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  • Original Article
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Lipomatosis-associated inflammation and excess collagen may contribute to lower relative resting energy expenditure in women with adiposis dolorosa

International Journal of Obesity volume 33, pages 1031–1038 (2009)Cite this article

Abstract

Background:

Adiposis dolorosa (AD) is a syndrome of obese and non-obese individuals whose hallmark is lipomatosis: unencapsulated painful fatty masses in subcutaneous fat. Lipomatosis may contain excess collagen and multi-nucleated giant (MNG) cells. Case reports suggest metabolic defects in AD.

Objectives:

(1) To determine whether women with AD have altered relative resting energy expenditure (REE per total body mass) compared with controls; and (2) to quantitate lipomatosis-associated collagen, MNGs and tissue and blood cytokines that may influence REE.

Methods:

A total of 10 women with AD were compared with age, body mass index, fat and weight-matched control women. Adipose tissue was obtained from five women with AD and five controls and evaluated for collagen and macrophages/MNGs. Fat mass and fat-free mass were identified by dual X-ray absorptiometry. REE was by determined indirect calorimetry and related to mass. Adipokines and cytokines were evaluated in blood and tissue.

Results:

Relative REE (REE per total body mass) was lower in women with AD compared with controls (P=0.007). Only lipomatosis (group) and total body mass were significant predictors of REE in forward stepwise regression (P<0.0001). Adipose interleukin (IL)-6 levels were elevated (P=0.03) and connective tissue was increased fourfold in lipomatosis compared with control tissue (P <0.0001). There was no difference in adipose tissue macrophages between groups; 30% of women with AD had MNG cells. Anti-inflammatory IL-13 levels were elevated (P=0.03), and cytokines important in the recruitment of monocytes, Fraktalkine (P=0.04) and macrophage inflammatory protein-1β (P=0.009), were significantly lower in the blood of women with AD compared with controls.

Conclusions:

The lower relative REE in women with AD compared with controls was associated with increased connective (non-metabolic) tissue in the lipomatosis, and inflammation, although underlying metabolic defects may be important as well. Understanding the pathophysiology and metabolism of lipomatosis in AD may contribute to a better understanding of metabolism in non-lipomatosis obesity.

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Acknowledgements

We thank Patrick Tso for his support, Dana Lee for performing and analyzing the cytokine and lipid data and Tom Storer for his critical read of the indirect calorimetry aspect of this paper. We also extend our sincere gratitude to the women who traveled and took time out from their lives to be a part of this study. This work was supported by GCRC Grant 5M01 RR000827 and NIH NIDDK Grant K23 DK 065038-05.

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Authors and Affiliations

  1. Department of Medicine, University of California San Diego, San Diego, CA, USA

    K L Herbst, A Chang & D L Boyle

  2. Department of Medicine, Veteran's Affairs San Diego Healthcare System, San Diego, CA, USA

    K L Herbst

  3. Department of Medicine, Boston University School of Medicine, Boston, MA, USA

    A D Coviello

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Herbst, K., Coviello, A., Chang, A. et al. Lipomatosis-associated inflammation and excess collagen may contribute to lower relative resting energy expenditure in women with adiposis dolorosa. Int J Obes 33, 1031–1038 (2009). https://doi.org/10.1038/ijo.2009.119

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