Suratos Cezar Thomas Reyes, Benitez James Albert Edward Lim, Urquiza Sheen Corvera, Sacro Cheryl Anne Lubaton
Department of Neurosciences, University of the Philippines Manila College of Medicine, Manila, Philippines.
Department of Pediatrics, University of the Philippines Manila College of Medicine, Manila, Philippines.
BMJ Case Rep. 2018 Apr 5;2018:bcr-2017-223429. doi: 10.1136/bcr-2017-223429.
Non-ketotic hyperglycaemic hemichorea-hemiballismus (NHHH) is commonly seen among elderly Asian women with type 2 diabetes mellitus. Here, we present a case of a 16-year-old Filipina with type 1 diabetes mellitus who is poorly compliant to her medications and subsequently developed right hemichorea-hemiballismus (HH). She was initially admitted with hyperglycaemia but was negative for ketonuria or metabolic acidosis. Neuroimaging showed bilateral lentiform nuclei and left caudate hyperdensities on CT and T1-weighted hyperintensity on MRI. Blood glucose was controlled with insulin. Haloperidol and clonazepam were started for the HH with gradual resolution of symptoms in 6 weeks. This is the fifth reported case of NHHH seen among the paediatric age group. NHHH in the paediatric population is clinically and radiographically similar to NHHH seen among adults. Correction of hyperglycaemia results in clinical improvement and radiographic resolution of lesions but persistent cases may necessitate specific treatment targeted towards the abnormal movements.
非酮症高血糖性偏侧舞蹈症-偏侧投掷症(NHHH)常见于患有2型糖尿病的老年亚洲女性。在此,我们报告一例16岁的菲律宾1型糖尿病患者,她对药物治疗依从性差,随后出现了右侧偏侧舞蹈症-偏侧投掷症(HH)。她最初因高血糖入院,但尿酮体或代谢性酸中毒检测呈阴性。神经影像学检查显示,CT上双侧豆状核和左侧尾状核高密度影,MRI上T1加权像呈高信号。通过胰岛素控制血糖。开始使用氟哌啶醇和氯硝西泮治疗HH,症状在6周内逐渐缓解。这是儿科年龄组中第五例报告的NHHH病例。儿科人群中的NHHH在临床和影像学上与成人中的NHHH相似。纠正高血糖可导致临床症状改善和病变的影像学消退,但持续性病例可能需要针对异常运动进行特定治疗。