Secondary haemophagocytic lymphohistiocytosis triggered by postnatally acquired cytomegalovirus infection in a late preterm infant.
Identifieur interne : 000015 ( PubMed/Corpus ); précédent : 000014; suivant : 000016Secondary haemophagocytic lymphohistiocytosis triggered by postnatally acquired cytomegalovirus infection in a late preterm infant.
Auteurs : Christine Silwedel ; Eric Frieauff ; Wolfgang Thomas ; Johannes G. Liese ; Christian P. SpeerSource :
- Infection [ 1439-0973 ] ; 2017.
Abstract
Haemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory condition with impairment of cytotoxic T-cells and natural killer cells. Causes in infants are mostly hereditary immune defects as well as various infectious triggering factors, amongst these cytomegalovirus (CMV). Vertical CMV transmission may occur in utero, during birth, and by breast feeding. Usually, a CMV infection transmitted via breast milk is symptomatic only in very immature preterm infants. We report on a late preterm infant born after 35 + 5 weeks of gestation with a birth weight of 1840 g, being admitted to our intensive care unit at the age of 9 weeks with acute enteritis and severe dehydration. After a prolonged recovery, the infant developed a sepsis-like condition with hyperpyrexia, hepatosplenomegaly, and pancytopenia. Combination with high ferritin levels (2809 μg/l), hypertriglyceridaemia (481 mg/dl), elevated soluble IL-2 receptor (sCD25, 9120 U/ml), and reduced perforin expression allowed diagnosis of HLH, caused by an acute CMV infection. Since connatal CMV infection had been ruled out earlier, we report the rare case of secondary HLH triggered by a postnatally acquired symptomatic CMV infection in an immunocompetent infant, most likely transmitted via breast milk. The infant was successfully treated with ganciclovir without need for immunosuppressive therapy.
DOI: 10.1007/s15010-016-0970-3
PubMed: 28093696
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Silwedel_C@ukw.de.</nlm:affiliation></affiliation></author><author><name sortKey="Frieauff, Eric" sort="Frieauff, Eric" uniqKey="Frieauff E" first="Eric" last="Frieauff">Eric Frieauff</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Thomas, Wolfgang" sort="Thomas, Wolfgang" uniqKey="Thomas W" first="Wolfgang" last="Thomas">Wolfgang Thomas</name><affiliation><nlm:affiliation>Department of Paediatrics, Mutterhaus der Borromaeerinnen, Trier, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Liese, Johannes G" sort="Liese, Johannes G" uniqKey="Liese J" first="Johannes G" last="Liese">Johannes G. Liese</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Speer, Christian P" sort="Speer, Christian P" uniqKey="Speer C" first="Christian P" last="Speer">Christian P. Speer</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2017">2017</date><idno type="RBID">pubmed:28093696</idno><idno type="pmid">28093696</idno><idno type="doi">10.1007/s15010-016-0970-3</idno><idno type="wicri:Area/PubMed/Corpus">000015</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">000015</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Secondary haemophagocytic lymphohistiocytosis triggered by postnatally acquired cytomegalovirus infection in a late preterm infant.</title><author><name sortKey="Silwedel, Christine" sort="Silwedel, Christine" uniqKey="Silwedel C" first="Christine" last="Silwedel">Christine Silwedel</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany. Silwedel_C@ukw.de.</nlm:affiliation></affiliation></author><author><name sortKey="Frieauff, Eric" sort="Frieauff, Eric" uniqKey="Frieauff E" first="Eric" last="Frieauff">Eric Frieauff</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Thomas, Wolfgang" sort="Thomas, Wolfgang" uniqKey="Thomas W" first="Wolfgang" last="Thomas">Wolfgang Thomas</name><affiliation><nlm:affiliation>Department of Paediatrics, Mutterhaus der Borromaeerinnen, Trier, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Liese, Johannes G" sort="Liese, Johannes G" uniqKey="Liese J" first="Johannes G" last="Liese">Johannes G. Liese</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Speer, Christian P" sort="Speer, Christian P" uniqKey="Speer C" first="Christian P" last="Speer">Christian P. Speer</name><affiliation><nlm:affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</nlm:affiliation></affiliation></author></analytic><series><title level="j">Infection</title><idno type="eISSN">1439-0973</idno><imprint><date when="2017" type="published">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Haemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory condition with impairment of cytotoxic T-cells and natural killer cells. Causes in infants are mostly hereditary immune defects as well as various infectious triggering factors, amongst these cytomegalovirus (CMV). Vertical CMV transmission may occur in utero, during birth, and by breast feeding. Usually, a CMV infection transmitted via breast milk is symptomatic only in very immature preterm infants. We report on a late preterm infant born after 35 + 5 weeks of gestation with a birth weight of 1840 g, being admitted to our intensive care unit at the age of 9 weeks with acute enteritis and severe dehydration. After a prolonged recovery, the infant developed a sepsis-like condition with hyperpyrexia, hepatosplenomegaly, and pancytopenia. Combination with high ferritin levels (2809 μg/l), hypertriglyceridaemia (481 mg/dl), elevated soluble IL-2 receptor (sCD25, 9120 U/ml), and reduced perforin expression allowed diagnosis of HLH, caused by an acute CMV infection. Since connatal CMV infection had been ruled out earlier, we report the rare case of secondary HLH triggered by a postnatally acquired symptomatic CMV infection in an immunocompetent infant, most likely transmitted via breast milk. The infant was successfully treated with ganciclovir without need for immunosuppressive therapy.</div></front></TEI><pubmed><MedlineCitation Status="Publisher" Owner="NLM"><PMID Version="1">28093696</PMID><DateCreated><Year>2017</Year><Month>01</Month><Day>17</Day></DateCreated><DateRevised><Year>2017</Year><Month>01</Month><Day>17</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1439-0973</ISSN><JournalIssue CitedMedium="Internet"><PubDate><Year>2017</Year><Month>Jan</Month><Day>16</Day></PubDate></JournalIssue><Title>Infection</Title><ISOAbbreviation>Infection</ISOAbbreviation></Journal><ArticleTitle>Secondary haemophagocytic lymphohistiocytosis triggered by postnatally acquired cytomegalovirus infection in a late preterm infant.</ArticleTitle><ELocationID EIdType="doi" ValidYN="Y">10.1007/s15010-016-0970-3</ELocationID><Abstract><AbstractText NlmCategory="UNASSIGNED">Haemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory condition with impairment of cytotoxic T-cells and natural killer cells. Causes in infants are mostly hereditary immune defects as well as various infectious triggering factors, amongst these cytomegalovirus (CMV). Vertical CMV transmission may occur in utero, during birth, and by breast feeding. Usually, a CMV infection transmitted via breast milk is symptomatic only in very immature preterm infants. We report on a late preterm infant born after 35 + 5 weeks of gestation with a birth weight of 1840 g, being admitted to our intensive care unit at the age of 9 weeks with acute enteritis and severe dehydration. After a prolonged recovery, the infant developed a sepsis-like condition with hyperpyrexia, hepatosplenomegaly, and pancytopenia. Combination with high ferritin levels (2809 μg/l), hypertriglyceridaemia (481 mg/dl), elevated soluble IL-2 receptor (sCD25, 9120 U/ml), and reduced perforin expression allowed diagnosis of HLH, caused by an acute CMV infection. Since connatal CMV infection had been ruled out earlier, we report the rare case of secondary HLH triggered by a postnatally acquired symptomatic CMV infection in an immunocompetent infant, most likely transmitted via breast milk. The infant was successfully treated with ganciclovir without need for immunosuppressive therapy.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Silwedel</LastName><ForeName>Christine</ForeName><Initials>C</Initials><AffiliationInfo><Affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany. Silwedel_C@ukw.de.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Frieauff</LastName><ForeName>Eric</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Thomas</LastName><ForeName>Wolfgang</ForeName><Initials>W</Initials><AffiliationInfo><Affiliation>Department of Paediatrics, Mutterhaus der Borromaeerinnen, Trier, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Liese</LastName><ForeName>Johannes G</ForeName><Initials>JG</Initials><AffiliationInfo><Affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Speer</LastName><ForeName>Christian P</ForeName><Initials>CP</Initials><AffiliationInfo><Affiliation>Children's Hospital, University Hospital Wuerzburg, Josef-Schneider-Str. 2, 97080, Würzburg, Germany.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2017</Year><Month>01</Month><Day>16</Day></ArticleDate></Article><MedlineJournalInfo><Country>Germany</Country><MedlineTA>Infection</MedlineTA><NlmUniqueID>0365307</NlmUniqueID><ISSNLinking>0300-8126</ISSNLinking></MedlineJournalInfo><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">Cytomegalovirus</Keyword><Keyword MajorTopicYN="N">Ganciclovir</Keyword><Keyword MajorTopicYN="N">Haemophagocytic lymphohistiocytosis</Keyword><Keyword MajorTopicYN="N">Preterm infant</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2016</Year><Month>07</Month><Day>19</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2016</Year><Month>11</Month><Day>28</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2017</Year><Month>1</Month><Day>18</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2017</Year><Month>1</Month><Day>18</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2017</Year><Month>1</Month><Day>18</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>aheadofprint</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">28093696</ArticleId><ArticleId IdType="doi">10.1007/s15010-016-0970-3</ArticleId><ArticleId IdType="pii">10.1007/s15010-016-0970-3</ArticleId></ArticleIdList></PubmedData></pubmed></record>Pour manipuler ce document sous Unix (Dilib)
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