非典型非酮症高血糖性偏侧舞蹈症三例

doi:10.11817/j.issn.1673-9248.2023.03.010

非酮症高血糖性偏侧舞蹈症是一种罕见的糖尿病并发症，尽管具有偏侧肢体的舞蹈样动作、非酮症高血糖、影像学表现为病变肢体对侧基底核区高信号等特点，但仍有部分患者以非典型的临床表现起病，且相关报道较少。本文通过对海南医学院第一附属医院收治的3例特殊的非酮症高血糖性偏侧舞蹈症患者的临床及影像学特点进行总结分析，以提高临床医师对罕见病的认识，减少不必要的漏诊、误诊。

关键词: 糖尿病, 非酮症高血糖, 偏侧舞蹈症

Non-ketotic hyperglycemic hemichorea is a rare clinical complication. It is characterized by dance-like movements in the paralyzed limb, non-ketotic hyperglycemia, and imaging manifestations of high signals in the contralateral basal ganglia of the diseased limb. However some patients still start with atypical clinical manifestations, and related reports are few. Hereon, the clinical and imaging characteristics of 3 atypical patients with non-ketotic hyperglycemic hemichorea admitted to the First Affiliated Hospital of Hainan Medical College were summarized and analyzed, so as to improve clinicians' understanding of rare diseases and reduce unnecessary missed diagnosis and misdiagnosis.

Key words: Diabetes Mellitus, Non-ketotic hyperglycemia, Hemichorea

图1 患者1 头颅影像学检查结果。图a：螺旋CT颅脑平扫可见双侧放射冠、基底节区腔隙性脑梗死，未见异常高密度灶；图b：磁共振脑功能成像（弥散加权成像）未见弥散受限；图c：磁共振颅脑平扫T1 加权像可见右侧基底节区异常高信号影、无明显水肿及占位；图d：磁共振颅脑平扫T2 加权像可见右侧基底节区呈等信号；图e：磁共振脑血管成像可见双侧大脑前动脉、大脑中动脉、大脑后动脉无明显狭窄、闭塞，脑动脉硬化

图2 患者2头颅影像学检查结果。图a：螺旋CT颅脑平扫示双侧放射冠区腔隙性脑梗死，未见异常高密度灶；图b：磁共振脑功能成像（弥散加权成像）未见弥散受限；图c：磁共振颅脑平扫T1加权像可见右侧基底节区异常高信号影、边界较清、无水肿及占位；图d：磁共振颅脑平扫T2加权像可见右侧基底核区呈等信号；图e：磁共振脑血管成像可见双侧颈内动脉、大脑中动脉、大脑后动脉走行僵直，管壁欠光滑，管腔粗细不均匀且部分呈串珠样狭窄

图3 患者3头颅影像学检查结果。图a：螺旋CT颅脑平扫可见左侧尾状核、豆状核区片状高密度灶，CT值为53~58 HU；图b：磁共振脑功能成像（弥散加权成像）未见弥散受限；图c：磁共振颅脑平扫T1加权像可见左侧基底核区片状高信号灶；图d：磁共振颅脑平扫T2加权像可见左侧基底核区片状低信号；图e：磁共振脑血管成像可见双侧大脑前动脉、大脑中动脉、大脑后动脉走行僵直，管壁欠光滑，管腔粗细不均匀，脑动脉硬化

1	Lin JB, Sng AA, Wang FS, et al. Acute hemichorea in a young type 1 diabetic [J]. Int J Neurosci, 2020, 130(7): 743-745.
2	Wang L, Song CL. Chorea associated with nonketotic hyperglycemia: an uncommon patient with bilateral movements [J]. J Clin Neurosci, 2015, 22(6): 1068-1069.
3	Zheng W, Chen L, Chen JH, et al. Hemichorea associated with non-ketotic hyperglycemia: a case report and literature review [J]. Front Neurol, 2020, 11: 96.
4	Zaitout Z. CT and MRI findings in the basal ganglia in non-ketotic hyperglycaemia associated hemichorea and hemi-ballismus (HC-HB) [J]. Neuroradiology, 2012, 54(10): 1119-1120.
5	Bedwell SF. Some observations on hemiballismus [J]. Neurology, 1960, 10: 619-622.
6	Jones HJ, Baker RA, Kott HS. Hypertensive putaminal hemorrhage presenting with hemichorea [J]. Stroke, 1985, 16(1): 130-131.
7	Chua C, Sun C, Hsu C, et al. "Diabetic striatopathy": clinical presentations, controversy, pathogenesis, treatments, and outcomes [J]. Sci Rep, 2020, 10(1): 1594.
8	Chen X, Ma C, Zhi L, et al. Hemichorea associated with nonketotic hyperglycemia: A case series of 16 patients and literature review [J]. J Clin Endocrinol Metab, 2023: dgad077. Online ahead of print.
9	葛建亮, 刘晓微, 曹红玉, 等. 非酮症性高血糖偏身舞蹈症5例临床特点分析并文献复习 [J]. 中国现代医药杂志, 2021, 23(4): 30-34.
10	Oh S, Lee K, Im J, et al. Chorea associated with non-ketotic hyperglycemia and hyperintensity basal ganglia lesion on T1-weighted brain MRI study: a meta-analysis of 53 cases including four present cases [J]. J Neurol Sci, 2002, 200(1-2): 57-62.
11	Satish PV, Pujitha K, Agrawal N, et al. Hemi-chorea in a patient with ketotic hyperglycemia: an unusual presentation [J]. J Clin Diagn Res, 2017, 11(5): OD24-OD25.
12	Galloway KY, Younus O, Quattrocchi G, et al. Diabetes and neurology: hemichorea-hemiballism in hyperglycaemia [J]. Clin Med (Lond), 2020, 20(Suppl 2): s21-s22.
13	Chang K, Tsou J, Chen S, et al. Temporal features of magnetic resonance imaging and spectroscopy in non-ketotic hyperglycemic chorea-ballism patients [J]. Eur J Neurol, 2010, 17(4): 589-593.
14	Ondo WG. Hyperglycemic nonketotic states and other metabolic imbalances [J]. Handb Clin Neurol, 2011, 100: 287-291.
15	Nath J, Jambhekar K, Rao C, et al. Radiological and pathological changes in hemiballism-hemichorea with striatal hyperintensity [J]. J Magn Reson Imaging, 2006, 23(4): 564-568.
16	Ahlskog JE, Nishino H, Evidente VG, et al. Persistent chorea triggered by hyperglycemic crisis in diabetics [J]. Mov Disord, 2001, 16(5): 890-898.
17	Kumar N, Prakash V. Pediatric hemichorea-hemiballismus associated with nonketotic hyperglycemia: a case report and review of the literature [J]. J Mov Disord, 2021, 14(3): 248-250.
18	Wang JH, Wu T, Deng BQ, et al. Hemichorea-hemiballismus associated with nonketotic hyperglycemia: a possible role of inflammation [J]. J Neurol Sci, 2009, 284(1-2): 198-202.
19	Sharma R, Buras E, Terashima T, et al. Hyperglycemia induces oxidative stress and impairs axonal transport rates in mice [J]. PLoS One, 2010, 5(10): e13463.
20	Achilles EI, Maus V, Fink GR, et al. [Hemichorea with contralateral high signal intensity putaminal lesion on T1-weighted images in non-ketotic hyperglycemia] [J]. Fortschr Neurol Psychiatr, 2016, 84(4): 222-225.
21	Ronald BP, Anthony EL. Hemiballism: revisiting a classic disorder [J]. Lancet Neurology, 2003, 2(11): 661-668.
22	Udare AS, Sankhe S, Mondel PK. Bilateral diabetic striatopathy [J]. Asian J Neurosurg, 2016, 11(2): 169.
23	梁国泰, 张育生. 非酮症高血糖性偏侧舞蹈症误诊脑出血一例 [J/OL]. 中华脑血管病杂志(电子版), 2021, 15(3): 196-198.
24	Ray S, Howlader S, Chakraborty S, et al. Hemichorea-hemiballism as the first presentation of type 2 diabetes [J]. Clin Diabetes, 2015, 33(2): 87-89.
25	Cho HS, Hong CT, Chan L. Hemichorea after hyperglycemia correction: a case report and a short review of hyperglycemia-related hemichorea at the euglycemic state [J]. Medicine (Baltimore), 2018, 97(10): e76.
26	Dubey S, Chatterjee S, Ghosh R, et al. Acute onset movement disorders in diabetes mellitus: a clinical series of 59 patients [J]. Eur J Neurol, 2022, 29(8): 2241-2248.
27	Tocco P, Barbieri F, Bonetti B, et al. Hemichorea-hemiballismus in patients with non-ketotic hyperglycemia [J]. Neurol Sci, 2016, 37(2): 297-298.
28	Son BC, Choi JG, Ko HC. Globus pallidus internus deep brain stimulation for disabling diabetic hemiballism/hemichorea [J]. Case Rep Neurol Med, 2017, 2017: 2165905.

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Three cases of atypical non-ketotic hyperglycemic hemichorea

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Three cases of atypical non-ketotic hyperglycemic hemichorea