Type B insulin-resistance syndrome presenting as autoimmune hypoglycemia, associated with systemic lupus erythematosus and interstitial lung disease.
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.
Division of Endocrinology and Metabolism, Department of Internal Medicine, Seoul National University Bundang Hospital, Seongnam, Korea.
Seon Mee
SM
Kang
Heung Yong
HY
Jin
Kyung Ae
KA
Lee
Ji Hyun
JH
Park
Hong Sun
HS
Baek
Tae Sun
TS
Park
D000328:Adult; D001323:Autoantibodies / Q000097:blood*; D015551:Autoimmunity; D015415:Biomarkers / Q000097:blood; D001786:Blood Glucose / Q000378:metabolism; D005260:Female; D006801:Humans; D007003:Hypoglycemia / Q000097:blood / Q000150:complications* / Q000276:immunology; D007328:Insulin / Q000097:blood; D007333:Insulin Resistance; D017563:Lung Diseases, Interstitial / Q000175:diagnosis / Q000209:etiology* / Q000276:immunology / Q000601:surgery; D008180:Lupus Erythematosus, Systemic / Q000150:complications* / Q000175:diagnosis / Q000276:immunology; D011972:Receptor, Insulin / Q000276:immunology*; D020775:Thoracic Surgery, Video-Assisted; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome
D001323:Autoantibodies; D015415:Biomarkers; D001786:Blood Glucose; D007328:Insulin; D011972:Receptor, Insulin
Autoimmune hypoglycemia; Lung diseases, interstitial; Lupus erythematosus, systemic