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乳突性垂体神经内分泌肿瘤/腺瘤与克罗恩病患者的肉芽肿性垂体炎相关:病例报告。

Lactotroph PitNET/adenoma associated to granulomatous hypophysitis in a patient with Crohn's disease: A case report.

机构信息

Pathology Unit, Department of Molecular and Translational Medicine, University of Brescia and Pathology Service Spedali Civili of Brescia, Brescia, Italy.

Neurosurgery, Department of Surgical Specialties, Radiological Sciences and Public Health, University of Brescia, Brescia, Italy.

出版信息

Neuropathology. 2023 Feb;43(1):104-109. doi: 10.1111/neup.12857. Epub 2022 Aug 10.

DOI:10.1111/neup.12857
PMID:35949102
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10087785/
Abstract

Granulomatous hypophysitis is a rare and poorly understood condition. Although certain cases are treated as primary pituitary autoimmune disorders, rare cases may be associated with pituitary neuroendocrine tumours (PitNETs) and systemic inflammatory diseases. Here, we report a case of a 47-year-old man that underwent endoscopic trans-sphenoidal excision of a pituitary mass diagnosed as PitNET. On histologic evaluation, the neoplasm showed an admixture of granulomas with extensive inflammatory infiltrate and lactotroph PitNET/adenoma. Careful anamnestic examination revealed a diagnosis of Crohn's disease 20 years prior. Although rarely done, both PitNET and Crohn's disease may be associated with granulomatous hypophysitis, and our patient had both conditions. During the 6-year follow-up, PitNETs and hypophysitis did not recur, while Crohn's disease was only partially controlled by medical therapy. To our knowledge, this is the first description of association of granulomatous hypophysitis, PitNET and Crohn's disease.

摘要

肉芽肿性垂体炎是一种罕见且尚未被充分认识的疾病。尽管某些病例被视为原发性垂体自身免疫性疾病,但罕见病例可能与垂体神经内分泌肿瘤(PitNET)和系统性炎症性疾病相关。在此,我们报告一例 47 岁男性患者,其接受了内镜经蝶窦切除垂体肿块的手术,该肿块被诊断为 PitNET。在组织学评估中,该肿瘤显示出混杂的肉芽肿,伴有广泛的炎症浸润和泌乳细胞型 PitNET/腺瘤。仔细的病史询问发现 20 年前患有克罗恩病的诊断。尽管很少见,但 PitNET 和克罗恩病都可能与肉芽肿性垂体炎相关,而我们的患者同时患有这两种疾病。在 6 年的随访中,PitNET 和垂体炎未再复发,而克罗恩病仅通过药物治疗部分得到控制。据我们所知,这是首例描述肉芽肿性垂体炎、PitNET 和克罗恩病之间关联的病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/598476785412/NEUP-43-104-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/4c1ef7f9d46e/NEUP-43-104-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/a77033c78b3f/NEUP-43-104-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/598476785412/NEUP-43-104-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/4c1ef7f9d46e/NEUP-43-104-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/a77033c78b3f/NEUP-43-104-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b5a1/10087785/598476785412/NEUP-43-104-g002.jpg

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本文引用的文献

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Granulomatous Sarcoidosis Mimics.肉芽肿性结节病的模仿者。
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A Remarkable Response of Granulomatous Hypophysitis to Infliximab in a Patient With a Background of Crohn's Disease-A Case Report.英夫利昔单抗治疗克罗恩病背景下的肉芽肿性垂体炎患者的显著疗效:病例报告。
Front Endocrinol (Lausanne). 2020 May 29;11:350. doi: 10.3389/fendo.2020.00350. eCollection 2020.
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垂体炎:新型垂体炎症性疾病的形式、诊断和治疗方法的最新进展。
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Intratumoral granulomatous reaction in recurrent pituitary adenoma: A unique presentation.复发性垂体腺瘤中的瘤内肉芽肿反应:一种独特的表现。
J Cancer Res Ther. 2017 Jul-Sep;13(3):580-582. doi: 10.4103/jcrt.JCRT_677_16.
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