Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos)
Introduction Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare entity that mimics various inflammatory strictures of the small intestine. Pediatric literature is scarce. We analyzed the clinical, radiological, endoscopic and histopathological features of children with CMUSE that...
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| Veröffentlicht in: | Indian journal of gastroenterology 2024-12, Vol.43 (6), p.1144-1155 |
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| creator | Samanta, Arghya Sen Sarma, Moinak Singh, Sumit Kumar Srivastava, Anshu Poddar, Ujjal Mandelia, Ankur Agrawal, Vinita Yachha, Surendra Kumar Lal, Richa |
| description | Introduction
Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare entity that mimics various inflammatory strictures of the small intestine. Pediatric literature is scarce. We analyzed the clinical, radiological, endoscopic and histopathological features of children with CMUSE that differentiate it from small bowel Crohn’s disease (SBCD) and gastrointestinal tuberculosis (GITB).
Methods
CMUSE was diagnosed by the following criteria: (1) unexplained small bowel strictures with superficial ulcers, (2) chronic/relapsing ulcers of small bowel after resection, (3) no signs of systemic inflammation, (4) absence of other known etiologies of small bowel ulcers. SBCD and GITB were diagnosed based on standard criteria. The clinical features, laboratory parameters, radioimaging, endoscopy (including video capsule endoscopy [VCE], intra-operative endoscopy), histopathological features and treatment outcome were noted.
Results
Out of 48, CMUSE was diagnosed in 13 (27%) isolated small bowel and ileocecal strictures, while GITB and SBCD accounted for 41% and 21% cases, respectively. Common presentations were sub-acute obstruction (46%), obscure gastrointestinal bleeding (38%) and protein-losing enteropathy (38%). CMUSE patients had significantly longer disease duration compared to SBCD and GITB (
p
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| doi_str_mv | 10.1007/s12664-024-01604-3 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_3070839467</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>3070839467</sourcerecordid><originalsourceid>FETCH-LOGICAL-c298t-62d7f61803eee58897bf9ff1e9f6cd993b388bdb6036fa322e873057676df8dc3</originalsourceid><addsrcrecordid>eNp9UctuFDEQHCEQCYEf4IB8DIcBz3jWD27R8pQicYGz5bHbG0cee3F7Eu2N3-BT-B2-BIcNHDlY7lZXVbequu75QF8NlIrXOIycTz0d2xs4nXr2oDulSmx6KobhYavHzdRLyelJ9wTxmt71ij3uTphUjSWm0-7n24C2hCUkU0NOJHuyBxdMLcESWw77mneQWr2ssQafrYlkjRZKXpFghZQxpB2BVKGEGpD4kheCi4mRzPkWItmWfJV-ff-BxAUEg0BMcmRnsJYcGg1r2x1JXWcodo1ND9-QC1KgzXEPtoYbaJtWdyDnt6FekZvgIOPLp90jbyLCs_v_rPv6_t2X7cf-8vOHT9uLy96OStaej054PkjKAGAjpRKzV94PoDy3Tik2MylnN3PKuDdsHEEKRjeCC-68dJaddedH3X3J39Z2rl6aZRCjSdBM0IwKKpmauGjQ8Qi17XQs4PW-WWvKQQ9U30Wmj5HpZr7-E5lmjfTiXn-dF3D_KH8zagB2BGAbpR0UfZ3X0jzD_8n-Bk0AqQs</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>3070839467</pqid></control><display><type>article</type><title>Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos)</title><source>MEDLINE</source><source>SpringerLink Journals</source><creator>Samanta, Arghya ; Sen Sarma, Moinak ; Singh, Sumit Kumar ; Srivastava, Anshu ; Poddar, Ujjal ; Mandelia, Ankur ; Agrawal, Vinita ; Yachha, Surendra Kumar ; Lal, Richa</creator><creatorcontrib>Samanta, Arghya ; Sen Sarma, Moinak ; Singh, Sumit Kumar ; Srivastava, Anshu ; Poddar, Ujjal ; Mandelia, Ankur ; Agrawal, Vinita ; Yachha, Surendra Kumar ; Lal, Richa</creatorcontrib><description>Introduction
Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare entity that mimics various inflammatory strictures of the small intestine. Pediatric literature is scarce. We analyzed the clinical, radiological, endoscopic and histopathological features of children with CMUSE that differentiate it from small bowel Crohn’s disease (SBCD) and gastrointestinal tuberculosis (GITB).
Methods
CMUSE was diagnosed by the following criteria: (1) unexplained small bowel strictures with superficial ulcers, (2) chronic/relapsing ulcers of small bowel after resection, (3) no signs of systemic inflammation, (4) absence of other known etiologies of small bowel ulcers. SBCD and GITB were diagnosed based on standard criteria. The clinical features, laboratory parameters, radioimaging, endoscopy (including video capsule endoscopy [VCE], intra-operative endoscopy), histopathological features and treatment outcome were noted.
Results
Out of 48, CMUSE was diagnosed in 13 (27%) isolated small bowel and ileocecal strictures, while GITB and SBCD accounted for 41% and 21% cases, respectively. Common presentations were sub-acute obstruction (46%), obscure gastrointestinal bleeding (38%) and protein-losing enteropathy (38%). CMUSE patients had significantly longer disease duration compared to SBCD and GITB (
p
< 0.001). SBCD (90.0%) and GITB (85%) cases had elevated C-reactive protein (CRP), none with CMUSE had elevated CRP (
p
< 0.001). The disease was localized in jejunum (100%) and proximal ileum (56%) in CMUSE, ileocecal region (85%) in GITB, but evenly distributed in small intestine in SBCD. Endoscopy showed evenly placed, superficial, circumferential ulcers with strictures in CMUSE, deep linear ulcers in SBCD and circumferential ulcers in GITB. Upfront immunosuppression was given in four; three (75%) of them relapsed. Only surgery was done in three with one (25%) having relapse. Upfront surgery followed by immunosuppression was used in six, but all relapsed and two required repeat surgery.
Conclusion
CMUSE is important but underdiagnosed in children. Lack of constitutional symptoms, normal inflammatory parameters and characteristic ulcers with strictures helped in differentiating CMUSE from GITB and SBCD.
Graphical Abstract</description><identifier>ISSN: 0254-8860</identifier><identifier>ISSN: 0975-0711</identifier><identifier>EISSN: 0975-0711</identifier><identifier>DOI: 10.1007/s12664-024-01604-3</identifier><identifier>PMID: 38902474</identifier><language>eng</language><publisher>New Delhi: Springer India</publisher><subject>Adolescent ; Capsule Endoscopy ; Child ; Child, Preschool ; Constriction, Pathologic - etiology ; Crohn Disease - complications ; Crohn Disease - diagnosis ; Diagnosis, Differential ; Enteritis - complications ; Enteritis - diagnosis ; Enteritis - pathology ; Female ; Gastroenterology ; Hepatology ; Humans ; Intestinal Obstruction - etiology ; Intestinal Obstruction - surgery ; Intestine, Small - pathology ; Male ; Medicine ; Medicine & Public Health ; Original Article ; Retrospective Studies ; Tuberculosis, Gastrointestinal - complications ; Tuberculosis, Gastrointestinal - diagnosis ; Ulcer - etiology ; Ulcer - pathology</subject><ispartof>Indian journal of gastroenterology, 2024-12, Vol.43 (6), p.1144-1155</ispartof><rights>Indian Society of Gastroenterology 2024. Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.</rights><rights>2024. Indian Society of Gastroenterology.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c298t-62d7f61803eee58897bf9ff1e9f6cd993b388bdb6036fa322e873057676df8dc3</cites><orcidid>0000-0003-2015-4069</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s12664-024-01604-3$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s12664-024-01604-3$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,776,780,27901,27902,41464,42533,51294</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/38902474$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Samanta, Arghya</creatorcontrib><creatorcontrib>Sen Sarma, Moinak</creatorcontrib><creatorcontrib>Singh, Sumit Kumar</creatorcontrib><creatorcontrib>Srivastava, Anshu</creatorcontrib><creatorcontrib>Poddar, Ujjal</creatorcontrib><creatorcontrib>Mandelia, Ankur</creatorcontrib><creatorcontrib>Agrawal, Vinita</creatorcontrib><creatorcontrib>Yachha, Surendra Kumar</creatorcontrib><creatorcontrib>Lal, Richa</creatorcontrib><title>Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos)</title><title>Indian journal of gastroenterology</title><addtitle>Indian J Gastroenterol</addtitle><addtitle>Indian J Gastroenterol</addtitle><description>Introduction
Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare entity that mimics various inflammatory strictures of the small intestine. Pediatric literature is scarce. We analyzed the clinical, radiological, endoscopic and histopathological features of children with CMUSE that differentiate it from small bowel Crohn’s disease (SBCD) and gastrointestinal tuberculosis (GITB).
Methods
CMUSE was diagnosed by the following criteria: (1) unexplained small bowel strictures with superficial ulcers, (2) chronic/relapsing ulcers of small bowel after resection, (3) no signs of systemic inflammation, (4) absence of other known etiologies of small bowel ulcers. SBCD and GITB were diagnosed based on standard criteria. The clinical features, laboratory parameters, radioimaging, endoscopy (including video capsule endoscopy [VCE], intra-operative endoscopy), histopathological features and treatment outcome were noted.
Results
Out of 48, CMUSE was diagnosed in 13 (27%) isolated small bowel and ileocecal strictures, while GITB and SBCD accounted for 41% and 21% cases, respectively. Common presentations were sub-acute obstruction (46%), obscure gastrointestinal bleeding (38%) and protein-losing enteropathy (38%). CMUSE patients had significantly longer disease duration compared to SBCD and GITB (
p
< 0.001). SBCD (90.0%) and GITB (85%) cases had elevated C-reactive protein (CRP), none with CMUSE had elevated CRP (
p
< 0.001). The disease was localized in jejunum (100%) and proximal ileum (56%) in CMUSE, ileocecal region (85%) in GITB, but evenly distributed in small intestine in SBCD. Endoscopy showed evenly placed, superficial, circumferential ulcers with strictures in CMUSE, deep linear ulcers in SBCD and circumferential ulcers in GITB. Upfront immunosuppression was given in four; three (75%) of them relapsed. Only surgery was done in three with one (25%) having relapse. Upfront surgery followed by immunosuppression was used in six, but all relapsed and two required repeat surgery.
Conclusion
CMUSE is important but underdiagnosed in children. Lack of constitutional symptoms, normal inflammatory parameters and characteristic ulcers with strictures helped in differentiating CMUSE from GITB and SBCD.
Graphical Abstract</description><subject>Adolescent</subject><subject>Capsule Endoscopy</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Constriction, Pathologic - etiology</subject><subject>Crohn Disease - complications</subject><subject>Crohn Disease - diagnosis</subject><subject>Diagnosis, Differential</subject><subject>Enteritis - complications</subject><subject>Enteritis - diagnosis</subject><subject>Enteritis - pathology</subject><subject>Female</subject><subject>Gastroenterology</subject><subject>Hepatology</subject><subject>Humans</subject><subject>Intestinal Obstruction - etiology</subject><subject>Intestinal Obstruction - surgery</subject><subject>Intestine, Small - pathology</subject><subject>Male</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Original Article</subject><subject>Retrospective Studies</subject><subject>Tuberculosis, Gastrointestinal - complications</subject><subject>Tuberculosis, Gastrointestinal - diagnosis</subject><subject>Ulcer - etiology</subject><subject>Ulcer - pathology</subject><issn>0254-8860</issn><issn>0975-0711</issn><issn>0975-0711</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9UctuFDEQHCEQCYEf4IB8DIcBz3jWD27R8pQicYGz5bHbG0cee3F7Eu2N3-BT-B2-BIcNHDlY7lZXVbequu75QF8NlIrXOIycTz0d2xs4nXr2oDulSmx6KobhYavHzdRLyelJ9wTxmt71ij3uTphUjSWm0-7n24C2hCUkU0NOJHuyBxdMLcESWw77mneQWr2ssQafrYlkjRZKXpFghZQxpB2BVKGEGpD4kheCi4mRzPkWItmWfJV-ff-BxAUEg0BMcmRnsJYcGg1r2x1JXWcodo1ND9-QC1KgzXEPtoYbaJtWdyDnt6FekZvgIOPLp90jbyLCs_v_rPv6_t2X7cf-8vOHT9uLy96OStaej054PkjKAGAjpRKzV94PoDy3Tik2MylnN3PKuDdsHEEKRjeCC-68dJaddedH3X3J39Z2rl6aZRCjSdBM0IwKKpmauGjQ8Qi17XQs4PW-WWvKQQ9U30Wmj5HpZr7-E5lmjfTiXn-dF3D_KH8zagB2BGAbpR0UfZ3X0jzD_8n-Bk0AqQs</recordid><startdate>20241201</startdate><enddate>20241201</enddate><creator>Samanta, Arghya</creator><creator>Sen Sarma, Moinak</creator><creator>Singh, Sumit Kumar</creator><creator>Srivastava, Anshu</creator><creator>Poddar, Ujjal</creator><creator>Mandelia, Ankur</creator><creator>Agrawal, Vinita</creator><creator>Yachha, Surendra Kumar</creator><creator>Lal, Richa</creator><general>Springer India</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0003-2015-4069</orcidid></search><sort><creationdate>20241201</creationdate><title>Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos)</title><author>Samanta, Arghya ; Sen Sarma, Moinak ; Singh, Sumit Kumar ; Srivastava, Anshu ; Poddar, Ujjal ; Mandelia, Ankur ; Agrawal, Vinita ; Yachha, Surendra Kumar ; Lal, Richa</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c298t-62d7f61803eee58897bf9ff1e9f6cd993b388bdb6036fa322e873057676df8dc3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Adolescent</topic><topic>Capsule Endoscopy</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Constriction, Pathologic - etiology</topic><topic>Crohn Disease - complications</topic><topic>Crohn Disease - diagnosis</topic><topic>Diagnosis, Differential</topic><topic>Enteritis - complications</topic><topic>Enteritis - diagnosis</topic><topic>Enteritis - pathology</topic><topic>Female</topic><topic>Gastroenterology</topic><topic>Hepatology</topic><topic>Humans</topic><topic>Intestinal Obstruction - etiology</topic><topic>Intestinal Obstruction - surgery</topic><topic>Intestine, Small - pathology</topic><topic>Male</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Original Article</topic><topic>Retrospective Studies</topic><topic>Tuberculosis, Gastrointestinal - complications</topic><topic>Tuberculosis, Gastrointestinal - diagnosis</topic><topic>Ulcer - etiology</topic><topic>Ulcer - pathology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Samanta, Arghya</creatorcontrib><creatorcontrib>Sen Sarma, Moinak</creatorcontrib><creatorcontrib>Singh, Sumit Kumar</creatorcontrib><creatorcontrib>Srivastava, Anshu</creatorcontrib><creatorcontrib>Poddar, Ujjal</creatorcontrib><creatorcontrib>Mandelia, Ankur</creatorcontrib><creatorcontrib>Agrawal, Vinita</creatorcontrib><creatorcontrib>Yachha, Surendra Kumar</creatorcontrib><creatorcontrib>Lal, Richa</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Indian journal of gastroenterology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Samanta, Arghya</au><au>Sen Sarma, Moinak</au><au>Singh, Sumit Kumar</au><au>Srivastava, Anshu</au><au>Poddar, Ujjal</au><au>Mandelia, Ankur</au><au>Agrawal, Vinita</au><au>Yachha, Surendra Kumar</au><au>Lal, Richa</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos)</atitle><jtitle>Indian journal of gastroenterology</jtitle><stitle>Indian J Gastroenterol</stitle><addtitle>Indian J Gastroenterol</addtitle><date>2024-12-01</date><risdate>2024</risdate><volume>43</volume><issue>6</issue><spage>1144</spage><epage>1155</epage><pages>1144-1155</pages><issn>0254-8860</issn><issn>0975-0711</issn><eissn>0975-0711</eissn><abstract>Introduction
Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare entity that mimics various inflammatory strictures of the small intestine. Pediatric literature is scarce. We analyzed the clinical, radiological, endoscopic and histopathological features of children with CMUSE that differentiate it from small bowel Crohn’s disease (SBCD) and gastrointestinal tuberculosis (GITB).
Methods
CMUSE was diagnosed by the following criteria: (1) unexplained small bowel strictures with superficial ulcers, (2) chronic/relapsing ulcers of small bowel after resection, (3) no signs of systemic inflammation, (4) absence of other known etiologies of small bowel ulcers. SBCD and GITB were diagnosed based on standard criteria. The clinical features, laboratory parameters, radioimaging, endoscopy (including video capsule endoscopy [VCE], intra-operative endoscopy), histopathological features and treatment outcome were noted.
Results
Out of 48, CMUSE was diagnosed in 13 (27%) isolated small bowel and ileocecal strictures, while GITB and SBCD accounted for 41% and 21% cases, respectively. Common presentations were sub-acute obstruction (46%), obscure gastrointestinal bleeding (38%) and protein-losing enteropathy (38%). CMUSE patients had significantly longer disease duration compared to SBCD and GITB (
p
< 0.001). SBCD (90.0%) and GITB (85%) cases had elevated C-reactive protein (CRP), none with CMUSE had elevated CRP (
p
< 0.001). The disease was localized in jejunum (100%) and proximal ileum (56%) in CMUSE, ileocecal region (85%) in GITB, but evenly distributed in small intestine in SBCD. Endoscopy showed evenly placed, superficial, circumferential ulcers with strictures in CMUSE, deep linear ulcers in SBCD and circumferential ulcers in GITB. Upfront immunosuppression was given in four; three (75%) of them relapsed. Only surgery was done in three with one (25%) having relapse. Upfront surgery followed by immunosuppression was used in six, but all relapsed and two required repeat surgery.
Conclusion
CMUSE is important but underdiagnosed in children. Lack of constitutional symptoms, normal inflammatory parameters and characteristic ulcers with strictures helped in differentiating CMUSE from GITB and SBCD.
Graphical Abstract</abstract><cop>New Delhi</cop><pub>Springer India</pub><pmid>38902474</pmid><doi>10.1007/s12664-024-01604-3</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0003-2015-4069</orcidid></addata></record> |
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| subjects | Adolescent Capsule Endoscopy Child Child, Preschool Constriction, Pathologic - etiology Crohn Disease - complications Crohn Disease - diagnosis Diagnosis, Differential Enteritis - complications Enteritis - diagnosis Enteritis - pathology Female Gastroenterology Hepatology Humans Intestinal Obstruction - etiology Intestinal Obstruction - surgery Intestine, Small - pathology Male Medicine Medicine & Public Health Original Article Retrospective Studies Tuberculosis, Gastrointestinal - complications Tuberculosis, Gastrointestinal - diagnosis Ulcer - etiology Ulcer - pathology |
| title | Discrimination of pediatric cryptogenic multifocal ulcerous stenosing enteritis from small bowel Crohn’s disease and gastrointestinal tuberculosis: A retrospective study (with videos) |
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