非酮症高血糖性偏身舞蹈症二例报道并文献复习
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摘要
目的探讨非酮症高血糖性偏身舞蹈症患者的临床特点、诊断和治疗、发病机制,提高对本病的认识,避免误诊、漏诊。方法分析2017-03-31和2017-04-17入住山西医科大学附属人民医院的2例非酮症高血糖性偏身舞蹈症患者的临床症状、体征、影像学资料,并复习相关文献。结果 2例均为老年女性糖尿病患者,突发单侧肢体舞蹈样动作,血糖水平升高,酮体阴性。其中1例患者颅脑磁共振成像(MRI)示舞蹈症状对侧基底核区T1加权像高信号及对应部位CT高密度影。经过积极降糖治疗,1例舞蹈样动作完全消失,另1例明显缓解。结论非酮症高血糖性偏身舞蹈症是一种罕见的糖尿病并发症,多见于血糖水平控制不佳的老年女性糖尿病患者。以非酮症性高血糖、单侧舞蹈样不自主运动及MRI对侧基底核区T1加权像高信号或CT高密度影为特点。发病初始立即使用胰岛素积极降糖治疗,能够缩短舞蹈样动作改善或消失时间,改善患者生活质量。
Objective To study the clinical manifestation,diagnosis,therapy and mechanism of hemichorea associated with non-ketotic hyperglycemia,so as to enhance awareness and avoid misdiagnosis or missed diagnosis of this disease.Methods Clinical symptoms,signs and imaging examinations of two patients with hemichorea associated with non-ketotic hyperglycemia admitted to Shanxi Provincial People's Hospital Affiliated to Shanxi Medical University on 31 March 2017 and 17 April 2017 were analyzed,and related literature were reviewed.Results Two patients were aged women with diabetes,who had acute onset hemichorea of unilateral limb,high blood glucose and negative ketone body.One subject had high signal intensity of T1-weighted MR imaging on the contralateral basal ganglia,and high density of corresponding lesion on CT.After controling glucose positively,choreiform movement of one subject had disappeared,and of the other one relieved obviously.Conclusion Hemichorea associated with non-ketotic hyperglycemia is a rare complication of diabetes mellitus,which is commonly seen in elderly women with bad controlled diabetes.It is characterized by non-ketotic hyperglycemia,unilateral involuntary choreiform movements,and contralateral basal ganglia hyper-intensity on T1-weighted MR imaging or high density on CT.Timely insulin therapy may reduce the time of improvement and disappearance of choreiform movements,and improve quality of life in patients.
Objective To study the clinical manifestation,diagnosis,therapy and mechanism of hemichorea associated with non-ketotic hyperglycemia,so as to enhance awareness and avoid misdiagnosis or missed diagnosis of this disease.Methods Clinical symptoms,signs and imaging examinations of two patients with hemichorea associated with non-ketotic hyperglycemia admitted to Shanxi Provincial People's Hospital Affiliated to Shanxi Medical University on 31 March 2017 and 17 April 2017 were analyzed,and related literature were reviewed.Results Two patients were aged women with diabetes,who had acute onset hemichorea of unilateral limb,high blood glucose and negative ketone body.One subject had high signal intensity of T1-weighted MR imaging on the contralateral basal ganglia,and high density of corresponding lesion on CT.After controling glucose positively,choreiform movement of one subject had disappeared,and of the other one relieved obviously.Conclusion Hemichorea associated with non-ketotic hyperglycemia is a rare complication of diabetes mellitus,which is commonly seen in elderly women with bad controlled diabetes.It is characterized by non-ketotic hyperglycemia,unilateral involuntary choreiform movements,and contralateral basal ganglia hyper-intensity on T1-weighted MR imaging or high density on CT.Timely insulin therapy may reduce the time of improvement and disappearance of choreiform movements,and improve quality of life in patients.
引文
[1]OH S H,LEE K Y,IM J H,et al.Chorea associated with nonketotic 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.DOI:10.1016/S0022-510X(02)00133-8.
[2]刘志锋,蔡金辉,刘庆余,等.非酮症性高血糖偏侧舞蹈症脑部CT及MRI表现[J].影像诊断与介入放射学,2016,25(2):133-137.DOI:10.3969/j.issn.1005-8001.2016.02.009.LIU Z F,CAI J H,LIU Q Y,et al.Cranial CT and MR imaging findings in non-ketotic hyperglycemia hemichorea[J].Diagnostic Imaging&Interventional Radiology,2016,25(2):133-137.DOI:10.3969/j.issn.1005-8001.2016.02.009.
[3]LEE S H,SHIN J A,KIM J H,et al.Chorea-ballism associated with nonketotic hyperglycaemia or diabetic ketoacidosis:characteristics of 25 patients in Korea[J].Diabetes Res Clin Pract,2011,93(2):e80-83.DOI:10.1016/j.diabres.2011.05.003.
[4]TAN Y,XIN X,XIAO Q,et al.Hemiballism-hemichorea induced by ketotic hyperglycemia:case report with PET study and review of the literature[J].Transl Neurodegener,2014,3:14.DOI:10.1186/2047-9158-3-14.
[5]AQUINO J H,SPITZ M,PEREIRA J S.Hemichorea-hemiballismus as the first sign of type 1b diabetes during adolescence and its recurrence in the setting of infection[J].J Child Neurol,2015,30(10):1362-1365.DOI:10.1177/0883073814553972.
[6]ROY U,DAS S K,MUKHERJEE A,et al.Irreversible hemichorea-hemiballism in a case of nonketotic hyperglycemia presenting as the initial manifestation of diabetes mellitus[J].Tremor Other Hyperkinet Mov(N Y),2016,6:393.DOI:10.7916/D8QZ2B3F.
[7]COSENTINO C,TORRES L,NUNEZ Y,et al.Hemichorea/hemiballism associated with hyperglycemia:report of 20cases[J].Tremor Other Hyperkinet Mov(N Y),2016,6:402.DOI:10.7916/D8DN454P.
[8]LIN C J,HUANG P.Delayed onset diabetic striatopathy:hemichorea-hemiballism one month after a hyperglycemic episode[J].Am J Emerg Med,2017,35(7):1036.e3-1036.e4.DOI:10.1016/j.ajem.2017.02.018.
[9]TUNG C S,GUO Y C,LAI C L,et al.Irreversible striatal neuroimaging abnormalities secondary to prolonged,uncontrolled diabetes mellitus in the setting of progressive focal neurological symptoms[J].Neurol Sci,2010,31(1):57-60.DOI:10.1007/s10072-009-0127-6.
[10]胡裕效.正常人和2型糖尿病人脑葡萄糖代谢局域及网络属性的初步研究[D].南京:南京大学,2014.HU Y X.Regional and networks characteristics of brain glucose metabolic networks in healthy and type 2 diabetes mellitus subjects[D].Nanjing:Nanjing University,2014.
[11]HSU J L,WANG H C,HSU W C.Hyperglycemia-induced unilateral basal ganglion lesions with and without hemichorea.A PETstudy[J].J Neurol,2004,251(12):1486-1490.DOI:10.1007/s00415-004-0571-4.
[12]LAI P H,CHEN P C,CHANG M H,et al.In vivo proton MRspectroscopy of chorea-ballismus in diabetes mellitus[J].Neuroradiology,2001,43(7):525-531.
[13]SHIMOMURA T,NOZAKI Y,TAMURA K.Hemichorea-hemiballism associated with nonketotic hyperglycemia and presenting with unilateral hyperintensity of the putamen on MRI T1-weighted images-a case report[J].No To Shinkei,1995,47(6):557-561.
[2]刘志锋,蔡金辉,刘庆余,等.非酮症性高血糖偏侧舞蹈症脑部CT及MRI表现[J].影像诊断与介入放射学,2016,25(2):133-137.DOI:10.3969/j.issn.1005-8001.2016.02.009.LIU Z F,CAI J H,LIU Q Y,et al.Cranial CT and MR imaging findings in non-ketotic hyperglycemia hemichorea[J].Diagnostic Imaging&Interventional Radiology,2016,25(2):133-137.DOI:10.3969/j.issn.1005-8001.2016.02.009.
[3]LEE S H,SHIN J A,KIM J H,et al.Chorea-ballism associated with nonketotic hyperglycaemia or diabetic ketoacidosis:characteristics of 25 patients in Korea[J].Diabetes Res Clin Pract,2011,93(2):e80-83.DOI:10.1016/j.diabres.2011.05.003.
[4]TAN Y,XIN X,XIAO Q,et al.Hemiballism-hemichorea induced by ketotic hyperglycemia:case report with PET study and review of the literature[J].Transl Neurodegener,2014,3:14.DOI:10.1186/2047-9158-3-14.
[5]AQUINO J H,SPITZ M,PEREIRA J S.Hemichorea-hemiballismus as the first sign of type 1b diabetes during adolescence and its recurrence in the setting of infection[J].J Child Neurol,2015,30(10):1362-1365.DOI:10.1177/0883073814553972.
[6]ROY U,DAS S K,MUKHERJEE A,et al.Irreversible hemichorea-hemiballism in a case of nonketotic hyperglycemia presenting as the initial manifestation of diabetes mellitus[J].Tremor Other Hyperkinet Mov(N Y),2016,6:393.DOI:10.7916/D8QZ2B3F.
[7]COSENTINO C,TORRES L,NUNEZ Y,et al.Hemichorea/hemiballism associated with hyperglycemia:report of 20cases[J].Tremor Other Hyperkinet Mov(N Y),2016,6:402.DOI:10.7916/D8DN454P.
[8]LIN C J,HUANG P.Delayed onset diabetic striatopathy:hemichorea-hemiballism one month after a hyperglycemic episode[J].Am J Emerg Med,2017,35(7):1036.e3-1036.e4.DOI:10.1016/j.ajem.2017.02.018.
[9]TUNG C S,GUO Y C,LAI C L,et al.Irreversible striatal neuroimaging abnormalities secondary to prolonged,uncontrolled diabetes mellitus in the setting of progressive focal neurological symptoms[J].Neurol Sci,2010,31(1):57-60.DOI:10.1007/s10072-009-0127-6.
[10]胡裕效.正常人和2型糖尿病人脑葡萄糖代谢局域及网络属性的初步研究[D].南京:南京大学,2014.HU Y X.Regional and networks characteristics of brain glucose metabolic networks in healthy and type 2 diabetes mellitus subjects[D].Nanjing:Nanjing University,2014.
[11]HSU J L,WANG H C,HSU W C.Hyperglycemia-induced unilateral basal ganglion lesions with and without hemichorea.A PETstudy[J].J Neurol,2004,251(12):1486-1490.DOI:10.1007/s00415-004-0571-4.
[12]LAI P H,CHEN P C,CHANG M H,et al.In vivo proton MRspectroscopy of chorea-ballismus in diabetes mellitus[J].Neuroradiology,2001,43(7):525-531.
[13]SHIMOMURA T,NOZAKI Y,TAMURA K.Hemichorea-hemiballism associated with nonketotic hyperglycemia and presenting with unilateral hyperintensity of the putamen on MRI T1-weighted images-a case report[J].No To Shinkei,1995,47(6):557-561.