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Cited by 28Diabetes Management and ResearchPancreatic function and diabetesGrowth Hormone and Insulin-like Growth Factors

Metabolic and endocrine studies in a case of lipoatrophic diabetes

Rossini, Aldo A., Self, Jane, Aoki, Thomas T., Goldman, Ralph F., Newmark, Stephen R., Meguid, Michael M., Soeldner, J.Stuart, Cahill, George F.·1977·Metabolism
DOI 10.1016/0026-0495(77)90085-3
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Abstract

A 20-yr-old female with congenital lipoatrophic diabetes was studied, with the following findings: (1) Serum insulin levels increased after both oral glucose and intravenous arginine administration; there was no growth hormone response to the latter. (2) The infusion of insulin (0.1 units and 0.5 units/kg) during the fed state and following at 110-hr fast produced only minimal changes of various fuels measured, with the exception of a decrease in the branched-chain amino acids. (3) There was a minimal production of ketones during the 110-hr fast. (4) Matabolic expenditure was markedly increased during the postabsorptive state (65-75 kcal/hr/sg m); it fell into the normal range during the 110-hr fast (31-35 kcal/hr/sq m). (5) Following meals, the patient experienced complaints ranging from cold and shivering to feeling hot with gross diaphoresis. These findings were associated with intermittent lability of her skin temperature, which varied 1 degree - 2 degrees F during a 3-hr period. (6) Progressive increases in doses of regular insulin before each meal resulted in up to a total of 9000 units/day being required before normal blood glucose levels were achieved. (7) A 2-wk therapeutic trial of pimozide provided no significant changes in a variety of hormones and fuels in the basal state or following insulin perturbations. (8) A variety of pituitary hormones and pituitary target organ hormones were studied in both the hypothyroid (Hashimoto's thyroiditis) and euthyroid state (following thyroid replacement). All the hormone responses were normal except that growth hormone did not rise during the slow wave sleep in either thyroid state.

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Crossref · 10.1016/0026-0495(77)90085-3

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24 of 24 references verified0 citation errors · 0 not checkable

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  • Lipodystrophy is a rare disorder characterized by selective loss of subcutaneous and visceral fat and is associated with hypertriglyceridemia, hepatic steatosis, and severe insulin resistance that often results in diabetes.

    “Lipodystrophy is a rare disorder that is characterized by selective loss of subcutaneous and visceral fat. This disease is associated with hypertriglyceridemia, hepatic steatosis, and severe insulin resistance that often results in diabetes (1-3).”

    From Leptin reverses insulin resistance and hepatic steatosis in patients with severe lipodystrophy (2002) · cites this paper as [1]

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Authors
Rossini, A. A., Self, J., Aoki, T. T., Goldman, R. F., Newmark, S. R., Meguid, M. M., Soeldner, J. S., & Cahill, G. F.
Affiliations
Harvard University (US)
Corresponding author
Rossini, Aldo A.
Venue
Metabolism
Publisher
Elsevier BV
Published
1977-06
Volume
26
Issue
6
Pages
637-650
ISSN
0026-0495 (ISSN-L)
DOI
10.1016/0026-0495(77)90085-3
OpenAlex
W1963665732
Cited by
28
References
24
Citations by year
2025: 1 · 2016: 1 · 2014: 1 · 2012: 2
FWCI
1.48
Type
journal article
Language
en
Open access
closed
Licence
Locations
  • Metabolism (publishedVersion)
  • PubMed (publishedVersion)
Topics
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Diabetes Management and Research
  • Health Sciences › Medicine › Surgery › Pancreatic function and diabetes
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Growth Hormone and Insulin-like Growth Factors
Keywords
lipoatrophic diabetes, congenital diabetes, insulin resistance, insulin requirement, growth hormone, branched-chain amino acids, ketogenesis, basal metabolic rate, skin temperature, pimozide, Hashimoto's thyroiditis, thyroid hormone replacement, slow-wave sleep, pituitary hormones
MeSH terms
Adult, Basal Metabolism, Blood Glucose, Body Temperature, Diabetes Mellitus, Fasting, Female, Glycosuria, Humans, Hypothyroidism, Insulin, Lipodystrophy, Pimozide, Proteinuria, Growth Hormone, Thyroid Hormones, Diabetes Complications

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