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Central Diabetes Insipidus in a Patient with Stiff Person Syndrome

Received: 14 August 2019     Accepted: 6 September 2019     Published: 19 November 2019
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Abstract

Background: Central diabetes insipidus (CDI) is a rare hypothalamic-pituitary disease due to the deficiency of arginine vasopressin (AVP) synthesis of which more than one third of etiologies are unidentified. Autoimmunity is associated with one third of patients with apparently idiopathic CDI. The most common antibody detected in auto immune CDI is autoantibodies to AVP-secreting cells (AVPcAb). Stiff-person syndrome (SPS) is a rare neurological disorder with features of an autoimmune disease. It is characterized by fluctuating muscle rigidity in the trunk and limbs and a heightened sensitivity to stimuli. It is well known that SPS is associated with multiple auto immune endocrinopathy including auto immune diabetes and auto immune thyroid disease. The association between SPS and CDI is not well documented in the literature. Case information: We report a case of 51 year old female who developed CDI while being treated for SPS unmasked by high dose steroids. Result: Anti amphiphysin is the only antibody detected in our patient. Animal studies showed a high expression of amphiphysin in anterior and posterior pituitary gland, data in human are still vague .One theory is that anti-amphiphysin antibodies attack the amphiphysin molecules in the AVP secreting cells and inhibit release of AVP. Conclusion: This antibody could hint towards the autoimmune cause of CDI.

Published in American Journal of Internal Medicine (Volume 7, Issue 6)
DOI 10.11648/j.ajim.20190706.13
Page(s) 151-153
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2019. Published by Science Publishing Group

Keywords

Central Diabetes Insipidus, Stiff Person Syndrome, Anti Amphyphysin Antibodies

References
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[2] Juul KV, Bichet DG, Nielsen S, Nørgaard JP. The physiological and pathophysiological functions of renal and extrarenal vasopressin V2 receptors. American Journal of Physiology-Renal Physiology. 2014; 306 (9).
[3] Iorgi ND, Napoli F, Allegri AEM, Olivieri I, Bertelli E, Gallizia A, et al. Diabetes Insipidus – Diagnosis and Management. Hormone Research in Paediatrics. 2012; 77 (2): 69–84.
[4] Murdaca G, Russo R, Spanò F, Ferone D, Albertelli M, Schenone A, Contatore M, Guastalla A, De Bellis A, Garibotto G, Puppo F. Autoimmune central diabetes insipidus in a patient with ureaplasma urealyticum infection and review on new triggers of immune response. Arch Endocrinol Metab. 2015 Dec; 59 (6): 554-8.
[5] Christ-Crain M, Bichet D G, Fenske W K, Goldman M B, Rittig S, Verbalis J G, & Verkman A S. Diabetes insipidus. Nature Reviews Disease Primers. 2019; 5 (1).
[6] Maghnie M, Ghirardello S, Bellis AD, Iorgi ND, Ambrosini L, Secco A, et al. Idiopathic central diabetes insipidus in children and young adults is commonly associated with vasopressin-cell antibodies and markers of autoimmunity. Clinical Endocrinology. 2006; 65 (4): 470–8.
[7] Pivonello R, Bellis AD, Faggiano A, Salle FD, Petretta M, Somma CD, et al. Central Diabetes Insipidus and Autoimmunity: Relationship between the Occurrence of Antibodies to Arginine Vasopressin-Secreting Cells and Clinical, Immunological, and Radiological Features in a Large Cohort of Patients with Central Diabetes Insipidus of Known and Unknown Etiology. The Journal of Clinical Endocrinology & Metabolism. 2003; 88 (4): 1629–36.
[8] Garrahy A, Moran C, Thompson C et al. Diagnosis and management of central diabetes insipidus in adults. Clinical Endocrinology. 2019; 90: 23–30.
[9] Martinez-Hernandez E, Arino H, McKeon A, et al. Clinical and immunologic investigations in patients with stiff-person spectrum disorder. JAMA Neurol. 2016; 73: 714–20.
[10] Rathbun J, Imber J et al. Stiff Person Syndrome and Type 1 Diabetes Mellitus: a Case of the Chicken or the Egg? Journal of Genral Internal Medicine. 2019; 34 (6): 1053-10574.
[11] Robert L, Julius C et al. Atlas of immunology. 2004 (book).
[12] Beate L, Rudiger WV, Helmut E. M, Manfred WK. et al. Amphiphysin, a novel protein associated with synaptic vesicles. The EMBO Journal vol. 1 1 no. 7 pp. 2521-2530, 1 992.
[13] Holst B, Madsen KL, Jansen AM, Jin C, Rickhag M, Lund VK, et al. PICK1 Deficiency Impairs Secretory Vesicle Biogenesis and Leads to Growth Retardation and Decreased Glucose Tolerance. PLoS Biology. 2013; 11 (4).
[14] L Qiaou. Et al. Paraneoplastic neurological syndromes associated with anti-amphiphysin antibodie. October 2016.
[15] Chin H, Quek T, Leow M. Central diabetes insipidus unmasked by corticosteroid therapy for cerebral metastases: beware the case with pituitary involvement and hypopituitarism. Journal of the Royal College of Physicians of Edinburgh. 2018; 47 (3): 247–9.
Cite This Article
  • APA Style

    Fatima Zarzour, Abir Bou Khalil, Sami Toufic Azar. (2019). Central Diabetes Insipidus in a Patient with Stiff Person Syndrome. American Journal of Internal Medicine, 7(6), 151-153. https://doi.org/10.11648/j.ajim.20190706.13

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    ACS Style

    Fatima Zarzour; Abir Bou Khalil; Sami Toufic Azar. Central Diabetes Insipidus in a Patient with Stiff Person Syndrome. Am. J. Intern. Med. 2019, 7(6), 151-153. doi: 10.11648/j.ajim.20190706.13

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    AMA Style

    Fatima Zarzour, Abir Bou Khalil, Sami Toufic Azar. Central Diabetes Insipidus in a Patient with Stiff Person Syndrome. Am J Intern Med. 2019;7(6):151-153. doi: 10.11648/j.ajim.20190706.13

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  • @article{10.11648/j.ajim.20190706.13,
      author = {Fatima Zarzour and Abir Bou Khalil and Sami Toufic Azar},
      title = {Central Diabetes Insipidus in a Patient with Stiff Person Syndrome},
      journal = {American Journal of Internal Medicine},
      volume = {7},
      number = {6},
      pages = {151-153},
      doi = {10.11648/j.ajim.20190706.13},
      url = {https://doi.org/10.11648/j.ajim.20190706.13},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ajim.20190706.13},
      abstract = {Background: Central diabetes insipidus (CDI) is a rare hypothalamic-pituitary disease due to the deficiency of arginine vasopressin (AVP) synthesis of which more than one third of etiologies are unidentified. Autoimmunity is associated with one third of patients with apparently idiopathic CDI. The most common antibody detected in auto immune CDI is autoantibodies to AVP-secreting cells (AVPcAb). Stiff-person syndrome (SPS) is a rare neurological disorder with features of an autoimmune disease. It is characterized by fluctuating muscle rigidity in the trunk and limbs and a heightened sensitivity to stimuli. It is well known that SPS is associated with multiple auto immune endocrinopathy including auto immune diabetes and auto immune thyroid disease. The association between SPS and CDI is not well documented in the literature. Case information: We report a case of 51 year old female who developed CDI while being treated for SPS unmasked by high dose steroids. Result: Anti amphiphysin is the only antibody detected in our patient. Animal studies showed a high expression of amphiphysin in anterior and posterior pituitary gland, data in human are still vague .One theory is that anti-amphiphysin antibodies attack the amphiphysin molecules in the AVP secreting cells and inhibit release of AVP. Conclusion: This antibody could hint towards the autoimmune cause of CDI.},
     year = {2019}
    }
    

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    AU  - Fatima Zarzour
    AU  - Abir Bou Khalil
    AU  - Sami Toufic Azar
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    PY  - 2019
    N1  - https://doi.org/10.11648/j.ajim.20190706.13
    DO  - 10.11648/j.ajim.20190706.13
    T2  - American Journal of Internal Medicine
    JF  - American Journal of Internal Medicine
    JO  - American Journal of Internal Medicine
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    AB  - Background: Central diabetes insipidus (CDI) is a rare hypothalamic-pituitary disease due to the deficiency of arginine vasopressin (AVP) synthesis of which more than one third of etiologies are unidentified. Autoimmunity is associated with one third of patients with apparently idiopathic CDI. The most common antibody detected in auto immune CDI is autoantibodies to AVP-secreting cells (AVPcAb). Stiff-person syndrome (SPS) is a rare neurological disorder with features of an autoimmune disease. It is characterized by fluctuating muscle rigidity in the trunk and limbs and a heightened sensitivity to stimuli. It is well known that SPS is associated with multiple auto immune endocrinopathy including auto immune diabetes and auto immune thyroid disease. The association between SPS and CDI is not well documented in the literature. Case information: We report a case of 51 year old female who developed CDI while being treated for SPS unmasked by high dose steroids. Result: Anti amphiphysin is the only antibody detected in our patient. Animal studies showed a high expression of amphiphysin in anterior and posterior pituitary gland, data in human are still vague .One theory is that anti-amphiphysin antibodies attack the amphiphysin molecules in the AVP secreting cells and inhibit release of AVP. Conclusion: This antibody could hint towards the autoimmune cause of CDI.
    VL  - 7
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Author Information
  • Department of Endocrinology and Metabolism, American University of Beirut Medical Center, Beirut, Lebanon

  • Department of Endocrinology and Metabolism, American University of Beirut Medical Center, Beirut, Lebanon

  • Department of Endocrinology and Metabolism, American University of Beirut Medical Center, Beirut, Lebanon

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