2022,"cited_by_count":1},{"year":2021,"cited_by_count":3}],"updated_date":"2026-10-16T09:59:22.235173","created_date":"2025-10-10T00:00:00"}