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未经治疗的巨大泌乳素瘤伴慢性脑脊液漏:一种罕见的并发症。

Untreated Giant Macroprolactinoma with Chronic Cerebrospinal Fluid Leakage: An Unusual Complication.

作者信息

Mad Naser Mohamad Nazrulhisham, Aziz Nor Azizah, Karim Noor Khairiah A

机构信息

Department of Medicine, Hospital Pulau Pinang, Jalan Residensi, 10990 Georgetown, Pulau Pinang, Malaysia.

Department of Cardiology, Hospital Pulau Pinang, Jalan Residensi, 10990 Georgetown, Pulau Pinang, Malaysia.

出版信息

Case Rep Endocrinol. 2019 Jan 15;2019:4825357. doi: 10.1155/2019/4825357. eCollection 2019.

DOI:10.1155/2019/4825357
PMID:30766735
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6350591/
Abstract

Macroprolactinoma has the potential to cause base of skull erosion and often extends into the sphenoid sinus. Rapid shrinkage of this invasive tumor following dopamine agonist therapy has been postulated to cause unplugging of the eroded area, leading to cerebrospinal fluid leakage. To the best of our knowledge, the occurrence of spontaneous cerebrospinal fluid leak in treatment-naive prolactinomas is very rare, the majority of which involve undiagnosed macroprolactinomas. We describe here a lady presented late with giant macroprolactinoma, complicated by cerebrospinal fluid leakage. This case raised the dilemma in the management pertaining to the role of either pharmacotherapy or surgical intervention, or combination of both. As she strictly refused surgery, she was treated with bromocriptine which was later changed to cabergoline. On follow-up, there was cessation of cerebrospinal fluid leak, marked reduction of serum prolactin level, and imaging evidence of tumor shrinkage. The majority of patients with medically induced cerebrospinal fluid leakage will require surgical procedures to overcome this complication; however, there are isolated cases of leakage resolution on continuing dopamine agonist therapy while awaiting surgery. The use of dopamine agonist does not necessarily cause worsening of cerebrospinal fluid leakage and instead may produce spontaneous resolution as in this case.

摘要

大泌乳素瘤有可能导致颅底骨质侵蚀,且常延伸至蝶窦。多巴胺激动剂治疗后这种侵袭性肿瘤迅速缩小,据推测会使侵蚀区域堵塞解除,从而导致脑脊液漏。据我们所知,未经治疗的泌乳素瘤出现自发性脑脊液漏的情况非常罕见,其中大多数涉及未被诊断出的大泌乳素瘤。我们在此描述一位因巨大泌乳素瘤就诊较晚且并发脑脊液漏的女性患者。该病例引发了治疗上的两难困境,即药物治疗、手术干预或两者联合的作用。由于她坚决拒绝手术,遂接受溴隐亭治疗,后来改为卡麦角林治疗。随访时,脑脊液漏停止,血清泌乳素水平显著降低,且有肿瘤缩小的影像学证据。大多数因药物治疗导致脑脊液漏的患者需要手术来解决这一并发症;然而,也有个别病例在等待手术期间继续使用多巴胺激动剂治疗时漏液得到缓解。使用多巴胺激动剂不一定会导致脑脊液漏恶化,反而可能像本例一样使漏液自行缓解。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9143/6350591/daeba9575fa2/CRIE2019-4825357.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9143/6350591/d837128fa73f/CRIE2019-4825357.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9143/6350591/daeba9575fa2/CRIE2019-4825357.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9143/6350591/d837128fa73f/CRIE2019-4825357.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9143/6350591/daeba9575fa2/CRIE2019-4825357.002.jpg

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本文引用的文献

1
Spontaneous and medically induced cerebrospinal fluid leakage in the setting of pituitary adenomas: review of the literature.垂体腺瘤患者并发自发性和医源性脑脊液漏:文献复习。
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Bromocriptine or cabergoline-induced cerebrospinal fluid rhinorrhea: A life-threatening complication during management of prolactinoma.溴隐亭或卡麦角林诱发的脑脊液鼻漏:泌乳素瘤治疗期间的一种危及生命的并发症。
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Spontaneous cerebrospinal fluid rhinorrhoea as the presenting feature of an invasive macroprolactinoma.
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Nonsurgical cerebrospinal fluid rhinorrhea in invasive macroprolactinoma: incidence, radiological, and clinicopathological features.侵袭性大泌乳素瘤的非手术性脑脊液鼻漏:发病率、影像学及临床病理特征
J Clin Endocrinol Metab. 2007 Oct;92(10):3829-35. doi: 10.1210/jc.2007-0373. Epub 2007 Jul 10.
7
Cerebrospinal fluid leakage as complication of treatment with cabergoline for macroprolactinomas.脑脊液漏作为卡麦角林治疗大泌乳素瘤的并发症
J Endocrinol Invest. 2006 Dec;29(11):1001-5. doi: 10.1007/BF03349214.
8
Giant prolactinomas: clinical management and long-term follow up.巨大泌乳素瘤:临床管理与长期随访
J Neurosurg. 2002 Aug;97(2):299-306. doi: 10.3171/jns.2002.97.2.0299.
9
Cerebrospinal fluid fistula as the presenting manifestation of pituitary adenoma: case report with a 4-year follow-up.脑脊液瘘作为垂体腺瘤的首发表现:病例报告及4年随访
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