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Hyukwon Chang  (Chang H) 2 Articles
Polyglandular Autoimmune Syndrome Type III with Primary Hypoparathyroidism
Sang Jin Kim, Sang-Yoon Kim, Han-Byul Kim, Hyukwon Chang, Ho-Chan Cho
Endocrinol Metab. 2013;28(3):236-240.   Published online September 13, 2013
DOI: https://doi.org/10.3803/EnM.2013.28.3.236
  • 4,571 View
  • 34 Download
  • 5 Crossref
AbstractAbstract PDFPubReader   

Polyglandular autoimmune syndrome is defined as multiple endocrine gland insufficiencies accompanied by autoimmune diseases of the endocrine and nonendocrine system. After Schmidt introduced a case of nontuberculosis adrenal gland dysfunction with thyroiditis in 1926, Neufeld defined polyglandular autoimmune syndrome by I, II, and III subtypes in 1980 by their presentation of occurrence age, heredity methods, relationship with human leukocyte antigen, and accompanying diseases. We report a case of a 32-year-old female with polyglandular autoimmune syndrome III accompanied by type 1 diabetes mellitus that was treated with insulin (36 units per day) for 11 years. She had insulin deficiency and Hashimoto thyroiditis as an autoimmune disorder. In addition, she had several features similar to Albright's hereditary osteodystrophy including short stature, truncal obesity, round face, short neck, low intelligence (full IQ 84), and decreased memory. Although Albright's hereditary osteodystrophy is morphological evidence of pseudohypoparathyroidism or pseudopseudohypoparathyroidism, she had primary hypoparathyroidism on laboratory results. Here, we report a case of polyglandular autoimmune syndrome III with type 1 diabetes mellitus, autoimmune thyroiditis, and primary hypoparathyroidism, accompanied by clinical features similar to Albright's hereditary osteodystrophy.

Citations

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  • Autoimmune polyglandular syndrome type III associated with antineutrophil cytoplasmic autoantibody-mediated crescentic glomerulonephritis
    Shiyuan Tian, Baofeng Xu, Ziwei Liu, Rui Liu
    Medicine.2020; 99(7): e19179.     CrossRef
  • Hypoparathyroidism: Genetics and Diagnosis
    Michael Mannstadt, Luisella Cianferotti, Rachel I Gafni, Francesca Giusti, Elizabeth Helen Kemp, Christian A Koch, Kelly L Roszko, Liam Yao, Gordon H Guyatt, Rajesh V Thakker, Weibo Xia, Maria-Luisa Brandi
    Journal of Bone and Mineral Research.2020; 37(12): 2615.     CrossRef
  • A rare case of polyglandular autoimmune syndrome type IIIc with primary antibody failure
    Adina Elena Stanciu, Florentina Sava, Gergely Toldi
    Gynecological Endocrinology.2018; 34(4): 283.     CrossRef
  • Autoimmune Polyendocrine Syndrome Type IIIC and Ankylosing Spondylitis; a Case Report
    Ali Javinani, Hamid Reza Aghaei Meybodi, Ahmad Reza Jamshidi, Farhad Gharibdoost, Hoda Kavosi
    Rheumatology Research.2016;[Epub]     CrossRef
  • Brief Review of Articles in 'Endocrinology and Metabolism' in 2013
    Won-Young Lee
    Endocrinology and Metabolism.2014; 29(3): 251.     CrossRef
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The Effect of Octreotide LAR on GH and TSH Co-Secreting Pituitary Adenoma.
Nam Keong Kim, Yu Jin Hah, Ho Young Lee, Sang Jin Kim, Mi Kyung Kim, Keun Gyu Park, Ealmaan Kim, Hyukwon Chang, Hye Soon Kim
Endocrinol Metab. 2010;25(4):378-381.   Published online December 1, 2010
DOI: https://doi.org/10.3803/EnM.2010.25.4.378
  • 1,931 View
  • 30 Download
AbstractAbstract PDF
Growth hormone (GH) and thyroid stimulating hormone (TSH)-secreting pituitary adenomas are very rare and they account for only 0.5% for all pituitary adenomas. These adenomas are usually treated with surgery, but this surgery is not easy because the tumor is usually huge and invasive. We reported here on a case of a GH-TSH-secreting adenoma in a 23-year-old male patient who was initially treated with octreotide LAR. He presented with symptoms of headache, palpitation and a visual defect that he had for the 3 months. He had hypertrophy of the frontal bone and enlargement of both the hands and feet. The visual field test showed bitemporal hemianopsia. The laboratory examinations showed high serum levels of free T4, TSH and free alpha-subunit. Additionally, the serum levels of GH and insulin-like growth factor-I (IGF-I) were increased. GH was not suppressed below 1microg/L by an oral 75g glucose loading test, and TSH was not stimulated by thyrotropin-releasing hormone (TRH). Because sellar MRI showed invasive macroadenoma encasing the vessels, we initially tried octreotide LAR for treatment. A year later, the IGF-I and thyroid function tests were normalized and the size of the tumor was reduced with cystic change. The symptoms of palpitation and headache were improved without a change of the visual field defect.
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