Reviewer’ s report
Title: A Case Report of Multiple Symmetric Lipomatosis in an East Asian Female
Version: 1Date: 03 Jun 2020
Reviewer's report:Review of Jung K and Lee S, "A case report of Madelung's disease in an East Asian female."
Submitted to BMC Women's Health.
Summary:Madelung's disease is a rare disorder characterised by fat accumulation in specific regions of the
body: type 1 (neck, shoulder), type 2 (gynoid), and type 3 (general). It is more common in males,and often presents due to cosmetic issues with the patient. Excessive alcohol consumption is oftena factor. Here the authors report a case of a 59-year old Korean female with type 1 Madelung'sdisease around the neck and shoulders. The patient also presented with a history of alcohol abuseand exhibited impaired liver function as a result. The fat masses were excised without difficulty,and histological examination revealed large dystrophic adipocyte morphology.
Major Points:This interesting and well-written report describes Madelung's disease, a heterogeneous disorder of
lipid accumulation that is observed in Mediterranean populations, but has not been widelydescribed in Asians. Similarly, more males have been described with Madelung's disease, althoughhas been disputed by a recent large study of Bavarians (Schiltz D et al. Plast Reconstr Surg Glob
Open. 2018 Apr; 6(4): e1722). Overall, the study is sufficiently novel, however.
The authors have covered the main issues associated with this condition (see below), although it is
difficult to generalise with one patient. A history of excessive alcohol consumption is discussed indetail, and is the likely cause of the aberrant liver function results. Interestingly, there was noevidence of neuropathy in this patient, and prior studies have assumed some polyneuropathy anddefective sympathetic activation of lipolysis as a contributing factor to the condition. Themanuscript might be improved by some further discussion of alternative mechanisms linkingalcohol consumption with excessive fat accumulation in this patient.
Does the expanded fat mass associate with a higher prevalence of any obesity-related metabolic
conditions, such as insulin resistance? Given the high fasting glucose levels, it might beinformative to assess this by measuring fasting insulin concentrations, and calculation of aHOMA-IR and HOMA-B scores (or similar) to estimate the degree of insulin resistance.
Apart from the high triglycerides (alcohol-induced?), there does not appear to be any
dyslipidaemia present (total, HDL- and LDL-cholesterol concentrations are all below the referencerange). Given that this type of fat mass is thought to originate from brown/beige fat, is possible
that this additional fat mass confers a metabolic benefit upon the patient?
Mildly elevated creatinene appears to be a common feature of this disorder (Bergler-Czop B et al.
Postepy Dermatol Alergol. 2014 May; 31(2): 121-124. Also Von Kemp et al. Acta Clinica Belgica1986; 41(4): 255-259). What do you think is its relevance in this setting? Kidney failure,dehydration, or do you think there is any relationship with brown fat given recent links withcreatine substrate cycles in the literature (see Gerngross C et al. J Nucl Med July 1, 2017 vol. 58no. 7 1104-1110).
Minor Points:
Do you have any anthropometric measurements (height, weight, BMI, waist, hip circumferences)
from this patient?
Line 106 - the patient may not be glucose intolerant as results from a glucose tolerance test were
not presented. It would be preferable to use the term "impaired fasting glucose".
What was the approximate weight of the adipose mass? If not recorded then can you estimate it
based on the volume of tissue?
Are the methods appropriate and well described?
If not, please specify what is required in your comments to the authors.
YesDoes the work include the necessary controls?
If not, please specify which controls are required in your comments to the authors.
Unable to assess
Are the conclusions drawn adequately supported by the data shown?
If not, please explain in your comments to the authors.
YesAre you able to assess any statistics in the manuscript or would you recommend an
additional statistical review?If an additional statistical review is recommended, please specify what aspects require furtherassessment in your comments to the editors.
Not relevant to this manuscriptQuality of written English
Please indicate the quality of language in the manuscript:
Acceptable
Declaration of competing interests
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