problendoПроблемы ЭндокринологииProblems of Endocrinology0375-96602308-1430Endocrinology Research Centre10.14341/probl13384problendo-13384Research ArticleКлиническая эндокринологияClinical endocrinologyГипофизит во время беременности с исходом в стойкий несахарный диабетHypophisitis in pregnant women with persistent diabetes insipidus in the outcomehttps://orcid.org/0000-0002-0327-4619ДзерановаЛ. К.DzeranovaL. K.

Дзеранова Лариса Константиновна - д.м.н.

Москва

Larisa K. Dzeranova - MD, PhD.

Moscow

dzeranovalk@yandex.ruhttps://orcid.org/0000-0001-6539-466XПигароваЕ. А.PigarovaE. A.

Пигарова Екатерина Александровна - д.м.н.

Москва

Ekaterina A. Pigarova

Moscow

kpigarova@gmail.comhttps://orcid.org/0000-0001-7470-1676ВоротниковаС. Ю.VorotnikovaS. Yu.

Воротникова Светлана Юрьевна - к.м.н.

Москва

Svetlana Y. Vorotnikova – MD.

Moscow

bra_svetix@list.ruhttps://orcid.org/0000-0001-8838-8908ВознесенскаяА. А.VoznesenskayaA. A.

Вознесенская Анастасия Александровна

117036, Москва, ул. Дм. Ульянова, д. 11

Anastasia A. Voznesenskaya - post-graduate student.

11 Dm. Ulyanova street, 117036 Moscow

Voznesenskaya.A.A@yandex.ruНациональный медицинский исследовательский центр эндокринологииРоссияEndocrinology Research CentreRussian Federation2024150920247041523Copyright © Дзеранова Л.К., Пигарова Е.А., Воротникова С.Ю., Вознесенская А.А., 20242024Дзеранова Л.К., Пигарова Е.А., Воротникова С.Ю., Вознесенская А.А.Dzeranova L.K., Pigarova E.A., Vorotnikova S.Y., Voznesenskaya A.A.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://www.probl-endojournals.ru/jour/article/view/13384

Аутоиммунный/лимфоцитарный гипофизит является одной из редких причин развития центрального несахарного диабета у взрослых пациентов и наиболее часто встречается среди женщин во втором или третьем триместрах беременности.

В многочисленных исследованиях показано, что лимфоцитарный гипофизит характеризуется весьма вариабельной клинической картиной с развитием неврологической симптоматики, нарушений со стороны зрения и гипопитуитаризма с частичным или полным выпадением функций гипофиза, а также рядом особенностей при магнитно-резонансной томографии (МРТ).

Изолированный лимфоцитарный инфудибулонейрогипофизит встречается значительно реже и затрагивает заднюю долю и ножку гипофиза с клинической картиной несахарного диабета.

В приведенном клиническом случае описывается развитие гипофизита у беременной пациентки с преимущественным поражением задней доли гипофиза и исходом в несахарный диабет, сохраняющимся через 6 лет после беременности и родов.

В статье рассмотрены аспекты дифференциальной диагностики несахарного диабета у беременных, а также особенности инструментальной диагностики и подходов к лечению гипофизита.

Autoimmune/lymphocytic hypophysitis is one of the rare causes of central diabetes insipidus in adults and is most common among women in the second or third trimester of pregnancy. Numerous studies have shown that lymphocytic hypophysitis is characterized by a very variable clinical signs with the development of neurological symptoms, visual disturbances and hypopituitarism with partial or complete loss of pituitary function, as well as a number of features in magnetic resonance imaging (MRI). Isolated lymphocytic indibuloneurohypophysitis occurs in fewer cases and involves the posterior lobe and stalk of the pituitary gland with a clinical presentation of diabetes insipidus. The above clinical case describes the development of hypophysitis in a pregnant woman with a predominant lesion of the posterior pituitary gland and an outcome in diabetes insipidus, which persists 6 years after pregnancy and childbirth. In the article some aspects of the differential diagnosis of diabetes insipidus in pregnant women, as well as instrumental diagnosis and treatment approaches of hypophysitis are discussed.

центральный несахарный диабетбеременностьлимфоцитарный гипофизитcentral diabetes insipiduspregnancylymphocytic hypophysitisОбследование и лечение пациентки осуществлялось за счет средств, выделенных по Гранту №121030100034-1 «Эндокринно-опосредованные нарушения осмотического гомеостаза: изучение этиологических и патогенетических факторов, разработка персонализированных подходов дифференциальной диагностики»ReferencesAnanthakrishnan S. Gestational diabetes insipidus: Diagnosis and management. Best Pract Res Clin Endocrinol Metab. 2020;34(5):101384. doi: https://doi.org/10.1016/j.beem.2020.101384Ananthakrishnan S. Gestational diabetes insipidus: Diagnosis and management. Best Pract Res Clin Endocrinol Metab. 2020;34(5):101384. doi:10.1016/j.beem.2020.101384Takeda R, Demura M, Sugimura Y, Miyamori I, Konoshita T, Yamamoto H. Pregnancy-associated diabetes insipidus in japan — a review based on quoting from the literatures reported during the period from 1982 to 2019. Endocr J. 2021;68(4):375-385. doi: https://doi.org/10.1507/endocrj.EJ20-0745Takeda R, Demura M, Sugimura Y, Miyamori I, Konoshita T, Yamamoto H. Pregnancy-associated diabetes insipidus in japan—a review based on quoting from the literatures reported during the period from 1982 to 2019. Endocr J. 2021;68(4):375-385. doi:10.1507/endocrj.EJ20-0745Gubbi S, Hannah-Shmouni F, Stratakis CA, Koch CA. Primary hypophysitis and other autoimmune disorders of the sellar and suprasellar regions. Rev Endocr Metab Disord. 2018;19(4):335-347. doi: https://doi.org/10.1007/s11154-018-9480-1Gubbi S, Hannah-Shmouni F, Stratakis CA, Koch CA. Primary hypophysitis and other autoimmune disorders of the sellar and suprasellar regions. Rev Endocr Metab Disord. 2018;19(4):335-347. doi:10.1007/s11154-018-9480-1Case A, Teratoid A. Neuroendocrinology and Pituitary Neuroendocrinology and Pituitary. 2020;4(May):639-640. doi: https://doi.org/10.1210/jendso/bvab048Case A, Teratoid A. Neuroendocrinology and Pituitary Neuroendocrinology and Pituitary. 2020;4(May):639-640. doi:10.1210/jendso/bvab048Benabderrahim K. Autoimmune hypophysitis and viral infection in a pregnant woman: A challengeable case. Pan Afr Med J. 2020;36:1-5. doi: https://doi.org/10.11604/pamj.2020.36.28.22454Benabderrahim K. Autoimmune hypophysitis and viral infection in a pregnant woman: A challengeable case. Pan Afr Med J. 2020;36:1-5. doi:10.11604/pamj.2020.36.28.22454Diego E, Ciudin A, Simo-Servat O, Ortiz A, Gutierrez-Carrasquilla L, Mesa J. A Case Report of Lymphocytic Hypophysitis Related to Pregnancy. Open J Endocr Metab Dis. 2015;05(12):171-176. doi: https://doi.org/10.4236/ojemd.2015.512021Diego E, Ciudin A, Simo-Servat O, Ortiz A, Gutierrez-Carrasquilla L, Mesa J. A Case Report of Lymphocytic Hypophysitis Related to Pregnancy. Open J Endocr Metab Dis. 2015;05(12):171-176. doi:10.4236/ojemd.2015.512021Yang JW, Duda B, Wo BL, Bédard MJ, Lavoie HB, Godbout A. A Rare Case of Severe Headache and Sudden-Onset Diabetes Insipidus During Pregnancy: Differential Diagnosis and Management of Lymphocytic Hypophysitis. AACE Clin Case Reports. 2016;2(1):30-35. doi: https://doi.org/10.4158/EP14590.CRYang JW, Duda B, Wo BL, Bédard MJ, Lavoie HB, Godbout A. A Rare Case of Severe Headache and Sudden-Onset Diabetes Insipidus During Pregnancy: Differential Diagnosis and Management of Lymphocytic Hypophysitis. AACE Clin Case Reports. 2016;2(1):e30-e35. doi:10.4158/EP14590.CRBiswas M, Thackare H, Jones MK, Bowen-Simpkins P. Lymphocytic hypophysitis and headache in pregnancy. BJOG An Int J Obstet Gynaecol. 2002;109(10):1184-1186. doi: https://doi.org/10.1016/S1470-0328(02)01658-0Biswas M, Thackare H, Jones MK, Bowen-Simpkins P. Lymphocytic hypophysitis and headache in pregnancy. BJOG An Int J Obstet Gynaecol. 2002;109(10):1184-1186. doi:10.1016/S1470-0328(02)01658-0Kuntal S, Manjambigai S. Lymphocytic hypophysitis in pregnancy leading to diabetes insipidus a rare disease and a diagnostic challenge. Eur J Obstet Gynecol Reprod Biol. 2016;206(2016):66. doi: https://doi.org/10.1016/j.ejogrb.2016.07.185Kuntal S, Manjambigai S. Lymphocytic hypophysitis in pregnancy leading to diabetes insipidus a rare disease and a diagnostic challenge. Eur J Obstet Gynecol Reprod Biol. 2016;206(2016):e66. doi:10.1016/j.ejogrb.2016.07.185Bellastella G, Bizzarro A, Aitella E, et al. Pregnancy may favour the development of severe autoimmune central diabetes insipidus in women with vasopressin cell antibodies: Description of two cases. Eur J Endocrinol. 2015;172(3):11-17. doi: https://doi.org/10.1530/EJE-14-0762Bellastella G, Bizzarro A, Aitella E, et al. Pregnancy may favour the development of severe autoimmune central diabetes insipidus in women with vasopressin cell antibodies: Description of two cases. Eur J Endocrinol. 2015;172(3):K11-K17. doi:10.1530/EJE-14-0762Blotner H, Kunkel P. Diabetes Insipidus and pregnancy: report of two cases. N Engl J Med. 1942;227:287e92Blotner H, Kunkel P. Diabetes Insipidus and pregnancy: report of two cases. N Engl J Med 1942;227:287e92Sainz Bueno JA, Villarejo Ortíz P, Hidalgo Amat J, Caballero Fernández V, Caballero Manzano M, Garrido Teruel R. Transient diabetes insipidus during pregnancy: A clinical case and a review of the syndrome. Eur J Obstet Gynecol Reprod Biol. 2005;118(2):251-254. doi: https://doi.org/10.1016/j.ejogrb.2004.04.036Sainz Bueno JA, Villarejo Ortíz P, Hidalgo Amat J, Caballero Fernández V, Caballero Manzano M, Garrido Teruel R. Transient diabetes insipidus during pregnancy: A clinical case and a review of the syndrome. Eur J Obstet Gynecol Reprod Biol. 2005;118(2):251-254. doi:10.1016/j.ejogrb.2004.04.036Bellastella A, Bizzarro A, Colella C, Bellastella G, Sinisi AA, De Bellis A. Subclinical diabetes insipidus. Best Pract Res Clin Endocrinol Metab. 2012;26(4):471-483. doi: https://doi.org/10.1016/j.beem.2011.11.008Bellastella A, Bizzarro A, Colella C, Bellastella G, Sinisi AA, De Bellis A. Subclinical diabetes insipidus. Best Pract Res Clin Endocrinol Metab. 2012;26(4):471-483. doi:10.1016/j.beem.2011.11.008Durr JA, Hoggard JG, Hunt JM, Schrier RW. Diabetes Insipidus in Pregnancy Associated with Abnormally High Circulating Vasopressinase Activity. N Engl J Med. 1987;316(17):1070-1074. doi: https://doi.org/10.1056/nejm198704233161707Durr JA, Hoggard JG, Hunt JM, Schrier RW. Diabetes Insipidus in Pregnancy Associated with Abnormally High Circulating Vasopressinase Activity. N Engl J Med. 1987;316(17):1070-1074. doi:10.1056/nejm198704233161707Проект клинических рекомендаций «Несахарный диабет», Российская ассоциация эндокринологов, 2022.Проект клинических рекомендаций «Несахарный диабет», Российская ассоциация эндокринологов, 2022.Dedov II, Mel’nichenko GA, Pigarova EA, et al. Federal clinical guidelines on diagnosis and treatment of diabetes insipidus in adults. Obe Metab. 2018;15(2):56-71. doi: https://doi.org/10.14341/OMET9670Dedov II, Mel’nichenko GA, Pigarova EA, et al. Federal clinical guidelines on diagnosis and treatment of diabetes insipidus in adults. Obe Metab. 2018;15(2):56-71. doi:10.14341/OMET9670Fenske W, Allolio B. Current state and future perspectives in the diagnosis of diabetes insipidus: A clinical review. J Clin Endocrinol Metab. 2012;97(10):3426-3437. doi: https://doi.org/10.1210/jc.2012-1981Fenske W, Allolio B. Current state and future perspectives in the diagnosis of diabetes insipidus: A clinical review. J Clin Endocrinol Metab. 2012;97(10):3426-3437. doi:10.1210/jc.2012-1981Briet JW. Diabetes insipidus, Sheehan’s syndrome and pregnancy. Eur J Obstet Gynecol Reprod Biol. 1998;77(2):201-203. doi: https://doi.org/10.1016/S0301-2115(97)00272-8Briet JW. Diabetes insipidus, Sheehan’s syndrome and pregnancy. Eur J Obstet Gynecol Reprod Biol. 1998;77(2):201-203. doi:10.1016/S0301-2115(97)00272-8Babey M, Kopp P, Robertson GL. Familial forms of diabetes insipidus: Clinical and molecular characteristics. Nat Rev Endocrinol. 2011;7(12):701-714. doi: https://doi.org/10.1038/nrendo.2011.100Babey M, Kopp P, Robertson GL. Familial forms of diabetes insipidus: Clinical and molecular characteristics. Nat Rev Endocrinol. 2011;7(12):701-714. doi:10.1038/nrendo.2011.100Araújo AN, Cunha M, Marques T, Bugalho MJGM. Multifactorial diabetes insipidus during pregnancy: A challenging diagnosis. BMJ Case Rep. 2021;14(3):2020-2022. doi: https://doi.org/10.1136/bcr-2020-238410Araújo AN, Cunha M, Marques T, Bugalho MJGM. Multifactorial diabetes insipidus during pregnancy: A challenging diagnosis. BMJ Case Rep. 2021;14(3):2020-2022. doi:10.1136/bcr-2020-238410Kluczyński Ł, Gilis-Januszewska A, Rogoziński D, Pantofliński J, Hubalewska-Dydejczyk A. Hypophysitis — new insights into diagnosis and treatment. Endokrynol Pol. 2019;70(3):260-269. doi: https://doi.org/10.5603/ep.a2019.0015Kluczyński Ł, Gilis-Januszewska A, Rogoziński D, Pantofliński J, Hubalewska-Dydejczyk A. Hypophysitis - new insights into diagnosis and treatment. Endokrynol Pol. 2019;70(3):260-269. doi:10.5603/ep.a2019.0015Vorontsov AV, Babaeva DM, Vladimirova VP, et al. Clinical and Radiological Diagnosis of Hypophysitis: a Review of Literature and Own Data. Probl Endokrinol (Mosk). 2022;68(2):16-33. doi: https://doi.org/10.14341/probl12777Vorontsov A V., Babaeva DM, Vladimirova VP, et al. Clinical and Radiological Diagnosis of Hypophysitis: a Review of Literature and Own Data. Probl Endokrinol (Mosk). 2022;68(2):16-33. doi:10.14341/probl12777Woodmansee WW. Pituitary Disorders in Pregnancy. Neurol Clin. 2019;37(1):63-83. doi: https://doi.org/10.1016/j.ncl.2018.09.009Woodmansee WW. Pituitary Disorders in Pregnancy. Neurol Clin. 2019;37(1):63-83. doi:10.1016/j.ncl.2018.09.009Bellastella G, Maiorino MI, Bizzarro A, et al. Revisitation of autoimmune hypophysitis: knowledge and uncertainties on pathophysiological and clinical aspects. Pituitary. 2016;19(6):625-642. doi: https://doi.org/10.1007/s11102-016-0736-zBellastella G, Maiorino MI, Bizzarro A, et al. Revisitation of autoimmune hypophysitis: knowledge and uncertainties on pathophysiological and clinical aspects. Pituitary. 2016;19(6):625-642. doi:10.1007/s11102-016-0736-zCaturegli P, Newschaffer C, Olivi A, Pomper MG, Burger PC, Rose NR. Autoimmune hypophysitis. Endocr Rev. 2005;26(5):599-614. doi: https://doi.org/10.1210/er.2004-0011Caturegli P, Newschaffer C, Olivi A, Pomper MG, Burger PC, Rose NR. Autoimmune hypophysitis. Endocr Rev. 2005;26(5):599-614. doi:10.1210/er.2004-0011Thodou E, Asa SL, Kontogeorgos G, Kovacs K, Horvath E, Ezzat S. Clinical case seminar: lymphocytic hypophysitis: clinicopathological findings. J Clin Endocrinol Metab. 1995;80(8):2302-2311. doi: https://doi.org/10.1210/jcem.80.8.762922327Thodou E, Asa SL, Kontogeorgos G, Kovacs K, Horvath E, Ezzat S. Clinical case seminar: lymphocytic hypophysitis: clinicopathological findings. J Clin Endocrinol Metab. 1995;80(8):2302-2311. doi:10.1210/jcem.80.8.762922327.Caturegli P, Lupi I, Landek-Salgado M, Kimura H, Rose NR. Pituitary autoimmunity: 30 years later. Autoimmun Rev. 2008;7(8):631-637. doi: https://doi.org/10.1016/j.autrev.2008.04.016Caturegli P, Lupi I, Landek-Salgado M, Kimura H, Rose NR. Pituitary autoimmunity: 30 years later. Autoimmun Rev. 2008;7(8):631-637. doi:10.1016/j.autrev.2008.04.016Zainudin S, Hea TB, Laporte J-P, Fang SC. Infundibuloneurohypophysitis as a Cause of Central Diabetes Insipidus in an Indian Woman Two Years Postpartum. J ASEAN Fed Endocr Soc. 2012;27(1):100-104. doi: https://doi.org/10.15605/jafes.027.01.17Zainudin, S., of Endocrinology, D., Hospital, S. G., Hea, T. B., Laporte, J.-P., & Fang, S. C. (2012). Infundibuloneurohypophysitis as a Cause of Central Diabetes Insipidus in an Indian Woman Two Years Postpartum. Journal of the ASEAN Federation of Endocrine Societies, 27(1), 100–104. https://doi.org/10.15605/JAFES.027.01.17Gutenberg A, Larsen J, Lupi I, Rohde V, Caturegli P. A radiologic score to distinguish autoimmune hypophysitis from nonsecreting pituitary adenoma preoperatively. Am J Neuroradiol. 2009;30(9):1766-1772. doi: https://doi.org/10.3174/ajnr.A1714Gutenberg A, Larsen J, Lupi I, Rohde V, Caturegli P. A radiologic score to distinguish autoimmune hypophysitis from nonsecreting pituitary adenoma preoperatively. Am J Neuroradiol. 2009;30(9):1766-1772. doi:10.3174/ajnr.A1714Leggett DAC, Hill PT, Anderson RJ. “Stalkitis” in a pregnant 32-year-old woman: A rare cause of diabetes insipidus. Australas Radiol. 1999;43(1):104-107. doi: https://doi.org/10.1046/j.1440-1673.1999.00597.xLeggett DAC, Hill PT, Anderson RJ. “Stalkitis” in a pregnant 32-year-old woman: A rare cause of diabetes insipidus. Australas Radiol. 1999;43(1):104-107. doi:10.1046/j.1440-1673.1999.00597.xIwama S, Sugimura Y, Kiyota A, et al. Rabphilin-3A as a targeted autoantigen in lymphocytic infundibulo-neurohypophysitis. J Clin Endocrinol Metab. 2015;100(7):E946-E954. doi: https://doi.org/10.1210/jc.2014-4209Iwama S, Sugimura Y, Kiyota A, et al. Rabphilin-3A as a targeted autoantigen in lymphocytic infundibulo-neurohypophysitis. J Clin Endocrinol Metab. 2015;100(7):E946-E954. doi:10.1210/jc.2014-4209Sakurai K, Yamashita R, Niituma S, et al. Usefulness of anti-rabphilin-3A antibodies for diagnosing central diabetes insipidus in the third trimester of pregnancy. Endocr J. 2017;64(6):645-650. doi: https://doi.org/10.1507/endocrj.EJ17-0028Sakurai K, Yamashita R, Niituma S, et al. Usefulness of anti-rabphilin-3A antibodies for diagnosing central diabetes insipidus in the third trimester of pregnancy. Endocr J. 2017;64(6):645-650. doi:10.1507/endocrj.EJ17-0028Park SM, Bae JC, Joung JY, et al. Clinical characteristics, management, and outcome of 22 cases of primary hypophysitis. Endocrinol Metab. 2014;29(4):470-478. doi: https://doi.org/10.3803/EnM.2014.29.4.470Park SM, Bae JC, Joung JY, et al. Clinical characteristics, management, and outcome of 22 cases of primary hypophysitis. Endocrinol Metab. 2014;29(4):470-478. doi:10.3803/EnM.2014.29.4.470Kristof RA, Van Roost D, Klingmüller D, Springer W, Schramm J. Lymphocytic hypophysitis: Non-invasive diagnosis and treatment by high dose methylprednisolone pulse therapy? J Neurol Neurosurg Psychiatry. 1999;67(3):398-402. doi: https://doi.org/10.1136/jnnp.67.3.398Kristof RA, Van Roost D, Klingmüller D, Springer W, Schramm J. Lymphocytic hypophysitis: Non-invasive diagnosis and treatment by high dose methylprednisolone pulse therapy? J Neurol Neurosurg Psychiatry. 1999;67(3):398-402. doi:10.1136/jnnp.67.3.398Angelousi A, Cohen C, Sosa S, et al. Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis. Horm Metab Res. 2018;50(4):296-302. doi: https://doi.org/10.1055/s-0044-101036Angelousi A, Cohen C, Sosa S, et al. Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis. Horm Metab Res. 2018;50(4):296-302. doi:10.1055/s-0044-101036Honegger J, Buchfelder M, Schlaffer S, et al. Treatment of primary hypophysitis in Germany. J Clin Endocrinol Metab. 2015;100(9):3460-3469. doi: https://doi.org/10.1210/jc.2015-2146Honegger J, Buchfelder M, Schlaffer S, et al. Treatment of primary hypophysitis in Germany. J Clin Endocrinol Metab. 2015;100(9):3460-3469. doi:10.1210/jc.2015-2146

The authors declare that there are no conflicts of interest present.