Clinical Features, Magnetic Resonance Imaging, and Treatment Experience of 20 Patients with Lymphocytic Hypophysitis in a Single Center
Lymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and focus on the management and prognosis of LYH. A retrospective study of patients with LYH was conducted between 2011 and 2018 at a single inst...
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| Vydané v: | World neurosurgery Ročník 127; s. e22 - e29 |
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| Hlavní autori: | , , , , , , |
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| Jazyk: | English |
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United States
Elsevier Inc
01.07.2019
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| Abstract | Lymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and focus on the management and prognosis of LYH.
A retrospective study of patients with LYH was conducted between 2011 and 2018 at a single institute. The patients were included by pathologic conformation and strict exclusion criteria. Clinical profile, imaging, and management data were collected.
Twenty patients with LYH (16 women and 4 men) were included. Ten patients were diagnosed histologically and the remaining 10 patients were confirmed clinically of exclusion criteria. The median age at diagnosis was 37 years (range, 16–58 years). Presenting symptoms were followed by polyuria/polydipsia (11, 55%), vision changes (10, 50%), headache (8, 40%), menstrual irregularities and amenorrhea (4, 20%), diplopia (1, 5%), or sexual dysfunction (1, 5%). Eight patients had partial anterior pituitary hormone dysfunction. The thyroid-stimulating hormone axis was most involved. Ten patients received transsphenoidal surgery, 5 patients experienced steroid pulse therapy, and observation was performed on 5 patients. Only 5 patients (25%) showed improvement of anterior pituitary dysfunction after initial management. Recovery of diabetes insipidus occurred in 2 patients (18%). The overall recurrence rate was 22.2%.
Nonoperative treatment is a better option for most patients with LYH because it is effective and noninvasive. Surgery is recommended for definitive diagnosis, severe or rapid progression of neurologic impairment, and glucocorticoid insensitivity. Periodic follow-up is mandatory in a patient's long-term management. |
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| AbstractList | Lymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and focus on the management and prognosis of LYH.
A retrospective study of patients with LYH was conducted between 2011 and 2018 at a single institute. The patients were included by pathologic conformation and strict exclusion criteria. Clinical profile, imaging, and management data were collected.
Twenty patients with LYH (16 women and 4 men) were included. Ten patients were diagnosed histologically and the remaining 10 patients were confirmed clinically of exclusion criteria. The median age at diagnosis was 37 years (range, 16–58 years). Presenting symptoms were followed by polyuria/polydipsia (11, 55%), vision changes (10, 50%), headache (8, 40%), menstrual irregularities and amenorrhea (4, 20%), diplopia (1, 5%), or sexual dysfunction (1, 5%). Eight patients had partial anterior pituitary hormone dysfunction. The thyroid-stimulating hormone axis was most involved. Ten patients received transsphenoidal surgery, 5 patients experienced steroid pulse therapy, and observation was performed on 5 patients. Only 5 patients (25%) showed improvement of anterior pituitary dysfunction after initial management. Recovery of diabetes insipidus occurred in 2 patients (18%). The overall recurrence rate was 22.2%.
Nonoperative treatment is a better option for most patients with LYH because it is effective and noninvasive. Surgery is recommended for definitive diagnosis, severe or rapid progression of neurologic impairment, and glucocorticoid insensitivity. Periodic follow-up is mandatory in a patient's long-term management. Lymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and focus on the management and prognosis of LYH.OBJECTIVELymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and focus on the management and prognosis of LYH.A retrospective study of patients with LYH was conducted between 2011 and 2018 at a single institute. The patients were included by pathologic conformation and strict exclusion criteria. Clinical profile, imaging, and management data were collected.METHODSA retrospective study of patients with LYH was conducted between 2011 and 2018 at a single institute. The patients were included by pathologic conformation and strict exclusion criteria. Clinical profile, imaging, and management data were collected.Twenty patients with LYH (16 women and 4 men) were included. Ten patients were diagnosed histologically and the remaining 10 patients were confirmed clinically of exclusion criteria. The median age at diagnosis was 37 years (range, 16-58 years). Presenting symptoms were followed by polyuria/polydipsia (11, 55%), vision changes (10, 50%), headache (8, 40%), menstrual irregularities and amenorrhea (4, 20%), diplopia (1, 5%), or sexual dysfunction (1, 5%). Eight patients had partial anterior pituitary hormone dysfunction. The thyroid-stimulating hormone axis was most involved. Ten patients received transsphenoidal surgery, 5 patients experienced steroid pulse therapy, and observation was performed on 5 patients. Only 5 patients (25%) showed improvement of anterior pituitary dysfunction after initial management. Recovery of diabetes insipidus occurred in 2 patients (18%). The overall recurrence rate was 22.2%.RESULTSTwenty patients with LYH (16 women and 4 men) were included. Ten patients were diagnosed histologically and the remaining 10 patients were confirmed clinically of exclusion criteria. The median age at diagnosis was 37 years (range, 16-58 years). Presenting symptoms were followed by polyuria/polydipsia (11, 55%), vision changes (10, 50%), headache (8, 40%), menstrual irregularities and amenorrhea (4, 20%), diplopia (1, 5%), or sexual dysfunction (1, 5%). Eight patients had partial anterior pituitary hormone dysfunction. The thyroid-stimulating hormone axis was most involved. Ten patients received transsphenoidal surgery, 5 patients experienced steroid pulse therapy, and observation was performed on 5 patients. Only 5 patients (25%) showed improvement of anterior pituitary dysfunction after initial management. Recovery of diabetes insipidus occurred in 2 patients (18%). The overall recurrence rate was 22.2%.Nonoperative treatment is a better option for most patients with LYH because it is effective and noninvasive. Surgery is recommended for definitive diagnosis, severe or rapid progression of neurologic impairment, and glucocorticoid insensitivity. Periodic follow-up is mandatory in a patient's long-term management.CONCLUSIONSNonoperative treatment is a better option for most patients with LYH because it is effective and noninvasive. Surgery is recommended for definitive diagnosis, severe or rapid progression of neurologic impairment, and glucocorticoid insensitivity. Periodic follow-up is mandatory in a patient's long-term management. |
| Author | Jia, Guijun Lv, Gang Wang, Jisheng Qian, Ke Zhu, Qiang Zhong, Liyong Yu, Shuqing |
| Author_xml | – sequence: 1 givenname: Qiang surname: Zhu fullname: Zhu, Qiang organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 2 givenname: Ke surname: Qian fullname: Qian, Ke organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 3 givenname: Guijun surname: Jia fullname: Jia, Guijun organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 4 givenname: Gang surname: Lv fullname: Lv, Gang organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 5 givenname: Jisheng surname: Wang fullname: Wang, Jisheng organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 6 givenname: Liyong surname: Zhong fullname: Zhong, Liyong organization: Department of Endocrinology, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China – sequence: 7 givenname: Shuqing orcidid: 0000-0002-8253-1811 surname: Yu fullname: Yu, Shuqing email: yushuqingttyy@163.com organization: Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, People's Republic of China |
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| CitedBy_id | crossref_primary_10_1111_jne_12954 crossref_primary_10_1210_clinem_dgab839 crossref_primary_10_1016_j_beem_2019_101371 crossref_primary_10_1016_j_wneu_2022_12_108 crossref_primary_10_1016_j_eprac_2022_06_009 crossref_primary_10_12998_wjcc_v10_i3_1041 crossref_primary_10_7759_cureus_30178 crossref_primary_10_1136_bcr_2021_242353 crossref_primary_10_1007_s11102_022_01232_0 crossref_primary_10_3389_fnins_2021_808111 crossref_primary_10_1111_jne_12995 crossref_primary_10_7759_cureus_59396 |
| Cites_doi | 10.1111/cen.13354 10.1007/s11102-014-0622-5 10.1007/s11102-016-0769-3 10.3803/EnM.2014.29.4.470 10.1007/s11102-013-0550-9 10.1210/er.2004-0011 10.1210/jc.2015-2146 10.1016/S0140-6736(16)30053-8 10.1111/j.1365-2265.2009.03765.x 10.1016/j.beem.2004.11.007 10.1007/s00701-005-0641-0 10.1530/eje.1.02183 10.1016/j.autrev.2008.04.016 10.1530/EJE-17-0910 10.1507/endocrj.K10E-334 10.1002/path.1700830241 10.1097/00006123-199704000-00010 10.3174/ajnr.A1714 10.1210/jc.2015-2152 10.1007/s11102-010-0252-5 |
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| Keywords | GH DI MRI Diabetes insipidus Lymphocytic hypophysitis ACTH TSH Steroid pulse therapy IGF PA Stalk thickening APA TSS FSH LYH LH |
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| Snippet | Lymphocytic hypophysitis (LYH) is a rare autoimmune inflammatory disease of the pituitary gland. In this study, we aim to characterize LYH at presentation and... |
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| SubjectTerms | Adolescent Adult Autoimmune Hypophysitis - complications Autoimmune Hypophysitis - diagnostic imaging Autoimmune Hypophysitis - pathology Autoimmune Hypophysitis - therapy Combined Modality Therapy Diabetes insipidus Diabetes Insipidus - etiology Diagnosis, Differential Diplopia - etiology Female Follow-Up Studies Glucocorticoids - therapeutic use Hemianopsia - etiology Hormone Replacement Therapy Humans Hypophysectomy - methods Lymphocytic hypophysitis Magnetic Resonance Imaging Male Methylprednisolone - therapeutic use Middle Aged Neuroimaging Pituitary Diseases - etiology Pituitary Hormones - blood Pituitary Hormones - therapeutic use Retrospective Studies Stalk thickening Steroid pulse therapy Treatment Outcome Young Adult |
| Title | Clinical Features, Magnetic Resonance Imaging, and Treatment Experience of 20 Patients with Lymphocytic Hypophysitis in a Single Center |
| URI | https://www.clinicalkey.com/#!/content/1-s2.0-S1878875019303663 https://dx.doi.org/10.1016/j.wneu.2019.01.250 https://www.ncbi.nlm.nih.gov/pubmed/30790734 https://www.proquest.com/docview/2185565347 |
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