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      Severe Hypothyroidism Caused by Hepatic Hemangioendothelioma in an Infant of a Diabetic Mother

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          Abstract

          Hemangioendotheliomas can express type 3 iodothyronine deiodinase and cause severe hypothyroidism. The risk of congenital malformations such as vertebral and cardiac abnormalities in infants of diabetic mothers is higher than babies of healthy women. Here we report an infant of a diabetic mother with hypothyroidism caused by liver hemangioendothelioma. Consumptive hypothyroidism should be an indicator to search for a vascular tumor in infants. Supranormal doses of L-thyroxine might be required for normalization of thyroid function until the tumor involutes or is resected.

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          Most cited references 7

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          Severe hypothyroidism caused by type 3 iodothyronine deiodinase in infantile hemangiomas.

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            Spontaneous regression of severe acquired infantile hypothyroidism associated with multiple liver hemangiomas.

            A 9-week-old infant presented with severe postnatal hypothyroidism. His hypothyroidism corrected only after his L-thyroxine dose was progressively increased to 28 micro g/kg/d. At 6 months of age, multiple clinically asymptomatic hepatic hemangiomas were detected and support a diagnosis of consumptive hypothyroidism as a result of increased type 3 iodothyronine deiodinase activity in the hemangiomas. Coincident with the involution of the hemangiomas, the child's hypothyroidism improved and L-thyroxin replacement could be stopped at the age of 3 years. Despite some degree of hypothyroidism for several weeks during infancy, his growth and development have been normal.
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              A 21-year-old woman with consumptive hypothyroidism due to a vascular tumor expressing type 3 iodothyronine deiodinase.

              We present a 21-yr-old female with a large hepatic vascular tumor and subclinical hypothyroidism. A high level of the thyroid hormone inactivating enzyme type 3 iodothyronine deiodinase (D3) was detected in her tumor, and the TSH of 26.2 mU/liter returned to normal after surgical resection of the mass. This indicates that the vascular tumor caused this adult's hypothyroidism as has now been documented in nine infants with this syndrome. This first example of consumptive hypothyroidism in an adult indicates that the inactivation rate of thyroid hormone by D3 in a vascular tumor can stress the secretory capacity even of the TSH-stimulated normal adult thyroid gland.
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                Author and article information

                Journal
                HRE
                Horm Res Paediatr
                10.1159/issn.1663-2818
                Hormone Research in Paediatrics
                S. Karger AG
                1663-2818
                1663-2826
                2005
                April 2005
                06 April 2005
                : 63
                : 2
                : 86-89
                Affiliations
                Divisions of aEndocrinology, bNeonatology, cOncology, and dCardiology, Department of Pediatrics, Medical Faculty, Ondokuz Mayıs University, Samsun, Turkey
                Article
                83879 Horm Res 2005;63:86–89
                10.1159/000083879
                15711094
                © 2005 S. Karger AG, Basel

                Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                Page count
                Figures: 3, References: 18, Pages: 4
                Categories
                Case Report

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