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

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

• Kang SM
• Jin HY
• Lee KA
• Park JH
• Baek HS
• Park TS

• KMID: 1793225
• DOI: http://doi.org/10.3904/kjim.2013.28.1.98

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.

Keyword

Autoimmune hypoglycemia; Lupus erythematosus, systemic; Lung diseases, interstitial

MeSH Terms

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