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      Hypothalamic lipoma and growth hormone deficiency

      case-report

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          Abstract

          Background

          Intracranial lipomas are rare, congenital lesions, most often located at the midline. Most hypothalamic lipomas are asymptomatic, but some cases have been associated with precocious puberty, hypothermia, headache and/or obesity.

          Case presentation

          A 7-year-old boy was referred for short stature and proved to be partially growth-hormone deficient. Magnetic resonance imaging (MRI) revealed a lipoma in the paramedian hypothalamus. Growth hormone treatment resulted in swift and uncomplicated catch-up growth.

          Conclusions

          The present case appears to be the first to link hypothalamic lipoma to GH deficiency. The neuro-endocrine pathophysiology underpinning this link remains to be explored.

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          Most cited references14

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          Reference intervals for insulin-like growth factor-1 (igf-i) from birth to senescence: results from a multicenter study using a new automated chemiluminescence IGF-I immunoassay conforming to recent international recommendations.

          Measurement of IGF-I is a cornerstone in diagnosis and monitoring of GH-related diseases, but considerable discrepancies exist between analytical methods. A recent consensus conference defined criteria for validation of IGF-I assays and for establishment of normative data. Our objectives were development and validation of a novel automated IGF-I immunoassay (iSYS; Immunodiagnostic Systems) according to international guidelines and establishment of method-specific age- and sex-adjusted reference intervals and analysis of their robustness. We conducted a multicenter study with samples from 12 cohorts from the United States, Canada, and Europe including 15 014 subjects (6697 males and 8317 females, 0-94 years of age). We measured concentrations of IGF-I as determined by the IDS iSYS IGF-I assay. A new IGF-I assay calibrated against the recommended standard (02/254) and insensitive to the 6 high-affinity IGF binding proteins was developed and rigorously validated. Age- and sex-adjusted reference intervals derived from a uniquely large cohort reflect the age-related pattern of IGF-I secretion: a decline immediately after birth followed by an increase until a pubertal peak (at 15 years of age). Later in life, values decrease continuously. The impact of gender is small, although across the lifespan, women have lower mean IGF-I concentrations. Geographical region, sampling setting (community or hospital based), and rigor of exclusion criteria in our large cohort did not affect the reference intervals. Using large cohorts of well-characterized subjects from different centers allowed construction of robust reference ranges for a new automated IGF-I assay. The strict adherence to recent consensus criteria for IGF-I assays might facilitate clinical application of the results.
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            Intracranial lipomas: importance of localization.

            Intracranial lipomas are rare congenital malformations. They are usually pericallosal asymptomatic midline lesions. Other brain malformations are often seen in association with intracranial lipomas. We describe the findings of imaging studies, including computed tomography (CT), magnetic resonance (MR) imaging, and MR angiography, along with a brief review of the literature. The frequency and the spectrum of the associated brain malformations are also discussed. We retrospectively reviewed CT and MR findings of 24 patients (14 female, 10 male, mean age 38.6 years) diagnosed with intracranial lipoma between December 2000 and June 2004 in two different radiology departments. Seventeen of the patients were diagnosed using cranial MR and seven with cranial CT. The CT density of all lesions was measured. Imaging characteristics of lipomas, morphological findings and associated malformations were described. The intracranial locations of the lipomas were left-sided quadrigeminal cistern (n=3), right-sided quadrigeminal cistern (n=4), interpeduncular cistern (n=1), sylvian fissure (n=3), interhemispheric fissure (n=3), choroid plexus (n=2), intercerebellar fissure (n=3), corpus fornicis (n=1) and the periphery of the corpus callosum (n=4). Eighteen of the intracranial lipomas were tubulonodular; six were curvilinear. Associated anomalies were observed in six patients. All of the patients with sylvian fissure lipoma had seizures. The two preferential sites of intracranial lipomas were pericallosal and dorsal mesencephalic. Most intracranial lipomas are found incidentally during neuroradiological investigations. CT and MR examination usually lead to the diagnosis, because of the very low attenuation values of lipomas on CT and the short T1 and T2 on MR. Midline anomalies and other malformations such as aneurysms are frequently associated with intracranial lipomas. Careful radiologic evaluation is therefore necessary to evaluate associated pathologies. Sylvian fissure lipomas should be considered in the differential diagnosis of patients with epilepsy.
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              60 YEARS OF NEUROENDOCRINOLOGY: The hypothalamo-GH axis: the past 60 years.

              At the time of the publication of Geoffrey Harris's monograph on 'Neural control of the pituitary gland' 60 years ago, the pituitary was recognised to produce a growth factor, and extracts administered to children with hypopituitarism could accelerate growth. Since then our understanding of the neuroendocrinology of the GH axis has included identification of the key central components of the GH axis: GH-releasing hormone and somatostatin (SST) in the 1970s and 1980s and ghrelin in the 1990s. Characterisation of the physiological control of the axis was significantly advanced by frequent blood sampling studies in the 1980s and 1990s; the pulsatile pattern of GH secretion and the factors that influenced the frequency and amplitude of the pulses have been defined. Over the same time, spontaneously occurring and targeted mutations in the GH axis in rodents combined with the recognition of genetic causes of familial hypopituitarism demonstrated the key factors controlling pituitary development. As the understanding of the control of GH secretion advanced, developments of treatments for GH axis disorders have evolved. Administration of pituitary-derived human GH was followed by the introduction of recombinant human GH in the 1980s, and, more recently, by long-acting GH preparations. For GH excess disorders, dopamine agonists were used first followed by SST analogues, and in 2005 the GH receptor blocker pegvisomant was introduced. This review will cover the evolution of these discoveries and build a picture of our current understanding of the hypothalamo-GH axis.
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                Author and article information

                Contributors
                anne.rochtus@uzleuven.be
                Journal
                Int J Pediatr Endocrinol
                Int J Pediatr Endocrinol
                International Journal of Pediatric Endocrinology
                BioMed Central (London )
                1687-9848
                1687-9856
                5 February 2020
                5 February 2020
                2020
                : 2020
                : 4
                Affiliations
                [1 ]ISNI 0000 0004 0626 3338, GRID grid.410569.f, Pediatric Neurology, , University Hospitals Leuven, ; 3000 Leuven, Belgium
                [2 ]ISNI 0000 0004 0626 3338, GRID grid.410569.f, Pediatric Endocrinology, , University Hospitals Leuven, ; Herestraat 49, 3000 Leuven, Belgium
                Author information
                http://orcid.org/0000-0002-3266-9126
                Article
                74
                10.1186/s13633-020-0074-9
                7001293
                1ff7bc35-cf88-4015-a3f1-5d621b81f70e
                © The Author(s). 2020

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 12 November 2019
                : 27 January 2020
                Funding
                Funded by: FundRef http://dx.doi.org/10.13039/501100004497, Onderzoeksraad, KU Leuven;
                Award ID: PDM/17/195
                Award Recipient :
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2020

                Pediatrics
                hypothalamic lipoma,growth hormone deficiency,intracranial lipoma,child
                Pediatrics
                hypothalamic lipoma, growth hormone deficiency, intracranial lipoma, child

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