Literature DB >> 28754454

Clinical spectra of neuromuscular manifestations in patients with lipodystrophy: A multicenter study.

Gulcin Akinci1, Haluk Topaloglu2, Tevfik Demir3, Ayca Ersen Danyeli4, Beril Talim5, Fatma Ela Keskin6, Pinar Kadioglu7, Enez Talip3, Canan Altay8, Guzin Fidan Yaylali9, Habib Bilen10, Banu Nur11, Leyla Demir12, Huseyin Onay13, Baris Akinci3.   

Abstract

Lipodystrophy is a heterogeneous group of disorders characterized by loss of adipose tissue. Here, we report on clinical spectra of neuromuscular manifestations of Turkish patients with lipodystrophy. Seventy-four patients with lipodystrophy and 20 healthy controls were included. Peripheral sensorimotor neuropathy was a common finding (67.4%) in lipodystrophic patients with diabetes. Neuropathic foot ulcers were observed in 4 patients. Drop foot developed in 1 patient with congenital generalized lipodystrophy type 1. Muscle symptoms and hypertrophy were consistent findings in congenital generalized lipodystrophy (21/21) and familial partial lipodystrophy (25/34); on the other hand, overt myopathy with elevated creatine kinase activity was a distinctive characteristic of congenital generalized lipodystrophy type 4. Muscle biopsies revealed myopathic changes at different levels. Accumulation of triglycerides was observed which contributes to insulin resistance. All patients with congenital generalized lipodystrophy suffered from tight Achilles tendons at various levels. Scoliosis was observed in congenital generalized lipodystrophy type 4 (2/2) and familial partial lipodystrophy type 2 (2/17). Atlantoaxial instability was unique to congenital generalized lipodystrophy type 4 (2/2). Bone cysts were detected in congenital generalized lipodystrophy type 1 (7/10) and congenital generalized lipodystrophy type 2 (2/8). Our study suggests that lipodystrophies are associated with a wide spectrum of neuromuscular abnormalities.
Copyright © 2017 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Insulin resistance; Lipodystrophy; Myopathy; Neuropathy

Mesh:

Substances:

Year:  2017        PMID: 28754454     DOI: 10.1016/j.nmd.2017.05.015

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  7 in total

Review 1.  Phenotypic and Genetic Characteristics of Lipodystrophy: Pathophysiology, Metabolic Abnormalities, and Comorbidities.

Authors:  Baris Akinci; Rasimcan Meral; Elif Arioglu Oral
Journal:  Curr Diab Rep       Date:  2018-11-08       Impact factor: 4.810

2.  Fatty Liver and Autoimmune Hepatitis: Two Forms of Liver Involvement in Lipodystrophies.

Authors:  Andreia Ribeiro; José Ricardo Brandão; Esmeralda Cleto; Manuela Santos; Teresa Borges; Ermelinda Santos Silva
Journal:  GE Port J Gastroenterol       Date:  2019-01-30

3.  A Patient with Berardinelli-Seip Syndrome, Novel AGPAT2 Splicesite Mutation and Concomitant Development of Non-diabetic Polyneuropathy

Authors:  Joanna Oswiecimska; Mateusz Dawidziuk; Tomasz Gambin; Katarzyna Ziora; Marta Marek; Sylwia Rzonca; D. Lys Guilbride; Shalini N. Jhangiani; Anna Obuchowicz; Alicja Sikora; James R. Lupski; Wojciech Wiszniewski; Pawel Gawlinski
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-12-19

Review 4.  Current Diagnosis, Treatment and Clinical Challenges in the Management of Lipodystrophy Syndromes in Children and Young People

Authors:  Samim Özen; Barış Akıncı; Elif A. Oral
Journal:  J Clin Res Pediatr Endocrinol       Date:  2019-08-22

5.  A systematic review and meta-analysis of the serum lipid profile in prediction of diabetic neuropathy.

Authors:  Zixin Cai; Yan Yang; Jingjing Zhang
Journal:  Sci Rep       Date:  2021-01-12       Impact factor: 4.379

6.  Altered acylated ghrelin response to food intake in congenital generalized lipodystrophy.

Authors:  Camilla O D Araújo; Renan M Montenegro; Amanda P Pedroso; Virgínia O Fernandes; Ana Paula D R Montenegro; Annelise B de Carvalho; Lila M Oyama; Carla S C Maia; Eliane B Ribeiro
Journal:  PLoS One       Date:  2021-01-07       Impact factor: 3.240

7.  Leptin mediates the regulation of muscle mass and strength by adipose tissue.

Authors:  Kelsey H Collins; Chang Gui; Erica V Ely; Kristin L Lenz; Charles A Harris; Farshid Guilak; Gretchen A Meyer
Journal:  J Physiol       Date:  2022-08-02       Impact factor: 6.228

  7 in total

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