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
Links to Exploration step
pubmed:25758258Le document en format XML
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<author><name sortKey="Mundy, Helen" sort="Mundy, Helen" uniqKey="Mundy H" first="Helen" last="Mundy">Helen Mundy</name>
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<author><name sortKey="Morley, David W" sort="Morley, David W" uniqKey="Morley D" first="David W" last="Morley">David W. Morley</name>
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<author><name sortKey="Maillot, Francois" sort="Maillot, Francois" uniqKey="Maillot F" first="François" last="Maillot">François Maillot</name>
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<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>
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<author><name sortKey="Lilburn, Maggie F" sort="Lilburn, Maggie F" uniqKey="Lilburn M" first="Maggie F" last="Lilburn">Maggie F. Lilburn</name>
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<author><name sortKey="Champion, Michael P" sort="Champion, Michael P" uniqKey="Champion M" first="Michael P" last="Champion">Michael P. Champion</name>
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<author><name sortKey="Morley, David W" sort="Morley, David W" uniqKey="Morley D" first="David W" last="Morley">David W. Morley</name>
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<series><title level="j">Orphanet journal of rare diseases</title>
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<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>
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<term>Adult</term>
<term>Cross-Over Studies</term>
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<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>
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<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>
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