Literature DB >> 26452737

Fever-triggered Brugada syndrome in an adult patient presenting with hemophagocytic syndrome induced by Chlamydophila pneumoniae.

Miguel Bigotte Vieira1, Nuno Gaibino2, Alexandra Pignatelli3, Anabela Oliveira2.   

Abstract

A previously healthy 29-year-old man was admitted to our hospital, with a 4-day history of fever (>39°C), rigours, diaphoresis, fatigue and retro-orbital headache. On examination, he was febrile (37.8°C) and tachycardic (110 bpm). Laboratory work up revealed bicytopenia (white cell count 1.37×10(9)/L, platelets 60×10(9)/L) and an increase in C reactive protein (9 mg/dL). The ECG showed ST segment elevation in V1, V2 and V3 leads. The patient was admitted and investigation was initiated revealing prolonged fever (>7 days), pancytopenia, hepatosplenomegaly, hyperferritinemia, hypertriglyceridemia, hypofibrinogenemia, elevated soluble CD25 and hemophagocytosis in bone marrow. Therefore, the patient presented 7 of the 8 diagnostic criteria of hemophagocytic syndrome. Laboratorial investigation for infectious causes was negative, except for IgA and IgG Chlamydophila pneumoniae. ECG re-evaluation on the day of discharge showed no ST segment elevation and no other abnormalities. Genetic testing for known mutations associated with hemophagocytic syndrome and Brugada syndrome did not show any mutations in these genes. 2015 BMJ Publishing Group Ltd.

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Year:  2015        PMID: 26452737      PMCID: PMC4600790          DOI: 10.1136/bcr-2014-208863

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  15 in total

1.  HLH-2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis.

Authors:  Jan-Inge Henter; Annacarin Horne; Maurizio Aricó; R Maarten Egeler; Alexandra H Filipovich; Shinsaku Imashuku; Stephan Ladisch; Ken McClain; David Webb; Jacek Winiarski; Gritta Janka
Journal:  Pediatr Blood Cancer       Date:  2007-02       Impact factor: 3.167

2.  Unmasking of brugada syndrome by lithium.

Authors:  Dawood Darbar; Tao Yang; Keith Churchwell; Arthur A M Wilde; Dan M Roden
Journal:  Circulation       Date:  2005-09-06       Impact factor: 29.690

Review 3.  How I treat hemophagocytic lymphohistiocytosis.

Authors:  Michael B Jordan; Carl E Allen; Sheila Weitzman; Alexandra H Filipovich; Kenneth L McClain
Journal:  Blood       Date:  2011-08-09       Impact factor: 22.113

4.  Chlamydia pneumoniae infection-related hemophagocytic lymphohistiocytosis and acute encephalitis and poliomyelitis-like flaccid paralysis.

Authors:  Kanae Yagi; Gen Kano; Mayumi Shibata; Izumi Sakamoto; Hirofumi Matsui; Shinsaku Imashuku
Journal:  Pediatr Blood Cancer       Date:  2010-11-05       Impact factor: 3.167

5.  Identification of novel MUNC13-4 mutations in familial haemophagocytic lymphohistiocytosis and functional analysis of MUNC13-4-deficient cytotoxic T lymphocytes.

Authors:  K Yamamoto; E Ishii; M Sako; S Ohga; K Furuno; N Suzuki; I Ueda; M Imayoshi; S Yamamoto; A Morimoto; H Takada; T Hara; S Imashuku; T Sasazuki; M Yasukawa
Journal:  J Med Genet       Date:  2004-10       Impact factor: 6.318

Review 6.  Chlamydophila pneumoniae.

Authors:  F Blasi; P Tarsia; S Aliberti
Journal:  Clin Microbiol Infect       Date:  2009-01       Impact factor: 8.067

7.  Q fever - a review.

Authors:  T J Marrie
Journal:  Can Vet J       Date:  1990-08       Impact factor: 1.008

8.  Determinants of sudden cardiac death in individuals with the electrocardiographic pattern of Brugada syndrome and no previous cardiac arrest.

Authors:  Josep Brugada; Ramon Brugada; Pedro Brugada
Journal:  Circulation       Date:  2003-11-17       Impact factor: 29.690

Review 9.  Hemophagocytic syndrome in children and adults.

Authors:  Iwona Malinowska; Maciej Machaczka; Katarzyna Popko; Alicja Siwicka; Małgorzata Salamonowicz; Barbara Nasiłowska-Adamska
Journal:  Arch Immunol Ther Exp (Warsz)       Date:  2014-02-09       Impact factor: 4.291

10.  Imaging characteristics of hemophagocytic lymphohistiocytosis.

Authors:  Nancy E Fitzgerald; Kenneth L MacClain
Journal:  Pediatr Radiol       Date:  2003-03-21
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  1 in total

Review 1.  Brugada pattern: a comprehensive review on the demographic and clinical spectrum.

Authors:  Hassan Abbas; Sohaib Roomi; Waqas Ullah; Asrar Ahmad; Ganesh Gajanan
Journal:  BMJ Case Rep       Date:  2019-07-11
  1 in total

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