Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review

PurposeXanthomatous hypophysitis (XHP) is an extremely rare form of primary hypophysitis for which there is a lack of clinical experience. A comprehensive understanding of its clinical characteristics, diagnosis and treatment is needed.MethodsHere, we report a case study and conduct a systematic rev...

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Autores principales:	Jianyu Zhu, Zhicheng Wang, Wenze Wang, Jinghua Fan, Yi Zhang, Xiaoxu Li, Jie Liu, Shenzhong Jiang, Kan Deng, Lian Duan, Yong Yao, Huijuan Zhu
Formato:	article
Lenguaje:	EN
Publicado:	Frontiers Media S.A. 2021
Materias:	xanthomatous hypophysitis clinical characteristics pathological examination surgery recurrence Diseases of the endocrine glands. Clinical endocrinology RC648-665
Acceso en línea:	https://doaj.org/article/718296eea8d04257abbe0c66dc2c0e1c
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spelling	oai:doaj.org-article:718296eea8d04257abbe0c66dc2c0e1c2021-11-09T10:15:14ZXanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review1664-239210.3389/fendo.2021.735655https://doaj.org/article/718296eea8d04257abbe0c66dc2c0e1c2021-10-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fendo.2021.735655/fullhttps://doaj.org/toc/1664-2392PurposeXanthomatous hypophysitis (XHP) is an extremely rare form of primary hypophysitis for which there is a lack of clinical experience. A comprehensive understanding of its clinical characteristics, diagnosis and treatment is needed.MethodsHere, we report a case study and conduct a systematic review of XHP. Thirty-six cases were included, and their clinical manifestations, endocrine assessment, imaging features, treatment and follow-up data were collected and analyzed.ResultsThe mean age at diagnosis was 39.1 years, and females were predominant (75.0%). The most common symptom was headache (68.6%), and 66.7% of female patients presented menstrual disorders. The most common pituitary dysfunction was growth hormone (GH) deficiency. More than half of patients exhibited central diabetes insipidus (CDI). The majority of patients had an imaging presentation of a cystic lesion with peripheral enhancement. Pituitary stalk thickening was observed in half of the patients. Total lesion resection was achieved in 57.1% of cases. The recurrence rate after partial resection and biopsy was significantly higher than that after total lesion resection (57.1% vs. 0.0%, P = 0.0147). The most common pituitary hormone abnormalities to resolve after surgery were hyperprolactinemia (100.0%) and GH deficiency (91.7%). The typical pathological feature was inflammatory infiltration of foamy histiocytes, which showed positivity for CD68.ConclusionDiagnosis of XHP is difficult when relying on clinical symptoms and imaging features. Therefore, surgical histopathology is necessary. Based on the available evidence, total lesion resection is recommended for treatment. However, the long-term prognosis for this rare disease remains unclear.Jianyu ZhuJianyu ZhuZhicheng WangWenze WangJinghua FanYi ZhangXiaoxu LiJie LiuShenzhong JiangKan DengLian DuanYong YaoHuijuan ZhuFrontiers Media S.A.articlexanthomatous hypophysitisclinical characteristicspathological examinationsurgeryrecurrenceDiseases of the endocrine glands. Clinical endocrinologyRC648-665ENFrontiers in Endocrinology, Vol 12 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	xanthomatous hypophysitis clinical characteristics pathological examination surgery recurrence Diseases of the endocrine glands. Clinical endocrinology RC648-665
spellingShingle	xanthomatous hypophysitis clinical characteristics pathological examination surgery recurrence Diseases of the endocrine glands. Clinical endocrinology RC648-665 Jianyu Zhu Jianyu Zhu Zhicheng Wang Wenze Wang Jinghua Fan Yi Zhang Xiaoxu Li Jie Liu Shenzhong Jiang Kan Deng Lian Duan Yong Yao Huijuan Zhu Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
description	PurposeXanthomatous hypophysitis (XHP) is an extremely rare form of primary hypophysitis for which there is a lack of clinical experience. A comprehensive understanding of its clinical characteristics, diagnosis and treatment is needed.MethodsHere, we report a case study and conduct a systematic review of XHP. Thirty-six cases were included, and their clinical manifestations, endocrine assessment, imaging features, treatment and follow-up data were collected and analyzed.ResultsThe mean age at diagnosis was 39.1 years, and females were predominant (75.0%). The most common symptom was headache (68.6%), and 66.7% of female patients presented menstrual disorders. The most common pituitary dysfunction was growth hormone (GH) deficiency. More than half of patients exhibited central diabetes insipidus (CDI). The majority of patients had an imaging presentation of a cystic lesion with peripheral enhancement. Pituitary stalk thickening was observed in half of the patients. Total lesion resection was achieved in 57.1% of cases. The recurrence rate after partial resection and biopsy was significantly higher than that after total lesion resection (57.1% vs. 0.0%, P = 0.0147). The most common pituitary hormone abnormalities to resolve after surgery were hyperprolactinemia (100.0%) and GH deficiency (91.7%). The typical pathological feature was inflammatory infiltration of foamy histiocytes, which showed positivity for CD68.ConclusionDiagnosis of XHP is difficult when relying on clinical symptoms and imaging features. Therefore, surgical histopathology is necessary. Based on the available evidence, total lesion resection is recommended for treatment. However, the long-term prognosis for this rare disease remains unclear.
format	article
author	Jianyu Zhu Jianyu Zhu Zhicheng Wang Wenze Wang Jinghua Fan Yi Zhang Xiaoxu Li Jie Liu Shenzhong Jiang Kan Deng Lian Duan Yong Yao Huijuan Zhu
author_facet	Jianyu Zhu Jianyu Zhu Zhicheng Wang Wenze Wang Jinghua Fan Yi Zhang Xiaoxu Li Jie Liu Shenzhong Jiang Kan Deng Lian Duan Yong Yao Huijuan Zhu
author_sort	Jianyu Zhu
title	Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
title_short	Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
title_full	Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
title_fullStr	Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
title_full_unstemmed	Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
title_sort	xanthomatous hypophysitis: a case report and comprehensive literature review
publisher	Frontiers Media S.A.
publishDate	2021
url	https://doaj.org/article/718296eea8d04257abbe0c66dc2c0e1c
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Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review

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