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      Caso original de lipo-hipertrofia simétrica: relato de caso e revisão de literatura Translated title: An original case of symmetric lipohypertrophy: case report and review of the literature

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          Abstract

          RESUMO Paciente do sexo feminino, saudável, meia-idade e com história prolongada de massas com crescimento lento localizadas bilateralmente e simetricamente nos membros superiores e inferiores, próximas às principais articulações. Exames de imagem e patológicos após excisão das massas revelaram massas constituídas por gordura subcutânea normal. Não houve evidências de lipomas bem-circunscritos. Na revisão de literatura, destacaramse diversos distúrbios lipo-hipertróficos, com possível apresentação simétrica. As características das afecções encontradas não eram, porém, totalmente concordantes com as características de nossa paciente. Embora não de modo absoluto, a doença que mais estreitamente se assemelhou ao caso foi lipodistrofia simétrica rara, conhecida como doença de Madelung. Dos 150 casos relatados, em apenas 3 foi descrito comprometimento dos membros inferiores, como ocorreu em nosso caso. Este relato apresenta descrição detalhada do caso, seu manejo e seguimento no pós-operatório. Os tipos distintos de lipodistrofias simétricas também são discutidos.

          Translated abstract

          ABSTRACT A middle-aged healthy woman who presented with longstanding history of slow growing masses located bilaterally and symmetrically on the upper and lower extremities closed to major joints. Imaging and pathology tests following excision of masses revealed normal subcutaneous fat. There was no evidence of well-circumscribed lipomas. A review of the literature identified a number of lipohypertrophic disorders, which may be present in a symmetrical fashion. The characteristics of the disorders, however, did not fully agree with characteristics observed in our patient. Although not absolute, the closest disease found to fit our case is a rare symmetrical lipodystrophy known as Madelung’s disease. Of 150 reported cases, only 3 described involvement of lower extremities as seen in our case. We report a detailed description of a case, its management and post-operative follow-up. Different types of symmetrical lipodytrophies are also discussed.

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          Most cited references10

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          Lipohypertrophy in children and adolescents with type 1 diabetes and the associated factors

          Background Despite the important implications of lipohypertrophy for diabetes control, there is a dearth of information and research about the subject in children. The aim of this study was to study the prevalence of lipohypertrophy in children with type 1 diabetes, and to evaluate the associated factors. Findings 119 children coming for regular follow up in the diabetes clinic were examined for the presence of lipohypertrophy by inspection and palpation. The last 4 readings of glycated hemoglobin (HbA1c) levels and other factors that may affect lipohypertrophy were documented. RESULTS: The patient's age ranged from 2 months to 21 years with a median of 10 years (inter-quartile range = 6). Lipohypertrophy occurred in 54.9% of patients, more commonly in males (62.7%) vs. females (48.4%) (P = 0.074). Grade 1 lipohypertrophy occurred in 42.5% and grade 2 in 12.4%. Lipohypertrophy was related significantly to the dose of insulin units per kg of body weight (Odds ratio [OR] = 16.4; 95% CI, 2.2 - 124.6; P = 0.007), the duration of diabetes, [OR] = 1.16; 95% CI, 1.05 - 1.32; P = 0.004)), and the body mass index (BMI) [OR] = 1.68; 95% CI, 1.25 - 2.15; P = 0.006). The mean HbA1c levels of patients with grade 1 and grade 2 lipohypertrophy did not differ from diabetics without lipohypertrophy (F = 0.178, P = 0.837) Conclusions The presence of lipohypertrophy was significantly associated with the duration of diabetes and the body mass index. Children with lipohypertrophy needed a significantly higher dose of insulin units/kg of body weight to achieve fair control compared to children without lipohypertrophy. Further studies are needed to ascertain the clinical meaning of these findings.
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            Lipodystrophy in HIV patients: its challenges and management approaches

            HIV-associated lipodystrophy is a term used to describe a constellation of body composition (lipoatrophy and lipohypertrophy) and metabolic (dyslipidemia and insulin resistance) alterations that accompany highly active antiretroviral therapy. These changes, which resemble metabolic syndrome, have been associated with a variety of adverse outcomes including accelerated cardiovascular disease. The body composition and metabolic changes appear to cluster in HIV infection, although they are distinct alterations and do not necessarily coexist. Epidemiological studies have demonstrated multiple pathogenic influences associated with host, disease, and treatment-related factors. The adverse treatment effects were more prominent in early regimens; continued drug development has led to the application of metabolically safer regimens with equal or greater potency than the regimens being replaced. Disease-related factors include HIV infection as well as inflammation, immune activation, and immune depletion. The body composition changes promote anxiety and depression in patients and may affect treatment adherence. Treatment of dyslipidemia and alterations in glucose metabolism is the same as in non-HIV-infected individuals. Lipoatrophy is managed by strategic choice of antivirals or by antiviral switching, and in some cases by plastic/reconstructive surgery. Lipohypertrophy has been managed mainly by lifestyle modification, ie, a hypocaloric diet and increased exercise. A growth hormone releasing factor, which reduces central fat, has recently become available for clinical use.
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              Harper’s textbook of pediatric dermatology

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                Author and article information

                Journal
                rbcp
                Revista Brasileira de Cirurgia Plástica
                Rev. Bras. Cir. Plást.
                Sociedade Brasileira de Cirurgia Plástica (São Paulo, SP, Brazil )
                1983-5175
                2177-1235
                2016
                : 31
                : 3
                : 424-427
                Affiliations
                [1] Beirut orgnameAmerican University of Beirut Medical Center Lebonon
                Article
                S1983-51752016000300424 S1983-5175(16)03100300424
                10.5935/2177-1235.2016rbcp0069
                a096d605-80b2-42e3-a5fd-6644785ae472

                This work is licensed under a Creative Commons Attribution 4.0 International License.

                History
                : 08 October 2015
                : 19 July 2015
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 10, Pages: 4
                Product

                SciELO Brazil

                Categories
                Relatos de Caso

                Lower extremity.,Lipodystrophy,Lipoma,Subcutaneous fat,Lipomatosis,multiple symmetrical,Lipodistrofia,Gordura subcutânea,Lipomatose,Simétrica múltipla,Membro inferior.

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