Hypothalamitis : A Novel Autoimmune Endocrine Disease. A Literature Review and Case Report
© The Author(s) 2020. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissionsoup.com..
CONTEXT: The relationship between the endocrine system and autoimmunity has been recognized for a long time and one of the best examples of autoimmune endocrine disease is autoimmune hypophysitis. A better understanding of autoimmune mechanisms and radiological, biochemical, and immunological developments has given rise to the definition of new autoimmune disorders including autoimmunity-related hypothalamic-pituitary disorders. However, whether hypothalamitis may occur as a distinct entity is still a matter of debate.
EVIDENCE ACQUISITION: Here we describe a 35-year-old woman with growing suprasellar mass, partial empty sella, central diabetes insipidus, hypopituitarism, and hyperprolactinemia.
EVIDENCE SYNTHESIS: Histopathologic examination of surgically removed suprasellar mass revealed lymphocytic infiltrate suggestive of an autoimmune disease with hypothalamic involvement. The presence of antihypothalamus antibodies to arginine vasopressin (AVP)-secreting cells (AVPcAb) at high titers and the absence of antipituitary antibodies suggested the diagnosis of isolated hypothalamitis. Some similar conditions have sometimes been reported in the literature but the simultaneous double finding of lymphocytic infiltrate and the presence of AVPcAb so far has never been reported.
CONCLUSIONS: We think that the hypothalamitis can be considered a new isolated autoimmune disease affecting the hypothalamus while the lymphocytic infundibuloneurohypophysitis can be a consequence of hypothalamitis with subsequent autoimmune involvement of the pituitary. To our knowledge this is the first observation of autoimmune hypothalamic involvement with central diabetes insipidus, partial empty sella, antihypothalamic antibodies and hypopituitarism.
| Medienart: |
E-Artikel |
|---|
| Erscheinungsjahr: |
2021 |
|---|---|
| Erschienen: |
2021 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:106 |
|---|---|
| Enthalten in: |
The Journal of clinical endocrinology and metabolism - 106(2021), 2 vom: 23. Jan., Seite e415-e429 |
| Sprache: |
Englisch |
|---|
| Beteiligte Personen: |
Türe, Uğur [VerfasserIn] |
|---|
| Links: |
|---|
| Themen: |
Case Reports |
|---|
| Anmerkungen: |
Date Completed 08.09.2021 Date Revised 08.09.2021 published: Print Citation Status MEDLINE |
|---|
| doi: |
10.1210/clinem/dgaa771 |
|---|
| funding: |
|
|---|---|
| Förderinstitution / Projekttitel: |
|
| PPN (Katalog-ID): |
NLM316735590 |
|---|
| LEADER | 01000naa a22002652 4500 | ||
|---|---|---|---|
| 001 | NLM316735590 | ||
| 003 | DE-627 | ||
| 005 | 20231225161726.0 | ||
| 007 | cr uuu---uuuuu | ||
| 008 | 231225s2021 xx |||||o 00| ||eng c | ||
| 024 | 7 | |a 10.1210/clinem/dgaa771 |2 doi | |
| 028 | 5 | 2 | |a pubmed24n1055.xml |
| 035 | |a (DE-627)NLM316735590 | ||
| 035 | |a (NLM)33104773 | ||
| 040 | |a DE-627 |b ger |c DE-627 |e rakwb | ||
| 041 | |a eng | ||
| 100 | 1 | |a Türe, Uğur |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Hypothalamitis |b A Novel Autoimmune Endocrine Disease. A Literature Review and Case Report |
| 264 | 1 | |c 2021 | |
| 336 | |a Text |b txt |2 rdacontent | ||
| 337 | |a ƒaComputermedien |b c |2 rdamedia | ||
| 338 | |a ƒa Online-Ressource |b cr |2 rdacarrier | ||
| 500 | |a Date Completed 08.09.2021 | ||
| 500 | |a Date Revised 08.09.2021 | ||
| 500 | |a published: Print | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © The Author(s) 2020. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissionsoup.com. | ||
| 520 | |a CONTEXT: The relationship between the endocrine system and autoimmunity has been recognized for a long time and one of the best examples of autoimmune endocrine disease is autoimmune hypophysitis. A better understanding of autoimmune mechanisms and radiological, biochemical, and immunological developments has given rise to the definition of new autoimmune disorders including autoimmunity-related hypothalamic-pituitary disorders. However, whether hypothalamitis may occur as a distinct entity is still a matter of debate | ||
| 520 | |a EVIDENCE ACQUISITION: Here we describe a 35-year-old woman with growing suprasellar mass, partial empty sella, central diabetes insipidus, hypopituitarism, and hyperprolactinemia | ||
| 520 | |a EVIDENCE SYNTHESIS: Histopathologic examination of surgically removed suprasellar mass revealed lymphocytic infiltrate suggestive of an autoimmune disease with hypothalamic involvement. The presence of antihypothalamus antibodies to arginine vasopressin (AVP)-secreting cells (AVPcAb) at high titers and the absence of antipituitary antibodies suggested the diagnosis of isolated hypothalamitis. Some similar conditions have sometimes been reported in the literature but the simultaneous double finding of lymphocytic infiltrate and the presence of AVPcAb so far has never been reported | ||
| 520 | |a CONCLUSIONS: We think that the hypothalamitis can be considered a new isolated autoimmune disease affecting the hypothalamus while the lymphocytic infundibuloneurohypophysitis can be a consequence of hypothalamitis with subsequent autoimmune involvement of the pituitary. To our knowledge this is the first observation of autoimmune hypothalamic involvement with central diabetes insipidus, partial empty sella, antihypothalamic antibodies and hypopituitarism | ||
| 650 | 4 | |a Case Reports | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Review | |
| 650 | 4 | |a diabetes insipidus | |
| 650 | 4 | |a hypophysitis | |
| 650 | 4 | |a hypopituitarism | |
| 650 | 4 | |a hypothalamitis | |
| 650 | 4 | |a pituitary | |
| 700 | 1 | |a De Bellis, Annamaria |e verfasserin |4 aut | |
| 700 | 1 | |a Harput, Mehmet Volkan |e verfasserin |4 aut | |
| 700 | 1 | |a Bellastella, Giuseppe |e verfasserin |4 aut | |
| 700 | 1 | |a Topcuoglu, Melih |e verfasserin |4 aut | |
| 700 | 1 | |a Yaltirik, Cumhur Kaan |e verfasserin |4 aut | |
| 700 | 1 | |a Cirillo, Paolo |e verfasserin |4 aut | |
| 700 | 1 | |a Yola, Rima Nur |e verfasserin |4 aut | |
| 700 | 1 | |a Sav, Aydın |e verfasserin |4 aut | |
| 700 | 1 | |a Kelestimur, Fahrettin |e verfasserin |4 aut | |
| 773 | 0 | 8 | |i Enthalten in |t The Journal of clinical endocrinology and metabolism |d 1945 |g 106(2021), 2 vom: 23. Jan., Seite e415-e429 |w (DE-627)NLM00001821X |x 1945-7197 |7 nnns |
| 773 | 1 | 8 | |g volume:106 |g year:2021 |g number:2 |g day:23 |g month:01 |g pages:e415-e429 |
| 856 | 4 | 0 | |u http://dx.doi.org/10.1210/clinem/dgaa771 |3 Volltext |
| 912 | |a GBV_USEFLAG_A | ||
| 912 | |a GBV_NLM | ||
| 951 | |a AR | ||
| 952 | |d 106 |j 2021 |e 2 |b 23 |c 01 |h e415-e429 | ||