8
views
0
recommends
+1 Recommend
0 collections
    0
    shares
      • Record: found
      • Abstract: found
      • Article: found
      Is Open Access

      Resolution of Consumptive Hypothyroidism Secondary to Infantile Hepatic Hemangiomatosis with a Combination of Propranolol and Levothyroxine

      case-report

      Read this article at

      Bookmark
          There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

          Abstract

          Infantile hepatic hemangiomas (IHH), particularly of the diffuse subtype can, in severe cases, be associated with hepatic and cardiac failure, compartment syndrome and consumptive hypothyroidism. Early recognition and treatment of these pathologies is paramount in order to minimise the risk of long-term sequelae.

          We report an interesting case of a female infant who presented with systemic compromise, in the absence of large or obvious cutaneous infantile hemangiomas. Imaging identified innumerable hepatic hemangiomas, consistent with diffuse infantile hepatic hemangiomatosis. Subsequent to this, thyroid function tests confirmed an associated but comparatively rare form of hypothyroidism, known as consumptive hypothyroidism. Following joint consultation with dermatology and endocrinology she was promptly treated with oral propranolol and levothyroxine, with subsequent improvement in her clinical parameters.

          This case reiterates the importance of aggressive investigation and management of consumptive hypothyroidism in any infant diagnosed with IHH, particularly when there is systemic compromise. We advocate propranolol as a single first line treatment for IHH, supported by thyroid replacement when appropriate.

          Related collections

          Most cited references20

          • Record: found
          • Abstract: found
          • Article: not found

          Infantile hemangiomas: how common are they? A systematic review of the medical literature.

          No published prospective studies have been published for several decades examining the incidence of hemangiomas. Older studies were performed before the delineation of "hemangiomas" from other vascular birthmarks was well-established. The objective of our study is to critically re-examine the literature reporting the incidence of infantile hemangiomas to determine if the true incidence is actually known. We performed both an electronic database search and hand search of the medical literature on the natural history of hemangiomas in full-term newborns and infants. A total of seven articles were found comprising two study populations: newborns 500 patients including both hospital-based and primary care settings. Study designs ranged from retrospective chart reviews to cross-sectional cohort studies. Descriptive nomenclature was not uniform between studies, and all had methodologic limitations including problems of definition and study design. Studies estimating the true incidence of infantile hemangiomas are all many decades old and have significant methodologic issues limiting their ability to determine hemangioma incidence. Future studies in primary care settings using the currently accepted classification schema of vascular birthmarks may more accurately define the incidence and potential impact of this common vascular tumor of infancy.
            Bookmark
            • Record: found
            • Abstract: not found
            • Article: not found

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

              Bookmark
              • Record: found
              • Abstract: found
              • Article: not found

              Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry.

              Hepatic hemangiomas, though histologically benign, may be associated with significant morbidity and mortality in afflicted infants. The literature presents much confusion regarding the natural history and treatment options for hepatic hemangiomas. Clinical manifestations range from asymptomatic self-limiting lesions to congestive heart failure associated with high-volume vascular shunting to fulminant hepatic failure with hypothyroidism, abdominal compartment syndrome, and death. There has been little rationale to choose among observation, corticosteroid, other pharmacologic agents, arterial embolization, hepatic artery ligation, resection, or liver transplantation for any given patient. We analyzed several recent retrospective radiologic analyses and pathologic studies to determine whether hepatic hemangiomas could be categorized, allowing prediction of their natural history and rational choice of therapies based upon their clinical presentation and radiographic appearance. We propose that hepatic hemangiomas do not represent a single entity but, rather, 3 principle categories of lesions: focal, multifocal, and diffuse. Because these 2 categories represent different anatomical and physiologic variants, so, too, may they respond differently to previously anecdotally applied treatment regimens. With input from international multidisciplinary authorities on hemangiomas, we developed and proposed a clinical practice algorithm for the evaluation and management of hepatic hemangiomas. Toward that end, we propose a plan to institute a web-based international hepatic hemangioma registry. Participants in the registry could obtain no-cost centralized review of imaging studies (and histology if available) and guidance regarding the management algorithm from an established multidisciplinary team. In exchange, the registry will facilitate the acquisition of systematic clinical and imaging information. Longitudinal observation of response to more directed treatment protocols may contribute greatly to the understanding of these potentially fatal tumors.
                Bookmark

                Author and article information

                Journal
                J Clin Res Pediatr Endocrinol
                J Clin Res Pediatr Endocrinol
                JCRPE
                Journal of Clinical Research in Pediatric Endocrinology
                Galenos Publishing
                1308-5727
                1308-5735
                September 2018
                31 July 2018
                : 10
                : 3
                : 294-298
                Affiliations
                [1 ]Royal Victoria Hospital, Clinic of Dermatology, Belfast, Northern Ireland, United Kingdom
                [2 ]Royal Belfast Hospital for Sick Children, Clinic of Paediatric Endocrinology, Growth and Diabetes, Belfast, Northern Ireland, United Kingdom
                Author notes
                * Address for Correspondence: Royal Victoria Hospital, Clinic of Dermatology, Belfast, Northern Ireland, United Kingdom Phone: +028 90240503 E-mail: victoriacampbell@ 123456doctors.org.uk
                Author information
                https://orcid.org/0000-0002-7626-3574
                https://orcid.org/0000-0002-6449-6610
                https://orcid.org/0000-0002-9442-4618
                https://orcid.org/0000-0001-8403-4627
                Article
                16731
                10.4274/jcrpe.4865
                6083462
                29537380
                63c4b697-df4e-4fe1-9193-a5e60729bc26
                © Copyright 2018, Journal of Clinical Research in Pediatric Endocrinology, Published by Galenos Publishing.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 21 August 2017
                : 28 December 2017
                Categories
                Case Report

                Pediatrics
                hemangioma,consumptive hypothyroidism,type 3 iodothyronine deiodinase,propranolol
                Pediatrics
                hemangioma, consumptive hypothyroidism, type 3 iodothyronine deiodinase, propranolol

                Comments

                Comment on this article