{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,18]],"date-time":"2026-03-18T01:22:13Z","timestamp":1773796933808,"version":"3.50.1"},"reference-count":33,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2024,4,8]],"date-time":"2024-04-08T00:00:00Z","timestamp":1712534400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Sleep Research"],"published-print":{"date-parts":[[2024,12]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Obstructive sleep apnea (OSA) is common in sickle cell disease (SCD) despite the absence of overweight, suggesting a specific pathophysiology. We previously showed that otherwise healthy children with increased pharyngeal compliance, a main endotype of OSA, exhibited decreased sympathetic modulation. Our objective was to assess whether modifications of heart rate variability (HRV) and compliance are associated in SCD. Cases (children with SCD, African or Caribbean ethnicity) and controls (otherwise healthy children, same ethnicity), aged 4\u201318\u2009years, were selected from our database of children referred for OSA and matched for sex, age, and obstructive apnea\u2013hypopnoea index (OAHI) score. The children underwent polysomnography and acoustic pharyngometry (to compute compliance). HRV analyses were performed from 5 min ECG recordings in wakeful, NREM, and REM sleep states and from the whole night. Twenty\u2010one pairs were analysed (median age 10.5\u2009years, 24 girls). Children with SCD had lower BMI z\u2010scores and more tonsil hypertrophy than control children. Children with SCD and OSA (OAHI \u22652\/hour) were characterised by lower compliance than children with SCD without OSA. An inverse relationship between compliance and SD2 (HRV from whole night, inversely related to sympathetic modulation) was evidenced (negative relationship in SCD: <jats:italic>R<\/jats:italic>\u2009=\u2009\u22120.63, <jats:italic>p<\/jats:italic>\u2009=\u20090.002 vs. positive relationship in controls <jats:italic>R<\/jats:italic>\u2009=\u20090.59, <jats:italic>p<\/jats:italic>\u2009=\u20090.006). In conclusion, while the decrease in sympathetic modulation in control children may contribute to increasing pharyngeal compliance, its decrease seems protective in children with sickle cell disease, which underlines the specificity of OSAS pathophysiology in SCD that could be due to sickle cell disease related smooth muscle dystonia.<\/jats:p>","DOI":"10.1111\/jsr.14209","type":"journal-article","created":{"date-parts":[[2024,4,9]],"date-time":"2024-04-09T06:55:50Z","timestamp":1712645750000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Case\u2013control study of heart rate variability and sleep apnea in childhood sickle cell disease"],"prefix":"10.1111","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0604-2482","authenticated-orcid":false,"given":"Plamen","family":"Bokov","sequence":"first","affiliation":[{"name":"AP\u2010HP, Robert Debr\u00e9 Hospital, Department of Physiology, INSERM NeuroDiderot Paris University  Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"B\u00e9reng\u00e8re","family":"Koehl","sequence":"additional","affiliation":[{"name":"AP\u2010HP, Robert Debr\u00e9 Hospital, Sickle Cell Disease Center, Hematology Unit Paris University  Paris France"},{"name":"UMR_S1134, BIGR, INSERM, Institut National de la Transfusion Sanguine, Laboratoire d'Excellence GR\u2010Ex  Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Benjamin","family":"Dudoignon","sequence":"additional","affiliation":[{"name":"AP\u2010HP, Robert Debr\u00e9 Hospital, Department of Physiology, INSERM NeuroDiderot Paris University  Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Malika","family":"Benkerrou","sequence":"additional","affiliation":[{"name":"Sickle Cell Disease Center, Hematology Unit Robert Debr\u00e9 Hospital  Paris France"},{"name":"INSERM UMR 1123, ECEVE  Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2786-0812","authenticated-orcid":false,"given":"Christophe","family":"Delclaux","sequence":"additional","affiliation":[{"name":"AP\u2010HP, Robert Debr\u00e9 Hospital, Department of Physiology, INSERM NeuroDiderot Paris University  Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,4,8]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.5664\/jcsm.10256"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.CIR.93.5.1043"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/jsr.13867"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.3389\/fphys.2020.00031"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.sleep.2019.03.003"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.sleep.2020.01.023"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/jsr.13337"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0031-3955(16)36806-7"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1113\/expphysiol.2010.056259"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/s40101-019-0193-2"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/PSY.0000000000000133"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.5665\/SLEEP.1046"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1183\/13993003.00385-2015"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.3389\/fneur.2022.775709"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.3389\/fcvm.2020.00019"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jelectrocard.2015.11.004"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.25632"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnins.2018.00997"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1177\/000348948909800302"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1097\/MCP.0000000000000724"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1155\/2018\/6920420"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.sleep.2016.01.021"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.3389\/fphys.2016.00557"},{"key":"e_1_2_9_25_1","first-page":"755","article-title":"Sleep\u2010disordered breathing in patients with sickle cell disease","volume":"97","author":"Raghunathan V. M.","year":"2018","journal-title":"Annals of Hematology"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.293.13.1653"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(03)14689-2"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.3389\/fpubh.2017.00258"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.13916"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pmedr.2021.101638"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1378\/chest.11-2013"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.chest.2020.01.012"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1093\/sleep\/zsaa157"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.smrv.2005.04.005"}],"container-title":["Journal of Sleep Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jsr.14209","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,2]],"date-time":"2025-04-02T00:54:09Z","timestamp":1743555249000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/jsr.14209"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,4,8]]},"references-count":33,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2024,12]]}},"alternative-id":["10.1111\/jsr.14209"],"URL":"https:\/\/doi.org\/10.1111\/jsr.14209","archive":["Portico"],"relation":{},"ISSN":["0962-1105","1365-2869"],"issn-type":[{"value":"0962-1105","type":"print"},{"value":"1365-2869","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,4,8]]},"assertion":[{"value":"2023-09-12","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-03-28","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-04-08","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e14209"}}