{"id":"https://openalex.org/W7156221908","doi":"https://doi.org/10.3389/fmed.2026.1818323","title":"Recurrent self-limiting abdominal pain with bowel wall edema misdiagnosed as gastroenteritis: a case report of C1-inhibitor-deficient hereditary angioedema","display_name":"Recurrent self-limiting abdominal pain with bowel wall edema misdiagnosed as gastroenteritis: a case report of C1-inhibitor-deficient hereditary angioedema","publication_year":2026,"publication_date":"2026-04-27","ids":{"openalex":"https://openalex.org/W7156221908","doi":"https://doi.org/10.3389/fmed.2026.1818323","pmid":"https://pubmed.ncbi.nlm.nih.gov/42125081"},"language":"en","primary_location":{"id":"doi:10.3389/fmed.2026.1818323","doi":"https://doi.org/10.3389/fmed.2026.1818323","is_oa":true,"landing_page_url":"https://doi.org/10.3389/fmed.2026.1818323","pdf_url":null,"source":{"id":"https://openalex.org/S2597052008","display_name":"Frontiers in Medicine","issn_l":"2296-858X","issn":["2296-858X"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1"],"host_organization":"https://openalex.org/P4310320527","host_organization_name":"Frontiers Media","host_organization_lineage":["https://openalex.org/P4310320527"],"host_organization_lineage_names":["Frontiers Media"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Frontiers in Medicine","raw_type":"journal-article"},"volume":null,"type":"article","indexed_in":["crossref","doaj","pubmed"],"open_access":{"is_oa":true,"oa_status":"gold","oa_url":"https://doi.org/10.3389/fmed.2026.1818323","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5134695148","display_name":"Binlong Zhang","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210164143","display_name":"Yueqing People's Hospital","ror":"https://ror.org/054t0kh70","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I4210164143"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Binlong Zhang","raw_affiliation_strings":["Department of Gastroenterology, Yueqing People\u2019s Hospital"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Gastroenterology, Yueqing People\u2019s Hospital","institution_ids":["https://openalex.org/I4210164143"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5134662376","display_name":"Haocheng Zhao","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210164143","display_name":"Yueqing People's Hospital","ror":"https://ror.org/054t0kh70","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I4210164143"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Haocheng Zhao","raw_affiliation_strings":["Department of Hematology, Yueqing People\u2019s Hospital"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Hematology, Yueqing People\u2019s Hospital","institution_ids":["https://openalex.org/I4210164143"]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":1,"corresponding_author_ids":[],"corresponding_institution_ids":["https://openalex.org/I4210164143"],"apc_list":{"value":3150,"currency":"CHF","value_usd":3801},"apc_paid":{"value":3150,"currency":"CHF","value_usd":3801},"fwci":0.0,"has_fulltext":true,"cited_by_count":0,"citation_normalized_percentile":{"value":0.26862532,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":null,"biblio":{"volume":"13","issue":null,"first_page":"1818323","last_page":"1818323"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T11917","display_name":"Coagulation, Bradykinin, Polyphosphates, and Angioedema","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2716","display_name":"Genetics"},"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/T11917","display_name":"Coagulation, Bradykinin, Polyphosphates, and Angioedema","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2716","display_name":"Genetics"},"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/T10993","display_name":"Complement system in diseases","score":0.0,"subfield":{"id":"https://openalex.org/subfields/2403","display_name":"Immunology"},"field":{"id":"https://openalex.org/fields/24","display_name":"Immunology and Microbiology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},{"id":"https://openalex.org/T11346","display_name":"Mast cells and histamine","score":0.0,"subfield":{"id":"https://openalex.org/subfields/2403","display_name":"Immunology"},"field":{"id":"https://openalex.org/fields/24","display_name":"Immunology and Microbiology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/hereditary-angioedema","display_name":"hereditary angioedema","score":0.9470000267028809},{"id":"https://openalex.org/keywords/c1-inhibitor-deficiency","display_name":"C1 inhibitor deficiency","score":0.8930000066757202},{"id":"https://openalex.org/keywords/type-i-hereditary-angioedema","display_name":"type I hereditary angioedema","score":0.9020000100135803},{"id":"https://openalex.org/keywords/abdominal-angioedema","display_name":"abdominal angioedema","score":0.8619999885559082},{"id":"https://openalex.org/keywords/bowel-wall-edema","display_name":"bowel wall edema","score":0.8579999804496765},{"id":"https://openalex.org/keywords/ascites","display_name":"ascites","score":0.7369999885559082},{"id":"https://openalex.org/keywords/acute-abdomen","display_name":"acute abdomen","score":0.46799999475479126},{"id":"https://openalex.org/keywords/complement-c4","display_name":"complement C4","score":0.41600000858306885},{"id":"https://openalex.org/keywords/d-dimer","display_name":"D-dimer","score":0.781000018119812},{"id":"https://openalex.org/keywords/small-intestine","display_name":"small intestine","score":0.3199999928474426},{"id":"https://openalex.org/keywords/acute-abdominal-pain","display_name":"acute abdominal pain","score":0.4410000145435333}],"concepts":[{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.9660000205039978},{"id":"https://openalex.org/C2777788586","wikidata":"https://www.wikidata.org/wiki/Q2096745","display_name":"Hereditary angioedema","level":2,"score":0.7408000230789185},{"id":"https://openalex.org/C2780955771","wikidata":"https://www.wikidata.org/wiki/Q183425","display_name":"Abdominal pain","level":2,"score":0.6786999702453613},{"id":"https://openalex.org/C2777995640","wikidata":"https://www.wikidata.org/wiki/Q1129007","display_name":"Angioedema","level":2,"score":0.6276999711990356},{"id":"https://openalex.org/C2779983558","wikidata":"https://www.wikidata.org/wiki/Q9597","display_name":"Abdomen","level":2,"score":0.5472999811172485},{"id":"https://openalex.org/C2778923028","wikidata":"https://www.wikidata.org/wiki/Q974869","display_name":"Leukocytosis","level":2,"score":0.5102999806404114},{"id":"https://openalex.org/C2780886150","wikidata":"https://www.wikidata.org/wiki/Q152234","display_name":"Edema","level":2,"score":0.49559998512268066},{"id":"https://openalex.org/C141071460","wikidata":"https://www.wikidata.org/wiki/Q40821","display_name":"Surgery","level":1,"score":0.46549999713897705},{"id":"https://openalex.org/C2778856562","wikidata":"https://www.wikidata.org/wiki/Q424317","display_name":"Acute abdomen","level":2,"score":0.4607999920845032},{"id":"https://openalex.org/C2780496750","wikidata":"https://www.wikidata.org/wiki/Q193889","display_name":"Ascites","level":2,"score":0.44130000472068787},{"id":"https://openalex.org/C2779802037","wikidata":"https://www.wikidata.org/wiki/Q40878","display_name":"Diarrhea","level":2,"score":0.37770000100135803},{"id":"https://openalex.org/C2777802486","wikidata":"https://www.wikidata.org/wiki/Q491741","display_name":"Neutrophilia","level":2,"score":0.36820000410079956},{"id":"https://openalex.org/C42219234","wikidata":"https://www.wikidata.org/wiki/Q131130","display_name":"Anesthesia","level":1,"score":0.33709999918937683},{"id":"https://openalex.org/C2778754067","wikidata":"https://www.wikidata.org/wiki/Q354111","display_name":"Abdominal wall","level":2,"score":0.3305000066757202},{"id":"https://openalex.org/C2779134260","wikidata":"https://www.wikidata.org/wiki/Q12136","display_name":"Disease","level":2,"score":0.31209999322891235},{"id":"https://openalex.org/C2992927888","wikidata":"https://www.wikidata.org/wiki/Q424317","display_name":"Acute abdominal pain","level":3,"score":0.3109000027179718},{"id":"https://openalex.org/C2778211319","wikidata":"https://www.wikidata.org/wiki/Q902379","display_name":"Icatibant","level":4,"score":0.28790000081062317},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.28679999709129333},{"id":"https://openalex.org/C126838900","wikidata":"https://www.wikidata.org/wiki/Q77604","display_name":"Radiology","level":1,"score":0.28529998660087585},{"id":"https://openalex.org/C2780801072","wikidata":"https://www.wikidata.org/wiki/Q837512","display_name":"Differential diagnosis","level":2,"score":0.27630001306533813},{"id":"https://openalex.org/C90924648","wikidata":"https://www.wikidata.org/wiki/Q120569","display_name":"Gastroenterology","level":1,"score":0.26600000262260437},{"id":"https://openalex.org/C2776608144","wikidata":"https://www.wikidata.org/wiki/Q410352","display_name":"Gabapentin","level":3,"score":0.2526000142097473},{"id":"https://openalex.org/C2779124032","wikidata":"https://www.wikidata.org/wiki/Q2536390","display_name":"Abdominal distension","level":2,"score":0.2526000142097473}],"mesh":[],"locations_count":4,"locations":[{"id":"doi:10.3389/fmed.2026.1818323","doi":"https://doi.org/10.3389/fmed.2026.1818323","is_oa":true,"landing_page_url":"https://doi.org/10.3389/fmed.2026.1818323","pdf_url":null,"source":{"id":"https://openalex.org/S2597052008","display_name":"Frontiers in Medicine","issn_l":"2296-858X","issn":["2296-858X"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1"],"host_organization":"https://openalex.org/P4310320527","host_organization_name":"Frontiers Media","host_organization_lineage":["https://openalex.org/P4310320527"],"host_organization_lineage_names":["Frontiers Media"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Frontiers in Medicine","raw_type":"journal-article"},{"id":"pmid:42125081","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/42125081","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":"Frontiers in medicine","raw_type":"Case Reports"},{"id":"pmh:oai:pubmedcentral.nih.gov:13159194","doi":null,"is_oa":true,"landing_page_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC13159194/","pdf_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC13159194/pdf/fmed-13-1818323.pdf","source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"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":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":"Front Med (Lausanne)","raw_type":"Text"},{"id":"pmh:oai:doaj.org/article:c49e3e4ab07d4949aae7560f875dba00","doi":null,"is_oa":true,"landing_page_url":"https://doaj.org/article/c49e3e4ab07d4949aae7560f875dba00","pdf_url":null,"source":{"id":"https://openalex.org/S4306401280","display_name":"DOAJ (DOAJ: Directory of Open Access Journals)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-sa","license_id":"https://openalex.org/licenses/cc-by-sa","version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":"Frontiers in Medicine, Vol 13 (2026)","raw_type":"article"}],"best_oa_location":{"id":"doi:10.3389/fmed.2026.1818323","doi":"https://doi.org/10.3389/fmed.2026.1818323","is_oa":true,"landing_page_url":"https://doi.org/10.3389/fmed.2026.1818323","pdf_url":null,"source":{"id":"https://openalex.org/S2597052008","display_name":"Frontiers in Medicine","issn_l":"2296-858X","issn":["2296-858X"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1"],"host_organization":"https://openalex.org/P4310320527","host_organization_name":"Frontiers Media","host_organization_lineage":["https://openalex.org/P4310320527"],"host_organization_lineage_names":["Frontiers Media"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Frontiers in Medicine","raw_type":"journal-article"},"sustainable_development_goals":[{"score":0.98580002784729,"display_name":"Good health and well-being","id":"https://metadata.un.org/sdg/3"}],"sustainable_development_goals_aurora":[{"score":0.8563296794891357,"display_name":"Good health and well-being","id":"https://metadata.un.org/sdg/3"}],"study_designs":[{"display_name":"Case Report","id":"https://openalex.org/study-designs/case-report"}],"awards":[],"funders":[],"has_content":{"pdf":false,"grobid_xml":false},"content_urls":null,"referenced_works_count":15,"referenced_works":["https://openalex.org/W2343704133","https://openalex.org/W2588081052","https://openalex.org/W2599824295","https://openalex.org/W2902818271","https://openalex.org/W2905117661","https://openalex.org/W2977229276","https://openalex.org/W3032400603","https://openalex.org/W3082923583","https://openalex.org/W3127297674","https://openalex.org/W3127819425","https://openalex.org/W4246423891","https://openalex.org/W4319660730","https://openalex.org/W4322604179","https://openalex.org/W4399211786","https://openalex.org/W4400270505"],"related_works":[],"abstract_inverted_index":{"Hereditary":[0],"angioedema":[1],"(HAE)":[2],"due":[3],"to":[4,33],"C1":[5],"esterase":[6],"inhibitor":[7],"(C1-INH)":[8],"deficiency":[9],"is":[10,221],"a":[11,38,131,139,159,167,222],"rare":[12],"bradykinin-mediated":[13],"disease":[14],"characterized":[15],"by":[16,200],"recurrent":[17],"episodes":[18,45],"of":[19,46,65,147,169,203,213,218,235],"angioedema.":[20],"Gastrointestinal":[21],"manifestations":[22],"can":[23],"resemble":[24],"an":[25],"acute":[26,70,81],"abdomen":[27],"and":[28,72,88,123,138,158,178,181,208,231],"are":[29],"often":[30],"misdiagnosed,":[31],"contributing":[32],"delayed":[34],"recognition.":[35],"We":[36],"describe":[37],"35-year-old":[39],"man":[40],"with":[41,49,74,80,105,121],"two":[42],"previous":[43],"self-limited":[44,196],"abdominal":[47,86,97,197,214],"pain":[48,87,198],"diarrhea":[50],"over":[51],"the":[52,114,233],"preceding":[53],"year,":[54],"beginning":[55],"in":[56],"August":[57],"2024,":[58],"each":[59],"lasting":[60],"approximately":[61],"five":[62],"days,":[63],"both":[64],"which":[66],"were":[67],"diagnosed":[68],"as":[69],"gastroenteritis":[71],"treated":[73],"symptomatic":[75],"intravenous":[76],"therapy.":[77],"He":[78],"presented":[79],"paroxysmal":[82],"right":[83],"lower":[84],"quadrant":[85],"watery":[89],"diarrhea,":[90],"without":[91],"fever,":[92],"nausea,":[93],"or":[94],"vomiting.":[95],"Contrast-enhanced":[96],"computed":[98],"tomography":[99],"revealed":[100],"segmental":[101],"ileal":[102],"wall":[103,206],"edema":[104,207],"ascites,":[106],"showing":[107],"slight":[108],"progression":[109],"on":[110],"repeat":[111],"imaging":[112,201],"later":[113],"same":[115],"day.":[116],"Laboratory":[117],"analysis":[118],"demonstrated":[119],"leukocytosis":[120],"neutrophilia":[122],"significantly":[124,132],"elevated":[125],"D-dimer":[126],"levels.":[127],"Complement":[128],"testing":[129,230],"showed":[130],"reduced":[133,160],"C4":[134,220],"concentration":[135],"(0.08":[136],"g/L)":[137],"normal":[140],"C3":[141],"level":[142,163],"(0.84":[143],"g/L),":[144],"raising":[145],"suspicion":[146],"HAE.":[148,172,215],"Further":[149],"evaluation":[150],"confirmed":[151],"profoundly":[152],"decreased":[153],"functional":[154],"C1-INH":[155,161],"activity":[156],"(<7%)":[157],"antigen":[162],"(18.89":[164],"\u03bcg/mL),":[165],"establishing":[166],"diagnosis":[168],"type":[170],"I":[171],"The":[173],"patient":[174],"improved":[175],"following":[176],"supportive":[177],"antispasmodic":[179],"treatment,":[180],"no":[182],"HAE-specific":[183],"bradykinin-targeted":[184],"therapy":[185],"was":[186],"administered":[187],"during":[188],"this":[189],"admission.":[190],"This":[191],"case":[192],"emphasizes":[193],"that":[194,226],"recurrent,":[195],"accompanied":[199],"evidence":[202],"transient":[204],"small-bowel":[205],"ascites":[209],"should":[210],"prompt":[211],"consideration":[212],"Early":[216],"assessment":[217],"complement":[219],"practical":[223],"screening":[224],"strategy":[225],"enables":[227],"timely":[228],"confirmatory":[229],"minimizes":[232],"likelihood":[234],"repeated":[236],"misdiagnosis.":[237]},"counts_by_year":[],"updated_date":"2026-09-26T08:19:03.415552","created_date":"2026-04-28T00:00:00"}
