{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,8]],"date-time":"2026-05-08T08:17:34Z","timestamp":1778228254044,"version":"3.51.4"},"reference-count":33,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2022,6,27]],"date-time":"2022-06-27T00:00:00Z","timestamp":1656288000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Aliment Pharmacol Ther"],"published-print":{"date-parts":[[2022,9]]},"abstract":"<jats:title>Summary<\/jats:title><jats:sec><jats:title>Background and Aims<\/jats:title><jats:p>To evaluate symptom presentation and underlying pathophysiology of colonic\/anorectal dysfunction in females with functional constipation (FC) and hypermobile Ehlers\u2013Danlos syndrome (hEDS)\/hypermobility spectrum disorder (HSD)<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Case\u2013control study of 67 consecutive female patients with an established diagnosis of hEDS\/HSD referred to a specialist centre for investigation of FC (Rome III criteria), age\u2010matched (1:2 ratio) to 134 female controls with FC scoring 0 on the validated 5\u2010point joint hypermobility questionnaire. Symptoms and results of colonic\/anorectal physiology testing were compared. An independent series of 72 consecutive females with hEDS\/HSD, referred to a separate hospital for investigation of FC, was used to validate physiological findings.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Females with hEDS\/HSD were more likely to report constipation for \u2265 5\u2009years (76.1% vs. 61.2%, <jats:italic>p<\/jats:italic>\u00a0=\u20090.035), and a greater proportion had a high Cleveland Clinic constipation score (\u226512: 97.0% vs. 87.3%; <jats:italic>p<\/jats:italic>\u00a0=\u20090.027). The proportions with delayed whole\u2010gut transit were similar between groups (35.3% vs. 41.7%; <jats:italic>p<\/jats:italic>\u00a0=\u20090.462), as were the proportions with functional or structural abnormalities on defaecography (functional: 47.8% vs. 36.6%; <jats:italic>p<\/jats:italic>\u00a0=\u20090.127; structural: 65.7% vs. 66.4%; <jats:italic>p<\/jats:italic>\u00a0=\u20090.916). However, rectal hyposensitivity was more common in those with hEDS\/HSD (43.3% vs. 20.1%; <jats:italic>p<\/jats:italic>\u00a0=\u20090.0006); this was confirmed in the validation cohort (rectal hyposensitivity: 45.8%).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Rectal hyposensitivity is a common pathophysiological factor in females with FC and hEDS\/HSD as confirmed in two separate cohorts. The rectal hyposensitivity may be due to altered rectal biomechanics\/neuronal pathway dysfunction. Management may be better focused on enhancement of sensory perception (e.g., sensory biofeedback).<\/jats:p><\/jats:sec>","DOI":"10.1111\/apt.17104","type":"journal-article","created":{"date-parts":[[2022,6,27]],"date-time":"2022-06-27T13:34:17Z","timestamp":1656336857000},"page":"802-813","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["Rectal hyposensitivity: a common pathophysiological finding in patients with constipation and associated hypermobile Ehlers\u2013Danlos syndrome"],"prefix":"10.1111","volume":"56","author":[{"given":"Anisa","family":"Choudhary","sequence":"first","affiliation":[{"name":"Centre for Neuroscience, Surgery and Trauma, Blizard Institute, Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry Queen Mary University of London  London UK"},{"name":"GI Physiology Unit Barts Health NHS Trust  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3158-6652","authenticated-orcid":false,"given":"Paul F.","family":"Vollebregt","sequence":"additional","affiliation":[{"name":"Centre for Neuroscience, Surgery and Trauma, Blizard Institute, Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry Queen Mary University of London  London UK"},{"name":"GI Physiology Unit Barts Health NHS Trust  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qasim","family":"Aziz","sequence":"additional","affiliation":[{"name":"Centre for Neuroscience, Surgery and Trauma, Blizard Institute, Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry Queen Mary University of London  London UK"},{"name":"The Princess Grace Hospital HCA Healthcare  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S. Mark","family":"Scott","sequence":"additional","affiliation":[{"name":"Centre for Neuroscience, Surgery and Trauma, Blizard Institute, Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry Queen Mary University of London  London UK"},{"name":"GI Physiology Unit Barts Health NHS Trust  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Asma","family":"Fikree","sequence":"additional","affiliation":[{"name":"Centre for Neuroscience, Surgery and Trauma, Blizard Institute, Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry Queen Mary University of London  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,6,27]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31552"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31538"},{"key":"e_1_2_8_4_1","doi-asserted-by":"crossref","first-page":"163","DOI":"10.1111\/j.1742-1241.2003.tb10455.x","article-title":"A simple questionnaire to detect hypermobility: an adjunct to the assessment of patients with diffuse musculoskeletal pain","volume":"57","author":"Hakim AJ","year":"2003","journal-title":"Int J Clin Pract"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cgh.2014.01.014"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cgh.2020.02.034"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/nmo.12665"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0080321"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2019.12.034"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1572-0241.2006.00604.x"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/0016-5085(86)90941-8"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.14309\/ajg.0000000000001039"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2982.2010.01562.x"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2982.2009.01421.x"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980428)77:1<31::AID-AJMG8>3.0.CO;2-O"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.eclinm.2020.100572"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2005.11.061"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02056950"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2005.11.064"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1136\/gut.44.1.77"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/nmo.13839"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cgh.2019.11.049"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/apt.15039"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1471-0528.2006.00860.x"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1067\/mob.2002.123819"},{"issue":"508","key":"e_1_2_8_26_1","first-page":"e4","article-title":"Rectal hyposensitivity: pathophysiological mechanisms","volume":"21","author":"Gladman MA","year":"2009","journal-title":"Neurogastroenterol Motil"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1111\/nmo.13679"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61031"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1111\/nmo.12520"},{"key":"e_1_2_8_30_1","first-page":"e14226","article-title":"Barostat or syringe\u2010assisted sensory biofeedback training for constipation with rectal hyposensitivity: a randomized controlled trial","volume":"34","author":"Rao SSC","year":"2021","journal-title":"Neurogastroenterol Motil"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1097\/SLA.0b013e318247d49f"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.14309\/ajg.0000000000000816"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2013.07.014"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1111\/apt.13149"}],"container-title":["Alimentary Pharmacology &amp; 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