QJM Advance Access originally published online on December 26, 2007
QJM 2008 101(1):31-40; doi:10.1093/qjmed/hcm118
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Cardiovascular risk factors in adult patients with multisystem Langerhans-cell histiocytosis: evidence of glucose metabolism abnormalities
From the 1Department of Pathophysiology, Division of Endocrinology, Laiko University Hospital, 2Department of Endocrinology & Diabetes, 251 Hellenic Air Force, Athens General Hospital, 3Department of Clinical Therapeutic, Vascular laboratory, "Alexandra" University Hospital, 4Department of Endocrinology, "G.Genimatas" General Hospital, Athens, Greece, 5Division of Endocrinology, Diabetes and Metabolism, Tufts-New England Medical Center, Boston, MA, USA and 6Department of Endocrinology, St Bartholomew's Hospital, London, UK
Address correspondence to K.I. Alexandraki, Department of Endocrinology, Laiko General Hospital, School of Medicine, National & Kapodistrian University of Athens Themistokleous 19 str, 15122 Amaroussion, Athens, Greece. email: krisalex{at}med.uoa.gr
Received 27 August 2007 and in revised form 1 October 2007
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Abstract |
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Background: Langerhans-cell histiocytosis (LCH) is a rare disease with features of chronic inflammation and it may also induce hypopituitarism, conditions associated with an increased risk of cardiovascular diseases.
Aim: Cardiovascular and metabolic risk profile investigation in multisystem LCH patients with and without anterior pituitary deficiency.
Design: Prospective, observational study.
Methods: Fourteen adult patients with LCH, 7 with and 7 without anterior pituitary deficiency, and 42 controls matched for age, body mass index (BMI) and smoking. Cardiovascular risk factors were estimated in all subjects: glucose and lipid profile, mathematical indices of insulin resistance (IR), blood pressure, structural arterial and functional endothelial properties (intima-media thickness, brachial artery flow-mediated dilatation). Cardiovascular risk factors were estimated in the three groups studied; the effect of disease activity and/or treatment was also determined in patients with LCH.
Results: Ten patients had diabetes insipidus, and 7 anterior pituitary hormone deficiencies: 8 patients had active disease and 11 had received systemic treatment. No difference was observed between the study groups in vascular parameters, in lipid profile or in blood pressure. However, the insulin resistance index GIR was decreased in patients with LCH without anterior pituitary deficiency compared to controls (P = 0.033). Three patients had impaired glucose tolerance and one diabetes mellitus type 2. These patients were older and had active disease; there was no association with hypopituitarism and/or previous treatment.
Conclusions: Adults patients with LCH have abnormalities of glucose metabolism that tend to occur in patients with active disease, and may be a consequence of the pro-inflammatory state.