Cardiac failure in an infant with Chediak-Higashi syndrome: a hypothesis of the effect of diadenosine polyphosphates.
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Cardiac failure in an infant with Chediak-Higashi syndrome: a hypothesis of the effect of diadenosine polyphosphates. / Ganschow, Rainer; Grabhorn, Enke; Lemke, Joachim; Lepler, Rudolf.
in: PEDIAT ALLERG IMM-UK, Jahrgang 13, Nr. 4, 4, 2002, S. 307-310.Publikationen: SCORING: Beitrag in Fachzeitschrift/Zeitung › SCORING: Zeitschriftenaufsatz › Forschung › Begutachtung
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TY - JOUR
T1 - Cardiac failure in an infant with Chediak-Higashi syndrome: a hypothesis of the effect of diadenosine polyphosphates.
AU - Ganschow, Rainer
AU - Grabhorn, Enke
AU - Lemke, Joachim
AU - Lepler, Rudolf
PY - 2002
Y1 - 2002
N2 - A 14-week-old boy with undiagnosed Chediak-Higashi syndrome developed fever with a high temperature and acute cardiac failure after having received a scheduled vaccination. We hypothesize that decreased concentrations and receptor binding of serum and tissue diadenosine polyphosphates, such as AP4A, AP5A, or AP6A, which are stored in various tissues and serve as extra-cellular signaling molecules or are secreted by cells in response to physiologically stressful stimuli, lead to the observed severe tachyarrhythmia. Diadenosine polyphosphates normally have a negative chronotropic and inotropic effect. This is the first report of severe cardiac failure in a child with Chediak-Higashi syndrome and we suggest that cardiac arrhythmias should be considered in such children in the event of high fever. Our hypothesis requires further investigation in other patients.
AB - A 14-week-old boy with undiagnosed Chediak-Higashi syndrome developed fever with a high temperature and acute cardiac failure after having received a scheduled vaccination. We hypothesize that decreased concentrations and receptor binding of serum and tissue diadenosine polyphosphates, such as AP4A, AP5A, or AP6A, which are stored in various tissues and serve as extra-cellular signaling molecules or are secreted by cells in response to physiologically stressful stimuli, lead to the observed severe tachyarrhythmia. Diadenosine polyphosphates normally have a negative chronotropic and inotropic effect. This is the first report of severe cardiac failure in a child with Chediak-Higashi syndrome and we suggest that cardiac arrhythmias should be considered in such children in the event of high fever. Our hypothesis requires further investigation in other patients.
M3 - SCORING: Zeitschriftenaufsatz
VL - 13
SP - 307
EP - 310
JO - PEDIAT ALLERG IMM-UK
JF - PEDIAT ALLERG IMM-UK
SN - 0905-6157
IS - 4
M1 - 4
ER -
