Type B insulin-resistance syndrome presenting as autoimmune hypoglycemia, associated with systemic lupus erythematosus and interstitial lung disease

Korean J Intern Med. 2013 Jan;28(1):98-102. doi: 10.3904/kjim.2013.28.1.98. Epub 2012 Dec 28.

Abstract

We describe an unusual case of systemic lupus erythematosus with pulmonary manifestations presenting as hypoglycemia due to anti-insulin receptor antibodies. A 38-year-old female suffered an episode of unconsciousness and was admitted to hospital where her blood glucose was found to be 18 mg/dL. During the hypoglycemic episode, her serum insulin level was inappropriately high (2,207.1 pmol/L; normal range, 18 to 173) and C-peptide level was elevated (1.7 nmol/L; normal range, 0.37 to 1.47). Further blood tests revealed the presence of antinuclear antibodies, anti-double-stranded DNA antibodies, and anti-Ro/SSA, anti-La/SSB, anti-ribonucleoprotein, and anti-insulin receptor antibodies. A computed tomography scan of the abdomen, aimed at tumor localization, such as an insulinoma, instead revealed ground-glass opacities in both lower lungs, and no abnormal finding in the abdomen. For a definitive diagnosis of the lung lesion, video-associated thoracoscopic surgery was performed and histopathological findings showed a pattern of fibrotic non-specific interstitial pneumonia.

Keywords: Autoimmune hypoglycemia; Lung diseases, interstitial; Lupus erythematosus, systemic.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Autoantibodies / blood*
  • Autoimmunity*
  • Biomarkers / blood
  • Blood Glucose / metabolism
  • Female
  • Humans
  • Hypoglycemia / blood
  • Hypoglycemia / complications*
  • Hypoglycemia / immunology
  • Insulin / blood
  • Insulin Resistance*
  • Lung Diseases, Interstitial / diagnosis
  • Lung Diseases, Interstitial / etiology*
  • Lung Diseases, Interstitial / immunology
  • Lung Diseases, Interstitial / surgery
  • Lupus Erythematosus, Systemic / complications*
  • Lupus Erythematosus, Systemic / diagnosis
  • Lupus Erythematosus, Systemic / immunology
  • Receptor, Insulin / immunology*
  • Thoracic Surgery, Video-Assisted
  • Tomography, X-Ray Computed
  • Treatment Outcome

Substances

  • Autoantibodies
  • Biomarkers
  • Blood Glucose
  • Insulin
  • Receptor, Insulin