Fatal infectious mononucleosis in a family

Two male siblings, one aged five and a half months (SB), and the other aged six months (VB), with fatal infectious mononucleosis phenotype of the X-linked lymphoproliferative syndrome, which resulted in the death of both infants, are presented. Both patients had been healthy, one until the ag...

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Main Authors: O Sanal, G Kale, F Ersoy, S Göğüş, Y Coşkun, M Cağlar, A I Berkel
Format: Article
Language:English
Published: Hacettepe University Institute of Child Health 1990-04-01
Series:The Turkish Journal of Pediatrics
Online Access:https://turkjpediatr.org/article/view/3799
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author O Sanal
G Kale
F Ersoy
S Göğüş
Y Coşkun
M Cağlar
A I Berkel
author_facet O Sanal
G Kale
F Ersoy
S Göğüş
Y Coşkun
M Cağlar
A I Berkel
author_sort O Sanal
collection DOAJ
description Two male siblings, one aged five and a half months (SB), and the other aged six months (VB), with fatal infectious mononucleosis phenotype of the X-linked lymphoproliferative syndrome, which resulted in the death of both infants, are presented. Both patients had been healthy, one until the age of five and a half months, and the other until the age of six months. Then, they developed a maculopapular rash, hepatosplenomegaly and lymphadenopathy. In one sibling, the serum IgG level was low, the IgM and IgA levels were high, and the proportion of E-rosette forming cells (E-RFC) and in vitro proliferative response to PHA were normal. In the other sibling, however, the serum IgG level was normal, the IgM and IgA levels were high and the stimulation index for proliferative response to PHA was reduced due to increased spontaneous blastogenesis. Anti-EBV antibodies were negative in both siblings, except for the IgM anti-VCA in V.B. A lymph node specimen could be studied in one infant and was found to be positive for the EBV genome. Postmortem histopathological findings included the absence of cortico-medullary differentiation and identifiable Hassal's corpuscles in the thymus and depletion of T-dependent regions of lymph nodes and spleen in V.B. Atypical mononuclear cell infiltration was detected in the portal areas of the postmortem liver biopsy in S.B.
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publishDate 1990-04-01
publisher Hacettepe University Institute of Child Health
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spelling doaj-art-35c2fe481bfb4d019930651786fdfd9f2025-08-20T02:59:41ZengHacettepe University Institute of Child HealthThe Turkish Journal of Pediatrics0041-43012791-64211990-04-01322Fatal infectious mononucleosis in a familyO Sanal0G KaleF ErsoyS GöğüşY CoşkunM CağlarA I BerkelDepartment of Pediatrics, Hacettepe University Faculty of Medicine, Ankara. Two male siblings, one aged five and a half months (SB), and the other aged six months (VB), with fatal infectious mononucleosis phenotype of the X-linked lymphoproliferative syndrome, which resulted in the death of both infants, are presented. Both patients had been healthy, one until the age of five and a half months, and the other until the age of six months. Then, they developed a maculopapular rash, hepatosplenomegaly and lymphadenopathy. In one sibling, the serum IgG level was low, the IgM and IgA levels were high, and the proportion of E-rosette forming cells (E-RFC) and in vitro proliferative response to PHA were normal. In the other sibling, however, the serum IgG level was normal, the IgM and IgA levels were high and the stimulation index for proliferative response to PHA was reduced due to increased spontaneous blastogenesis. Anti-EBV antibodies were negative in both siblings, except for the IgM anti-VCA in V.B. A lymph node specimen could be studied in one infant and was found to be positive for the EBV genome. Postmortem histopathological findings included the absence of cortico-medullary differentiation and identifiable Hassal's corpuscles in the thymus and depletion of T-dependent regions of lymph nodes and spleen in V.B. Atypical mononuclear cell infiltration was detected in the portal areas of the postmortem liver biopsy in S.B. https://turkjpediatr.org/article/view/3799
spellingShingle O Sanal
G Kale
F Ersoy
S Göğüş
Y Coşkun
M Cağlar
A I Berkel
Fatal infectious mononucleosis in a family
The Turkish Journal of Pediatrics
title Fatal infectious mononucleosis in a family
title_full Fatal infectious mononucleosis in a family
title_fullStr Fatal infectious mononucleosis in a family
title_full_unstemmed Fatal infectious mononucleosis in a family
title_short Fatal infectious mononucleosis in a family
title_sort fatal infectious mononucleosis in a family
url https://turkjpediatr.org/article/view/3799
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AT gkale fatalinfectiousmononucleosisinafamily
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AT sgogus fatalinfectiousmononucleosisinafamily
AT ycoskun fatalinfectiousmononucleosisinafamily
AT mcaglar fatalinfectiousmononucleosisinafamily
AT aiberkel fatalinfectiousmononucleosisinafamily