Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center
Purpose Mucopolysaccharidosis (MPS) encompasses a group of rare lysosomal storage disorders that are associated with the accumulation of glycosaminoglycans in organs and tissues. Respiratory disorders occur in all MPS types. In these patients, the prevalence of obstructive sleep apnea syndrome (OSAS...
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| Veröffentlicht in: | Sleep & breathing 2014-12, Vol.18 (4), p.791-797 |
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| creator | Moreira, Gustavo A. Kyosen, Sandra O. Patti, Camilla L. Martins, Ana Maria Tufik, Sergio |
| description | Purpose
Mucopolysaccharidosis (MPS) encompasses a group of rare lysosomal storage disorders that are associated with the accumulation of glycosaminoglycans in organs and tissues. Respiratory disorders occur in all MPS types. In these patients, the prevalence of obstructive sleep apnea syndrome (OSAS), which may confer additional morbidity, remains overlooked, and the results of the few existing studies are controversial. The present study aimed to characterize the prevalence of OSAS in patients with MPS types I, II, and VI in a reference center.
Methods
Forty-five patients with MPS (I,
n
= 17; II,
n
= 16; and VI;
n
= 12) in the Centro de Referência em Erros Inatos do Metabolismo, who underwent full-night polysomnography, were enrolled in a retrospective study. Demographic data and clinical history were collected from medical records of the first medical consultation.
Results
The prevalence of OSAS in patients with MPS was 69.8 %. MPS type I patients seemed to be more susceptible to OSA-induced hypoxemia, as indicated by reduced mean SpO
2
levels during both NREM and rapid eye movement sleep as well as during SpO
2
nadir.
Conclusions
Patients with MPS displayed a high prevalence of OSAS, often with moderate to high severity. Together, our results reinforce the need for OSAS screening in all patients with MPS. |
| doi_str_mv | 10.1007/s11325-014-0944-6 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_1621214256</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>3481838641</sourcerecordid><originalsourceid>FETCH-LOGICAL-c472t-2fddfb96eccc147fca4a0998ea3c11f7ea3cfc15d3385e278a226bdc7bc7fdcb3</originalsourceid><addsrcrecordid>eNp1kU1v1DAQhiNERUvhB3BBlhASBwIe58Pxsar4WKkSHICr5UzGNFXWCZ5k0f77Ot0FqkocrLE0z7zz8WbZC5DvQEr9ngEKVeUSylyasszrR9kZVErloKV5fPeXualAnWZPmW9kAhsDT7JTVWpZNVKfZbuvkXZuoIAkRi_Glue44NzvSPBANAk3BXKiD2Jyc09hZvG7n6_FdsFxGoc9O8RrF_tu5J7FvJ-Ixeat2KTnQid-bNZSJyJ5indNMGlQfJadeDcwPT_G8-z7xw_fLj_nV18-bS4vrnIstZpz5bvOt6YmRIRSe3Slk8Y05AoE8HqNHqHqiqKpSOnGKVW3HeoWte-wLc6zNwfdKY6_FuLZbntGGgYXaFzYQq1AQamqOqGvHqA34xJDmi5RYAwopU2i4EBhHJnTWnaK_dbFvQVpV1PswRSbbm1XU-yq_PKovLRb6v5W_HEhAa-PgGN0g48uYM__uMaoRhuZOHXgOKXCT4r3Rvxv91sKz6WM</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1619912279</pqid></control><display><type>article</type><title>Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center</title><source>MEDLINE</source><source>SpringerLink Journals - AutoHoldings</source><creator>Moreira, Gustavo A. ; Kyosen, Sandra O. ; Patti, Camilla L. ; Martins, Ana Maria ; Tufik, Sergio</creator><creatorcontrib>Moreira, Gustavo A. ; Kyosen, Sandra O. ; Patti, Camilla L. ; Martins, Ana Maria ; Tufik, Sergio</creatorcontrib><description>Purpose
Mucopolysaccharidosis (MPS) encompasses a group of rare lysosomal storage disorders that are associated with the accumulation of glycosaminoglycans in organs and tissues. Respiratory disorders occur in all MPS types. In these patients, the prevalence of obstructive sleep apnea syndrome (OSAS), which may confer additional morbidity, remains overlooked, and the results of the few existing studies are controversial. The present study aimed to characterize the prevalence of OSAS in patients with MPS types I, II, and VI in a reference center.
Methods
Forty-five patients with MPS (I,
n
= 17; II,
n
= 16; and VI;
n
= 12) in the Centro de Referência em Erros Inatos do Metabolismo, who underwent full-night polysomnography, were enrolled in a retrospective study. Demographic data and clinical history were collected from medical records of the first medical consultation.
Results
The prevalence of OSAS in patients with MPS was 69.8 %. MPS type I patients seemed to be more susceptible to OSA-induced hypoxemia, as indicated by reduced mean SpO
2
levels during both NREM and rapid eye movement sleep as well as during SpO
2
nadir.
Conclusions
Patients with MPS displayed a high prevalence of OSAS, often with moderate to high severity. Together, our results reinforce the need for OSAS screening in all patients with MPS.</description><identifier>ISSN: 1520-9512</identifier><identifier>EISSN: 1522-1709</identifier><identifier>DOI: 10.1007/s11325-014-0944-6</identifier><identifier>PMID: 24705807</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer Berlin Heidelberg</publisher><subject>Adolescent ; Biological and medical sciences ; Brazil ; Carbohydrates (enzymatic deficiencies). Glycogenosis ; Child ; Child, Preschool ; Comorbidity ; Cross-Sectional Studies ; Dentistry ; Disorders of higher nervous function. Focal brain diseases. Central vestibular syndrome and deafness. Brain stem syndromes ; Errors of metabolism ; Female ; Fundamental and applied biological sciences. Psychology ; Humans ; Hypoxia ; Infant ; Internal Medicine ; Male ; Medical sciences ; Medical screening ; Medicine ; Medicine & Public Health ; Metabolic diseases ; Metabolism ; Mucopolysaccharidosis I - diagnosis ; Mucopolysaccharidosis I - epidemiology ; Mucopolysaccharidosis II - diagnosis ; Mucopolysaccharidosis II - epidemiology ; Mucopolysaccharidosis VI - diagnosis ; Mucopolysaccharidosis VI - epidemiology ; Nervous system (semeiology, syndromes) ; Neurology ; Original Article ; Otorhinolaryngology ; Pediatrics ; Pneumology ; Pneumology/Respiratory System ; Polysomnography ; Respiration ; Respiratory system : syndromes and miscellaneous diseases ; Retrospective Studies ; Sleep Apnea, Obstructive - diagnosis ; Sleep Apnea, Obstructive - epidemiology ; Sleep disorders ; Sleep. Vigilance ; Vertebrates: nervous system and sense organs ; Young Adult</subject><ispartof>Sleep & breathing, 2014-12, Vol.18 (4), p.791-797</ispartof><rights>Springer-Verlag Berlin Heidelberg 2014</rights><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c472t-2fddfb96eccc147fca4a0998ea3c11f7ea3cfc15d3385e278a226bdc7bc7fdcb3</citedby><cites>FETCH-LOGICAL-c472t-2fddfb96eccc147fca4a0998ea3c11f7ea3cfc15d3385e278a226bdc7bc7fdcb3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s11325-014-0944-6$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s11325-014-0944-6$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,780,784,27924,27925,41488,42557,51319</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=28928790$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/24705807$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Moreira, Gustavo A.</creatorcontrib><creatorcontrib>Kyosen, Sandra O.</creatorcontrib><creatorcontrib>Patti, Camilla L.</creatorcontrib><creatorcontrib>Martins, Ana Maria</creatorcontrib><creatorcontrib>Tufik, Sergio</creatorcontrib><title>Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center</title><title>Sleep & breathing</title><addtitle>Sleep Breath</addtitle><addtitle>Sleep Breath</addtitle><description>Purpose
Mucopolysaccharidosis (MPS) encompasses a group of rare lysosomal storage disorders that are associated with the accumulation of glycosaminoglycans in organs and tissues. Respiratory disorders occur in all MPS types. In these patients, the prevalence of obstructive sleep apnea syndrome (OSAS), which may confer additional morbidity, remains overlooked, and the results of the few existing studies are controversial. The present study aimed to characterize the prevalence of OSAS in patients with MPS types I, II, and VI in a reference center.
Methods
Forty-five patients with MPS (I,
n
= 17; II,
n
= 16; and VI;
n
= 12) in the Centro de Referência em Erros Inatos do Metabolismo, who underwent full-night polysomnography, were enrolled in a retrospective study. Demographic data and clinical history were collected from medical records of the first medical consultation.
Results
The prevalence of OSAS in patients with MPS was 69.8 %. MPS type I patients seemed to be more susceptible to OSA-induced hypoxemia, as indicated by reduced mean SpO
2
levels during both NREM and rapid eye movement sleep as well as during SpO
2
nadir.
Conclusions
Patients with MPS displayed a high prevalence of OSAS, often with moderate to high severity. Together, our results reinforce the need for OSAS screening in all patients with MPS.</description><subject>Adolescent</subject><subject>Biological and medical sciences</subject><subject>Brazil</subject><subject>Carbohydrates (enzymatic deficiencies). Glycogenosis</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Comorbidity</subject><subject>Cross-Sectional Studies</subject><subject>Dentistry</subject><subject>Disorders of higher nervous function. Focal brain diseases. Central vestibular syndrome and deafness. Brain stem syndromes</subject><subject>Errors of metabolism</subject><subject>Female</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>Humans</subject><subject>Hypoxia</subject><subject>Infant</subject><subject>Internal Medicine</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Medical screening</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Metabolic diseases</subject><subject>Metabolism</subject><subject>Mucopolysaccharidosis I - diagnosis</subject><subject>Mucopolysaccharidosis I - epidemiology</subject><subject>Mucopolysaccharidosis II - diagnosis</subject><subject>Mucopolysaccharidosis II - epidemiology</subject><subject>Mucopolysaccharidosis VI - diagnosis</subject><subject>Mucopolysaccharidosis VI - epidemiology</subject><subject>Nervous system (semeiology, syndromes)</subject><subject>Neurology</subject><subject>Original Article</subject><subject>Otorhinolaryngology</subject><subject>Pediatrics</subject><subject>Pneumology</subject><subject>Pneumology/Respiratory System</subject><subject>Polysomnography</subject><subject>Respiration</subject><subject>Respiratory system : syndromes and miscellaneous diseases</subject><subject>Retrospective Studies</subject><subject>Sleep Apnea, Obstructive - diagnosis</subject><subject>Sleep Apnea, Obstructive - epidemiology</subject><subject>Sleep disorders</subject><subject>Sleep. Vigilance</subject><subject>Vertebrates: nervous system and sense organs</subject><subject>Young Adult</subject><issn>1520-9512</issn><issn>1522-1709</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><recordid>eNp1kU1v1DAQhiNERUvhB3BBlhASBwIe58Pxsar4WKkSHICr5UzGNFXWCZ5k0f77Ot0FqkocrLE0z7zz8WbZC5DvQEr9ngEKVeUSylyasszrR9kZVErloKV5fPeXualAnWZPmW9kAhsDT7JTVWpZNVKfZbuvkXZuoIAkRi_Glue44NzvSPBANAk3BXKiD2Jyc09hZvG7n6_FdsFxGoc9O8RrF_tu5J7FvJ-Ixeat2KTnQid-bNZSJyJ5indNMGlQfJadeDcwPT_G8-z7xw_fLj_nV18-bS4vrnIstZpz5bvOt6YmRIRSe3Slk8Y05AoE8HqNHqHqiqKpSOnGKVW3HeoWte-wLc6zNwfdKY6_FuLZbntGGgYXaFzYQq1AQamqOqGvHqA34xJDmi5RYAwopU2i4EBhHJnTWnaK_dbFvQVpV1PswRSbbm1XU-yq_PKovLRb6v5W_HEhAa-PgGN0g48uYM__uMaoRhuZOHXgOKXCT4r3Rvxv91sKz6WM</recordid><startdate>20141201</startdate><enddate>20141201</enddate><creator>Moreira, Gustavo A.</creator><creator>Kyosen, Sandra O.</creator><creator>Patti, Camilla L.</creator><creator>Martins, Ana Maria</creator><creator>Tufik, Sergio</creator><general>Springer Berlin Heidelberg</general><general>Springer</general><general>Springer Nature B.V</general><scope>IQODW</scope><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>0-V</scope><scope>3V.</scope><scope>7T5</scope><scope>7TK</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>88G</scope><scope>88J</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>ALSLI</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>H94</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2M</scope><scope>M2R</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>PSYQQ</scope><scope>Q9U</scope><scope>7X8</scope></search><sort><creationdate>20141201</creationdate><title>Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center</title><author>Moreira, Gustavo A. ; Kyosen, Sandra O. ; Patti, Camilla L. ; Martins, Ana Maria ; Tufik, Sergio</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c472t-2fddfb96eccc147fca4a0998ea3c11f7ea3cfc15d3385e278a226bdc7bc7fdcb3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>Adolescent</topic><topic>Biological and medical sciences</topic><topic>Brazil</topic><topic>Carbohydrates (enzymatic deficiencies). Glycogenosis</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Comorbidity</topic><topic>Cross-Sectional Studies</topic><topic>Dentistry</topic><topic>Disorders of higher nervous function. Focal brain diseases. Central vestibular syndrome and deafness. Brain stem syndromes</topic><topic>Errors of metabolism</topic><topic>Female</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>Humans</topic><topic>Hypoxia</topic><topic>Infant</topic><topic>Internal Medicine</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Medical screening</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Metabolic diseases</topic><topic>Metabolism</topic><topic>Mucopolysaccharidosis I - diagnosis</topic><topic>Mucopolysaccharidosis I - epidemiology</topic><topic>Mucopolysaccharidosis II - diagnosis</topic><topic>Mucopolysaccharidosis II - epidemiology</topic><topic>Mucopolysaccharidosis VI - diagnosis</topic><topic>Mucopolysaccharidosis VI - epidemiology</topic><topic>Nervous system (semeiology, syndromes)</topic><topic>Neurology</topic><topic>Original Article</topic><topic>Otorhinolaryngology</topic><topic>Pediatrics</topic><topic>Pneumology</topic><topic>Pneumology/Respiratory System</topic><topic>Polysomnography</topic><topic>Respiration</topic><topic>Respiratory system : syndromes and miscellaneous diseases</topic><topic>Retrospective Studies</topic><topic>Sleep Apnea, Obstructive - diagnosis</topic><topic>Sleep Apnea, Obstructive - epidemiology</topic><topic>Sleep disorders</topic><topic>Sleep. Vigilance</topic><topic>Vertebrates: nervous system and sense organs</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Moreira, Gustavo A.</creatorcontrib><creatorcontrib>Kyosen, Sandra O.</creatorcontrib><creatorcontrib>Patti, Camilla L.</creatorcontrib><creatorcontrib>Martins, Ana Maria</creatorcontrib><creatorcontrib>Tufik, Sergio</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Social Sciences Premium Collection</collection><collection>ProQuest Central (Corporate)</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Psychology Database (Alumni)</collection><collection>Social Science Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>Social Science Premium Collection</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Psychology Database</collection><collection>Social Science Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest One Psychology</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><jtitle>Sleep & breathing</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Moreira, Gustavo A.</au><au>Kyosen, Sandra O.</au><au>Patti, Camilla L.</au><au>Martins, Ana Maria</au><au>Tufik, Sergio</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center</atitle><jtitle>Sleep & breathing</jtitle><stitle>Sleep Breath</stitle><addtitle>Sleep Breath</addtitle><date>2014-12-01</date><risdate>2014</risdate><volume>18</volume><issue>4</issue><spage>791</spage><epage>797</epage><pages>791-797</pages><issn>1520-9512</issn><eissn>1522-1709</eissn><abstract>Purpose
Mucopolysaccharidosis (MPS) encompasses a group of rare lysosomal storage disorders that are associated with the accumulation of glycosaminoglycans in organs and tissues. Respiratory disorders occur in all MPS types. In these patients, the prevalence of obstructive sleep apnea syndrome (OSAS), which may confer additional morbidity, remains overlooked, and the results of the few existing studies are controversial. The present study aimed to characterize the prevalence of OSAS in patients with MPS types I, II, and VI in a reference center.
Methods
Forty-five patients with MPS (I,
n
= 17; II,
n
= 16; and VI;
n
= 12) in the Centro de Referência em Erros Inatos do Metabolismo, who underwent full-night polysomnography, were enrolled in a retrospective study. Demographic data and clinical history were collected from medical records of the first medical consultation.
Results
The prevalence of OSAS in patients with MPS was 69.8 %. MPS type I patients seemed to be more susceptible to OSA-induced hypoxemia, as indicated by reduced mean SpO
2
levels during both NREM and rapid eye movement sleep as well as during SpO
2
nadir.
Conclusions
Patients with MPS displayed a high prevalence of OSAS, often with moderate to high severity. Together, our results reinforce the need for OSAS screening in all patients with MPS.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer Berlin Heidelberg</pub><pmid>24705807</pmid><doi>10.1007/s11325-014-0944-6</doi><tpages>7</tpages></addata></record> |
| fulltext | fulltext |
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| ispartof | Sleep & breathing, 2014-12, Vol.18 (4), p.791-797 |
| issn | 1520-9512 1522-1709 |
| language | eng |
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| subjects | Adolescent Biological and medical sciences Brazil Carbohydrates (enzymatic deficiencies). Glycogenosis Child Child, Preschool Comorbidity Cross-Sectional Studies Dentistry Disorders of higher nervous function. Focal brain diseases. Central vestibular syndrome and deafness. Brain stem syndromes Errors of metabolism Female Fundamental and applied biological sciences. Psychology Humans Hypoxia Infant Internal Medicine Male Medical sciences Medical screening Medicine Medicine & Public Health Metabolic diseases Metabolism Mucopolysaccharidosis I - diagnosis Mucopolysaccharidosis I - epidemiology Mucopolysaccharidosis II - diagnosis Mucopolysaccharidosis II - epidemiology Mucopolysaccharidosis VI - diagnosis Mucopolysaccharidosis VI - epidemiology Nervous system (semeiology, syndromes) Neurology Original Article Otorhinolaryngology Pediatrics Pneumology Pneumology/Respiratory System Polysomnography Respiration Respiratory system : syndromes and miscellaneous diseases Retrospective Studies Sleep Apnea, Obstructive - diagnosis Sleep Apnea, Obstructive - epidemiology Sleep disorders Sleep. Vigilance Vertebrates: nervous system and sense organs Young Adult |
| title | Prevalence of obstructive sleep apnea in patients with mucopolysaccharidosis types I, II, and VI in a reference center |
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