Document Detail


Sweet syndrome, cutis laxa, and fatal cardiac manifestations in a 2-year-old girl.
MedLine Citation:
PMID:  21720474     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A 2-year-old black girl was diagnosed with Sweet syndrome and cutis laxa, and she was given corticosteroid therapy. At that presentation, a cardiac evaluation revealed nothing unusual. Nine days later, she emergently presented with respiratory distress, and circulatory collapse rapidly developed. A 2-dimensional Doppler echocardiogram showed a dilated and poorly contractile left ventricle, severe aortic regurgitation, and a large aneurysm of the sinus of Valsalva. Despite resuscitative efforts, the child died. Inspection on autopsy revealed a markedly enlarged heart and 2 large aneurysms of the sinus of Valsalva. Histologic analysis disclosed acute necrosis in the cardiac apex and interventricular septum, and focal chronic inflammatory and granulation tissue in the aortic wall.Because cardiac lesions may remain clinically silent, we recommend that children with Sweet syndrome and cutis laxa undergo complete cardiac evaluation, including 2-dimensional Doppler echocardiography, by a pediatric cardiologist. Subsequent monitoring is also appropriate. Early recognition and aggressive therapeutic treatment could prevent fatal cardiac complications.
Authors:
Malini Guhamajumdar; Brojendra Agarwala
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Texas Heart Institute journal / from the Texas Heart Institute of St. Luke's Episcopal Hospital, Texas Children's Hospital     Volume:  38     ISSN:  1526-6702     ISO Abbreviation:  Tex Heart Inst J     Publication Date:  2011  
Date Detail:
Created Date:  2011-07-01     Completed Date:  2011-10-31     Revised Date:  2012-01-23    
Medline Journal Info:
Nlm Unique ID:  8214622     Medline TA:  Tex Heart Inst J     Country:  United States    
Other Details:
Languages:  eng     Pagination:  285-7     Citation Subset:  IM    
Affiliation:
Section of Pediatric Cardiology, University of Chicago Comer Children's Hospital, Chicago, Illinois 60637-1470, USA.
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MeSH Terms
Descriptor/Qualifier:
Adrenal Cortex Hormones / therapeutic use
Aortic Aneurysm
Aortic Valve Insufficiency / etiology
Autopsy
Child, Preschool
Cutis Laxa / complications*,  diagnosis,  drug therapy
Echocardiography, Doppler
Fatal Outcome
Female
Heart Diseases / etiology*,  pathology,  ultrasonography
Humans
Sinus of Valsalva
Sweet Syndrome / complications*,  diagnosis,  drug therapy
Ventricular Dysfunction, Left / etiology
Chemical
Reg. No./Substance:
0/Adrenal Cortex Hormones
Comments/Corrections
Comment In:
Tex Heart Inst J. 2011;38(5):608-9   [PMID:  22163152 ]

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