A pilot longitudinal study of the use of waxy maize heat modified starch in the treatment of adults with glycogen storage disease type I: a randomized double-blind cross-over study.
Identifieur interne : 003047 ( PubMed/Corpus ); précédent : 003046; suivant : 003048A pilot longitudinal study of the use of waxy maize heat modified starch in the treatment of adults with glycogen storage disease type I: a randomized double-blind cross-over study.
Auteurs : Kaustuv Bhattacharya ; Helen Mundy ; Maggie F. Lilburn ; Michael P. Champion ; David W. Morley ; François MaillotSource :
- Orphanet journal of rare diseases [ 1750-1172 ] ; 2015.
English descriptors
- KwdEn :
- MESH :
- chemical , chemistry : Starch.
- chemical , drug effects : Blood Glucose.
- diet therapy : Glycogen Storage Disease Type I.
- chemical , pharmacology : Starch.
- Adolescent, Adult, Cross-Over Studies, Double-Blind Method, Female, Humans, Male, Pilot Projects, Young Adult.
Abstract
Uncooked corn-starch (UCCS) has been the mainstay of therapy for the hepatic glycogen storage diseases (GSD) but is not always effective. A new starch (WMHMS) has demonstrated a more favourable short-term metabolic profile.
DOI: 10.1186/s13023-015-0229-6
PubMed: 25758258
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">A pilot longitudinal study of the use of waxy maize heat modified starch in the treatment of adults with glycogen storage disease type I: a randomized double-blind cross-over study.</title><author><name sortKey="Bhattacharya, Kaustuv" sort="Bhattacharya, Kaustuv" uniqKey="Bhattacharya K" first="Kaustuv" last="Bhattacharya">Kaustuv Bhattacharya</name><affiliation><nlm:affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Kaustuv.Bhattacharya@health.nsw.gov.au.</nlm:affiliation></affiliation></author><author><name sortKey="Mundy, Helen" sort="Mundy, Helen" uniqKey="Mundy H" first="Helen" last="Mundy">Helen Mundy</name><affiliation><nlm:affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Helen.Mundy@gstt.nhs.uk.</nlm:affiliation></affiliation></author><author><name sortKey="Lilburn, Maggie F" sort="Lilburn, Maggie F" uniqKey="Lilburn M" first="Maggie F" last="Lilburn">Maggie F. Lilburn</name><affiliation><nlm:affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Maggie.lilburn@btinternet.com.</nlm:affiliation></affiliation></author><author><name sortKey="Champion, Michael P" sort="Champion, Michael P" uniqKey="Champion M" first="Michael P" last="Champion">Michael P. Champion</name><affiliation><nlm:affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Michael.Champion@gstt.nhs.uk.</nlm:affiliation></affiliation></author><author><name sortKey="Morley, David W" sort="Morley, David W" uniqKey="Morley D" first="David W" last="Morley">David W. Morley</name><affiliation><nlm:affiliation>Department of Epidemiology & Biostatistics, Imperial College, SW7 2AZ, London, UK. d.w.morley@gmail.com.</nlm:affiliation></affiliation></author><author><name sortKey="Maillot, Francois" sort="Maillot, Francois" uniqKey="Maillot F" first="François" last="Maillot">François Maillot</name><affiliation><nlm:affiliation>CHRU de Tours, 37044, Tours, Cedex, France. francois.maillot@univ-tours.fr.</nlm:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2015">2015</date><idno type="RBID">pubmed:25758258</idno><idno type="pmid">25758258</idno><idno type="doi">10.1186/s13023-015-0229-6</idno><idno type="wicri:Area/PubMed/Corpus">003047</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">003047</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">A pilot longitudinal study of the use of waxy maize heat modified starch in the treatment of adults with glycogen storage disease type I: a randomized double-blind cross-over study.</title><author><name sortKey="Bhattacharya, Kaustuv" sort="Bhattacharya, Kaustuv" uniqKey="Bhattacharya K" first="Kaustuv" last="Bhattacharya">Kaustuv Bhattacharya</name><affiliation><nlm:affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Kaustuv.Bhattacharya@health.nsw.gov.au.</nlm:affiliation></affiliation></author><author><name sortKey="Mundy, Helen" sort="Mundy, Helen" uniqKey="Mundy H" first="Helen" last="Mundy">Helen Mundy</name><affiliation><nlm:affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Helen.Mundy@gstt.nhs.uk.</nlm:affiliation></affiliation></author><author><name sortKey="Lilburn, Maggie F" sort="Lilburn, Maggie F" uniqKey="Lilburn M" first="Maggie F" last="Lilburn">Maggie F. Lilburn</name><affiliation><nlm:affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Maggie.lilburn@btinternet.com.</nlm:affiliation></affiliation></author><author><name sortKey="Champion, Michael P" sort="Champion, Michael P" uniqKey="Champion M" first="Michael P" last="Champion">Michael P. Champion</name><affiliation><nlm:affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Michael.Champion@gstt.nhs.uk.</nlm:affiliation></affiliation></author><author><name sortKey="Morley, David W" sort="Morley, David W" uniqKey="Morley D" first="David W" last="Morley">David W. Morley</name><affiliation><nlm:affiliation>Department of Epidemiology & Biostatistics, Imperial College, SW7 2AZ, London, UK. d.w.morley@gmail.com.</nlm:affiliation></affiliation></author><author><name sortKey="Maillot, Francois" sort="Maillot, Francois" uniqKey="Maillot F" first="François" last="Maillot">François Maillot</name><affiliation><nlm:affiliation>CHRU de Tours, 37044, Tours, Cedex, France. francois.maillot@univ-tours.fr.</nlm:affiliation></affiliation></author></analytic><series><title level="j">Orphanet journal of rare diseases</title><idno type="eISSN">1750-1172</idno><imprint><date when="2015" type="published">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Blood Glucose (drug effects)</term><term>Cross-Over Studies</term><term>Double-Blind Method</term><term>Female</term><term>Glycogen Storage Disease Type I (diet therapy)</term><term>Humans</term><term>Male</term><term>Pilot Projects</term><term>Starch (chemistry)</term><term>Starch (pharmacology)</term><term>Young Adult</term></keywords><keywords scheme="MESH" type="chemical" qualifier="chemistry" xml:lang="en"><term>Starch</term></keywords><keywords scheme="MESH" type="chemical" qualifier="drug effects" xml:lang="en"><term>Blood Glucose</term></keywords><keywords scheme="MESH" qualifier="diet therapy" xml:lang="en"><term>Glycogen Storage Disease Type I</term></keywords><keywords scheme="MESH" type="chemical" qualifier="pharmacology" xml:lang="en"><term>Starch</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Cross-Over Studies</term><term>Double-Blind Method</term><term>Female</term><term>Humans</term><term>Male</term><term>Pilot Projects</term><term>Young Adult</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Uncooked corn-starch (UCCS) has been the mainstay of therapy for the hepatic glycogen storage diseases (GSD) but is not always effective. A new starch (WMHMS) has demonstrated a more favourable short-term metabolic profile.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">25758258</PMID><DateCreated><Year>2015</Year><Month>04</Month><Day>18</Day></DateCreated><DateCompleted><Year>2016</Year><Month>01</Month><Day>21</Day></DateCompleted><DateRevised><Year>2016</Year><Month>06</Month><Day>24</Day></DateRevised><Article PubModel="Electronic"><Journal><ISSN IssnType="Electronic">1750-1172</ISSN><JournalIssue CitedMedium="Internet"><Volume>10</Volume><PubDate><Year>2015</Year><Month>Feb</Month><Day>15</Day></PubDate></JournalIssue><Title>Orphanet journal of rare diseases</Title><ISOAbbreviation>Orphanet J Rare Dis</ISOAbbreviation></Journal><ArticleTitle>A pilot longitudinal study of the use of waxy maize heat modified starch in the treatment of adults with glycogen storage disease type I: a randomized double-blind cross-over study.</ArticleTitle><Pagination><MedlinePgn>18</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1186/s13023-015-0229-6</ELocationID><Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Uncooked corn-starch (UCCS) has been the mainstay of therapy for the hepatic glycogen storage diseases (GSD) but is not always effective. A new starch (WMHMS) has demonstrated a more favourable short-term metabolic profile.</AbstractText><AbstractText Label="OBJECTIVE" NlmCategory="OBJECTIVE">To determine efficacy and safety of a new uncooked starch (WMHMS) compared to UCCS over 16 weeks treatment with each.</AbstractText><AbstractText Label="METHOD" NlmCategory="METHODS">A double-blind cross-over study of 10 adults (aged 16 - 38 years, six male) with GSD Ia and Ib. After an individualised fast, subjects were randomised to take a 50 g starch-load of either WMHMS or UCCS. Starch-loads terminated when blood glucose was < 3.0 mmol/L or the subject felt subjectively hypoglycaemic. Anonymous biochemical profiles were assessed by 2 investigators and a starch administration schedule recommended. Each starch was delivered in coded sachets and intake was monitored for the following 16 weeks. After a washout period, the protocol was repeated with the alternative product.</AbstractText><AbstractText Label="RESULTS" NlmCategory="RESULTS">4 subjects failed to establish therapy on the cross-over limb. Data from 7 paired starch load showed: longer median fasting duration with WMHMS (7.5 versus 5 hours; p = 0.023), slower decrease in the glucose curve (0.357 versus 0.632 mmol/hr p = 0.028) and less area under insulin curves for the first 4 hours (p = 0.03). Two of six subjects took 50% or less WMHMS compared to UCCS and one took more. Plasma triglycerides, cholesterol and uric acid were unchanged after each study phase.</AbstractText><AbstractText Label="CONCLUSION" NlmCategory="CONCLUSIONS">WMHMS leads to significant reduction in insulin release and reduced starch use in some GSD patients.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Bhattacharya</LastName><ForeName>Kaustuv</ForeName><Initials>K</Initials><AffiliationInfo><Affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Kaustuv.Bhattacharya@health.nsw.gov.au.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Discipline of Paediatrics and Child Health, The Children's Hospital at Westmead Clinical School, University of Sydney, Sydney, Australia. Kaustuv.Bhattacharya@health.nsw.gov.au.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Mundy</LastName><ForeName>Helen</ForeName><Initials>H</Initials><AffiliationInfo><Affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Helen.Mundy@gstt.nhs.uk.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lilburn</LastName><ForeName>Maggie F</ForeName><Initials>MF</Initials><AffiliationInfo><Affiliation>Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, WC1N 3BG, London, UK. Maggie.lilburn@btinternet.com.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Champion</LastName><ForeName>Michael P</ForeName><Initials>MP</Initials><AffiliationInfo><Affiliation>Evelina London Children's Hospital; St Thomas's Hospital, SE1 7EH, London, UK. Michael.Champion@gstt.nhs.uk.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Morley</LastName><ForeName>David W</ForeName><Initials>DW</Initials><AffiliationInfo><Affiliation>Department of Epidemiology & Biostatistics, Imperial College, SW7 2AZ, London, UK. d.w.morley@gmail.com.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Maillot</LastName><ForeName>François</ForeName><Initials>F</Initials><AffiliationInfo><Affiliation>CHRU de Tours, 37044, Tours, Cedex, France. francois.maillot@univ-tours.fr.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D016449">Randomized Controlled Trial</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2015</Year><Month>02</Month><Day>15</Day></ArticleDate></Article><MedlineJournalInfo><Country>England</Country><MedlineTA>Orphanet J Rare Dis</MedlineTA><NlmUniqueID>101266602</NlmUniqueID><ISSNLinking>1750-1172</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D001786">Blood Glucose</NameOfSubstance></Chemical><Chemical><RegistryNumber>9005-25-8</RegistryNumber><NameOfSubstance UI="D013213">Starch</NameOfSubstance></Chemical></ChemicalList><CitationSubset>IM</CitationSubset><CommentsCorrectionsList><CommentsCorrections RefType="Cites"><RefSource>Eur J Pediatr. 1993;152 Suppl 1:S56-9</RefSource><PMID Version="1">8319727</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Clin Endocrinol (Oxf). 1995 Jun;42(6):601-6</RefSource><PMID Version="1">7634500</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>J Inherit Metab Dis. 1995;18(4):462-72</RefSource><PMID Version="1">7494404</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Pediatr Nephrol. 1995 Dec;9(6):705-10</RefSource><PMID 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Version="1">1779622</PMID></CommentsCorrections></CommentsCorrectionsList><MeshHeadingList><MeshHeading><DescriptorName UI="D000293" MajorTopicYN="N">Adolescent</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D001786" MajorTopicYN="N">Blood Glucose</DescriptorName><QualifierName UI="Q000187" MajorTopicYN="N">drug effects</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D018592" MajorTopicYN="N">Cross-Over Studies</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D004311" MajorTopicYN="N">Double-Blind Method</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005953" MajorTopicYN="N">Glycogen Storage Disease Type I</DescriptorName><QualifierName UI="Q000178" MajorTopicYN="Y">diet therapy</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D010865" MajorTopicYN="N">Pilot Projects</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D013213" MajorTopicYN="N">Starch</DescriptorName><QualifierName UI="Q000737" MajorTopicYN="Y">chemistry</QualifierName><QualifierName UI="Q000494" MajorTopicYN="Y">pharmacology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D055815" MajorTopicYN="N">Young Adult</DescriptorName></MeshHeading></MeshHeadingList><OtherID Source="NLM">PMC4340332</OtherID></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2014</Year><Month>11</Month><Day>30</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2015</Year><Month>01</Month><Day>18</Day></PubMedPubDate><PubMedPubDate 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