DOI: 10.1530/EJE-15-1025 Printed in Great Britain
Published by Bioscientifica Ltd.
European Journal of Endocrinology
www.eje-online.org © 2016 European Society of Endocrinology
MANAGEMENT OF ENDOCRINE DISEASE
Klinefelter syndrome, cardiovascular system,
and thromboembolic disease: review of
literature and clinical perspectives
Andrea Salzano
1
, Michele Arcopinto
2
, Alberto M Marra
3
, Emanuele Bobbio
1
,
Daniela Esposito
4
, Giacomo Accardo
4
, Francesco Giallauria
1
, Eduardo Bossone
5
,
Carlo Vigorito
1
, Andrea Lenzi
6
, Daniela Pasquali
4
, Andrea M Isidori
6
and
Antonio Cittadini
1
1
Department of Translational Medical Sciences, University “Federico II”, Naples, Italy,
2
Department of
Cardiac Surgery, IRCCS Policlinico San Donato, Milan, Italy,
3
IRCSS SDN, Via E. Gianturco 113, Naples, Italy,
4
Department of Cardiothoracic and Respiratory Science, Endocrinology Unit, Second University of Naples,
Italy,
5
Department of Cardiology and Cardiac Surgery, University Hospital “Scuola Medica Salernitana”,
Salerno, Italy and
6
Department of Experimental Medicine, Sapienza University of Rome, Rome, Italy
Abstract
Klinefelter syndrome (KS) is the most frequently occurring sex chromosomal aberration in males, with an incidence
of about 1 in 500 – 700 newborns. Data acquired from large registry-based studies revealed an increase in mortality
rates among KS patients when compared with mortality rates among the general population. Among all causes of
death, metabolic, cardiovascular, and hemostatic complication seem to play a pivotal role. KS is associated, as are other
chromosomal pathologies and genetic diseases, with cardiac congenital anomalies that contribute to the increase in
mortality. The aim of the current study was to systematically review the relationships between KS and the cardiovascular
system and hemostatic balance. In summary , patients with KS display an increased cardiovascular risk profle, characterized
by increased prevalence of metabolic abnormalities including Diabetes mellitus (DM), dyslipidemia, and alterations
in biomarkers of cardiovascular disease. KS does not, however, appear to be associated with arterial hypertension.
Moreover, KS patients are characterized by subclinical abnormalities in left ventricular (LV) systolic and diastolic function
and endothelial function, which, when associated with chronotropic incompetence may led to reduced cardiopulmonary
performance. KS patients appear to be at a higher risk for cardiovascular disease, attributing to an increased risk of
thromboembolic events with a high prevalence of recurrent venous ulcers, venous insuffciency, recurrent venous
and arterial thromboembolism with higher risk of deep venous thrombosis or pulmonary embolism. It appears that
cardiovascular involvement in KS is mainly due to chromosomal abnormalities rather than solely on low serum testosterone
levels. On the basis of evidence acquisition and authors’ own experience, a fowchart
addressing the management of cardiovascular function and prognosis of KS patients has
been developed for clinical use.
175:1 R27–R40 Andrea Salzano and others KS cardiovascular system, and
thromboembolic disease
Correspondence
should be addressed
to Antonio Cittadini
Email
antonio.cittadini@unina.it
European Journal of
Endocrinology
(2016) 175, R27–R40
Review
Introduction
Klinefelter syndrome (KS) is the most common abnormality
of sex chromosomes (47, XXY or a mosaic karyotype) and
is characterized by hypergonadotropic hypogonadism (1).
Data indicate the incidence of KS to be as high as 1/660 of
newborns (2, 3). Despite its frst mention being 70 years
ago (1), little data are available with regard to the morbidity
and mortality of KS. Data from recent large registry-based
studies (4, 5, 6, 7, 8) indicated an increase in mortality in
KS patients when compared with the general population.
Interestingly, mortality was specifcally increased by