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当组织学诊断变得虚假时:一例孟乔森综合征病例。

When a Histologic Diagnosis Becomes Factitious: A Case of Munchausen Syndrome.

作者信息

Teixeira Mafalda, Santos Pedro, Bastos Furtado Ana, Delgado Alves José

机构信息

Department of Internal Medicine IV, Hospital Professor Doutor Fernando Fonseca, Amadora, PRT.

Department of Internal Medicine IV, Systemic Immune-Mediated Diseases Unit (UDIMS), Hospital Professor Doutor Fernando Fonseca, Amadora, PRT.

出版信息

Cureus. 2022 Sep 7;14(9):e28876. doi: 10.7759/cureus.28876. eCollection 2022 Sep.

Abstract

Recurrent episodes of hypoglycemia are uncommon in non-diabetic patients. The workup investigation must confirm hypoglycemia and distinguish between endogenous versus exogenous hyperinsulinism. Simultaneous measurements of plasma glucose, insulin, C-peptide, and a screen for oral hypoglycemic agents should be performed. According to the results, further imaging studies may be necessary. A 43-year-old woman suffering from recurrent hypoglycemia presented to the emergency room (ER) with a hypoglycemic coma. She has had multiple episodes of documented hypoglycemia for the last 13 years. The case was initially investigated, and laboratory studies revealed endogenous hyperinsulinism. Screening for sulfonylureas, anti-insulin and anti-insulin receptor antibodies were negative. Body imaging and positron emission tomography (PET) with Ga-DOTANOC did not show evidence of an insulinoma. The patient was submitted to a pancreatectomy, which revealed nesidioblastosis in the histologic examination. Since then, the patient became hyperglycemic but the insulin doses were progressively reduced until new episodes of hypoglycemia recurred and the insulin was stopped. Again, inappropriately high levels of insulin were found at the time of hypoglycemic episodes. Computed tomography (CT) and PET scans did not find evidence of an insulinoma. A C-peptide was later found to be negative and insulin ampoules were found in her possession, making a diagnosis of a factitious disorder. Although rare, factious disorders are frequently overlooked and challenging to diagnose. Since they are very resource and time-consuming, self-inflicted illnesses should always be considered and ruled out beforehand.

摘要

低血糖反复发作在非糖尿病患者中并不常见。检查必须确认低血糖,并区分内源性与外源性高胰岛素血症。应同时测量血浆葡萄糖、胰岛素、C肽,并筛查口服降糖药。根据结果,可能需要进一步的影像学检查。一名43岁患有复发性低血糖的女性因低血糖昏迷被送往急诊室。在过去13年里,她有多次记录在案的低血糖发作。该病例最初进行了检查,实验室研究显示为内源性高胰岛素血症。磺脲类药物、抗胰岛素和抗胰岛素受体抗体筛查均为阴性。身体成像和用镓-多他环素进行正电子发射断层扫描(PET)未显示胰岛素瘤的迹象。患者接受了胰腺切除术,组织学检查显示为胰岛细胞增殖症。从那时起,患者血糖升高,但胰岛素剂量逐渐减少,直到再次出现低血糖发作并停用胰岛素。再次,在低血糖发作时发现胰岛素水平异常升高。计算机断层扫描(CT)和PET扫描未发现胰岛素瘤的迹象。后来发现C肽为阴性,且在她身上发现了胰岛素安瓿,从而诊断为做作性障碍。尽管罕见,但做作性障碍经常被忽视且诊断具有挑战性。由于它们非常耗费资源和时间,因此应始终考虑并事先排除自我造成的疾病。

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本文引用的文献

1
Hyperinsulinaemic hypoglycaemia in children and adults.儿童和成人的高胰岛素血症性低血糖症。
Lancet Diabetes Endocrinol. 2017 Sep;5(9):729-742. doi: 10.1016/S2213-8587(16)30323-0. Epub 2016 Dec 1.
2
Nesidioblastosis in adults.成人胰岛细胞瘤。
Neoplasma. 2014;61(3):252-6. doi: 10.4149/neo_2014_047.
3
Hyperinsulinaemic hypoglycaemia.高胰岛素血症性低血糖症
Horm Metab Res. 2014 Mar;46(3):157-70. doi: 10.1055/s-0034-1367063. Epub 2014 Feb 20.
4
Diagnosis and management of insulinoma.胰岛素瘤的诊断与治疗。
World J Gastroenterol. 2013 Feb 14;19(6):829-37. doi: 10.3748/wjg.v19.i6.829.

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