The Korean journal of internal medicine

  • 제28권1호
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  • Pages.98-102
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  • 2013
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  • 1226-3303(pISSN)
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  • 2005-6648(eISSN)
대한내과학회 (The Korean Association of Internal Medicine)
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Type B insulin-resistance syndrome presenting as autoimmune hypoglycemia, associated with systemic lupus erythematosus and interstitial lung disease

  • Kang, Seon Mee (Division of Endocrinology and Metabolism, Department of Internal Medicine, Seoul National University Bundang Hospital) ;
  • Jin, Heung Yong (Division of Endocrinology and Metabolism, Department of Internal Medicine, Research Institute of Clinical Medicine, Chonbuk National University Medical School) ;
  • Lee, Kyung Ae (Division of Endocrinology and Metabolism, Department of Internal Medicine, Research Institute of Clinical Medicine, Chonbuk National University Medical School) ;
  • Park, Ji Hyun (Division of Endocrinology and Metabolism, Department of Internal Medicine, Research Institute of Clinical Medicine, Chonbuk National University Medical School) ;
  • Baek, Hong Sun (Division of Endocrinology and Metabolism, Department of Internal Medicine, Research Institute of Clinical Medicine, Chonbuk National University Medical School) ;
  • Park, Tae Sun (Division of Endocrinology and Metabolism, Department of Internal Medicine, Research Institute of Clinical Medicine, Chonbuk National University Medical School)
  • 발행 : 2013.01.01
⟨ 이전 논문 다음 논문 ⟩

초록

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.

키워드

참고문헌

  1. Redmon JB, Nuttall FQ. Autoimmune hypoglycemia. Endocrinol Metab Clin North Am 1999;28:603-618. https://doi.org/10.1016/S0889-8529(05)70090-6
  2. Arioglu E, Andewelt A, Diabo C, Bell M, Taylor SI, Gorden P. Clinical course of the syndrome of autoantibodies to the insulin receptor (type B insulin resistance): a 28-year perspective. Medicine (Baltimore) 2002;81:87-100. https://doi.org/10.1097/00005792-200203000-00001
  3. Kahn CR, Flier JS, Bar RS, et al. The syndromes of insulin resistance and acanthosis nigricans. Insulinreceptor disorders in man. N Engl J Med 1976;294:739-745. https://doi.org/10.1056/NEJM197604012941401
  4. Flier JS, Eastman RC, Minaker KL, Matteson D, Rowe JW. Acanthosis nigricans in obese women with hyperandrogenism: characterization of an insulin-resistant state distinct from the type A and B syndromes. Diabetes 1985;34:101-107.
  5. Lupsa BC, Chong AY, Cochran EK, Soos MA, Semple RK, Gorden P. Autoimmune forms of hypoglycemia. Medicine (Baltimore) 2009;88:141-153. https://doi.org/10.1097/MD.0b013e3181a5b42e
  6. Chon S, Choi MC, Lee YJ, et al. Autoimmune hypoglycemia in a patient with characterization of insulin receptor autoantibodies. Diabetes Metab J 2011;35:80-85. https://doi.org/10.4093/dmj.2011.35.1.80
  7. Kim MJ, Shim MS, Kim MK, et al. A case of autoimmune hypoglycemia induced by insulin autoantibody and insulin receptor autoantibody. Diabetes Monit 2002;3:177-184.
  8. Pego-Reigosa JM, Medeiros DA, Isenberg DA. Respiratory manifestations of systemic lupus erythematosus: old and new concepts. Best Pract Res Clin Rheumatol 2009;23:469-480. https://doi.org/10.1016/j.berh.2009.01.002
  9. Orens JB, Martinez FJ, Lynch JP 3rd. Pleuropulmonary manifestations of systemic lupus erythematosus. Rheum Dis Clin North Am 1994;20:159-193.
  10. Tansey D, Wells AU, Colby TV, et al. Variations in histological patterns of interstitial pneumonia between connective tissue disorders and their relationship to prognosis. Histopathology 2004;44:585-596. https://doi.org/10.1111/j.1365-2559.2004.01896.x

피인용 문헌

  1. Type B insulin resistance syndrome vol.23, pp.4, 2013, https://doi.org/10.1097/med.0000000000000263
  2. The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis vol.54, pp.11, 2013, https://doi.org/10.1515/cclm-2015-1255
  3. The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis vol.54, pp.11, 2013, https://doi.org/10.1515/cclm-2015-1255
  4. Combined Immunosuppressive Therapy Induces Remission in Patients With Severe Type B Insulin Resistance: A Prospective Cohort Study vol.41, pp.11, 2013, https://doi.org/10.2337/dc18-0884
  5. Type B insulin resistance syndrome associated with connective tissue disease and psoriasis vol.2020, pp.None, 2013, https://doi.org/10.1530/edm-20-0027