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      Massive labial lipomatous hypertrophy in familial partial lipodystrophy seen on computed tomographic angiography

      case-report
      Author(s): , BS, , MD * , , MD
      Publication date (Electronic):
      Journal: Radiology Case Reports
      Publisher: Elsevier
      Keywords: Lipomatous hypertrophy, Familial partial lipodystrophy, CTA

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          Abstract

          We present a 28-year-old female with a rare familial partial lipodystrophy. Originally presenting at the age of 14, she began experiencing hypertrophy of the fat in the mons pubis and labia majora regions. By the age of 24 she had disfiguring hypertrophy of these areas with severe fatty overgrowth, similar in nature to that experienced by her father and paternal grandmother. During her workup and planning for suction lipectomy, she underwent computed tomography angiography with the imaging manifestation of severe massive subcutaneous fat hypertrophy; the imaging appearance was only able to be explained after a thorough review of the patient's history and medical literature.

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          NAFLD in lipodystrophy

          Stergios Polyzos, Nikolaos Perakakis, Christos S. Mantzoros (2019)
          Lipodystrophy is a group of clinically heterogeneous, inherited or acquired, disorders characterized by complete or partial absence of subcutaneous adipose tissue that may occur simultaneously with the pathological, ectopic, accumulation of fat in other regions of the body, including the liver. Fatty liver adds significantly to hepatic and extra-hepatic morbidity in patients with lipodystrophy. Lipodystrophy is strongly associated with severe insulin resistance and related comorbidities, such as hyperglycemia, hyperlipidemia and nonalcoholic fatty liver disease (NAFLD), but other hepatic diseases may co-exist in some types of lipodystrophy, including autoimmune hepatitis in acquired lipodystrophies, or viral hepatitis in human immunodeficiency virus (HIV)-associated lipodystrophy. The aim of this review is to summarize evidence linking lipodystrophy with hepatic disease and to provide a special focus on potential therapeutic perspectives of leptin replacement therapy and adiponectin upregulation in lipodystrophy.
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            Lipodystrophies, dyslipidaemias and atherosclerotic cardiovascular disease

            Iram Hussain, Nivedita Patni, Abhimanyu Garg (2018)
            Lipodystrophies are rare, heterogeneous, genetic or acquired, disorders characterised by varying degrees of body fat loss and associated metabolic complications, including insulin resistance, dyslipidaemias, hepatic steatosis and predisposition to atherosclerotic cardiovascular disease (ASCVD). The four main types of lipodystrophy, excluding antiretroviral therapy-induced lipodystrophy in HIV-infected patients, are congenital generalised lipodystrophy (CGL), familial partial lipodystrophy (FPLD), acquired generalised lipodystrophy (AGL) and acquired partial lipodystrophy (APL). This paper reviews the literature related to the prevalence of dyslipidaemias and ASCVD in patients with lipodystrophies. Patients with CGL, AGL and FPLD have increased prevalence of dyslipidaemia but those with APL do not. Patients with CGL as well as AGL present in childhood, and have severe dyslipidaemias (mainly hypertriglyceridaemia) and early onset diabetes mellitus as a consequence of extreme fat loss. However, only a few patients with CGL and AGL have been reported to develop coronary heart disease. In contrast, data from some small cohorts of FPLD patients reveal increased prevalence of ASCVD especially among women. Patients with APL have a relatively low prevalence of hypertriglyceridaemia and diabetes mellitus. Overall, patients with lipodystrophies appear to be at high risk of ASCVD due to increased prevalence of dyslipidaemia and diabetes and efforts should be made to manage these metabolic complications aggressively to prevent ASCVD.
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              Long-term effectiveness and safety of metreleptin in the treatment of patients with partial lipodystrophy.

              Elif A Oral, Phillip Gorden, Elaine Cochran … (2019)
              To evaluate the effects of metreleptin in patients with partial lipodystrophy (PL).
              Article 0 comments Cited 28 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                Neil J. Hansen
                Journal
                Journal ID (nlm-ta): Radiol Case Rep
                Journal ID (iso-abbrev): Radiol Case Rep
                Title: Radiology Case Reports
                Publisher: Elsevier
                ISSN (Electronic): 1930-0433
                Publication date PMC-release: 22 August 2019
                Publication date Collection: October 2019
                Publication date (Electronic): 22 August 2019
                Volume: 14
                Issue: 10
                Pages: 1311-1313
                Affiliations
                [0001]University of Nebraska College of Medicine, Department of Radiology, 981045 Nebraska Medical Center, Omaha, NE 68198-1045
                Author notes
                [* ]Corresponding Author. njhansen@ 123456unmc.edu
                Article
                Publisher ID: S1930-0433(19)30271-7
                DOI: 10.1016/j.radcr.2019.08.007
                PMC ID: 6727031
                SO-VID: 2f2f0f70-0d6f-4a97-a0d3-d5c25db71a79
                Copyright © © 2019 The Authors
                License:

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 18 June 2019
                Date revision received : 2 August 2019
                Date accepted : 3 August 2019
                Categories
                Subject: Multisystem

                Keywords: lipomatous hypertrophy,familial partial lipodystrophy,cta
                Data availability:
                Keywords: lipomatous hypertrophy, familial partial lipodystrophy, cta

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