Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2017 Sep 12;17(1):54.
doi: 10.1186/s12880-017-0228-2.

Case report of hyperglycemic nonketotic chorea with rapid radiological resolution

H M M T B Herath  1 , S P Pahalagamage  2 , Sunethra Senanayake  2
Affiliations
Case Reports

Case report of hyperglycemic nonketotic chorea with rapid radiological resolution

H M M T B Herath et al. BMC Med Imaging. .

Abstract

Background: Hemichorea is a rare manifestation of nonketotic hyperglycemia that usually affects elderly Asian women with poor glycemic control. Non-contrast computerized Tomography and T1- weighted Magnetic Resonance Imaging shows characteristic hyperintense basal ganglia lesions.

Case presentation: A Fifty-seven year old Sri Lankan female presented with a two-day history of right upper limb chorea. She had been diagnosed with diabetes mellitus one year ago, but was not on any treatment and did not have any micro vascular or macro vascular complications. Random blood sugar was 420 mg/dl and full blood count, liver function tests, renal function tests, inflammatory markers, thyroid function tests, Urine protein / creatinine ratio, electrocardiogram and 2D Echo were normal. Arterial blood gas did not show acidosis and ketone bodies were not detected in urine. Non-contrast computerized Tomography brain on day 1 showed left side hyperdense lentiform and caudate nuclei and MRI on day 3 showed slightly high signal intensity of left side basal ganglia on T1- weighted images and low signal intensity on T2-weighted and Fluid-attenuated inversion recovery images. She was started on insulin and a low dose of clonazepam and glycemic control was achieved on day 3. Two days later, the chorea completely disappeared. CT brain was repeated 4 days and 10 days following glycemic control, which showed rapid resolution of CT changes. Clonazepam was stopped in 2 weeks and chorea did not recur.

Conclusion: This is a rare manifestation of diabetes in Sri lanka and diagnosing this rare entity will direct clinicians to achieve optimum glycemic control as the treatment which will lead to rapid clinical response without any other medications. In this case report we high light that with the clinical improvement, repeating a CT scan even after a very short period like 2 weeks will show rapid radiological resolution. This repeat imaging can also be useful to confirm the diagnosis, which will minimize unnecessary investigations and treatments. Further cases of hyperglycemic nonketotic chorea with brain imaging performed within short intervals is needed to evaluate the nature of rapid radiological changes, which will be useful to understand the pathology of this condition.

Keywords: Hyperglycemic nonketotic chorea; Rapid radiological resolution.

PubMed Disclaimer

Conflict of interest statement

Ethics approval and consent to participate

Not applicable.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Competing interests

The Authors declare that there they have no competing interest.

Publisher’s Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Figures

Fig. 1
Fig. 1
Serial NCCT brain showing resolution of left side high intensity basal ganglia. a: Day 1 NCCT brain showing left side hyperdense lentiform and caudate nuclei shown by black arrows. b: NCCT brain 4 days after glycemic control showing rapid resolution of left side hyperdense lentiform and caudate nuclei (The hyperdense area is shown by blue arrows). c: NCCT brain 10 days after glycemic control showing rapid resolution of left side hyperdense lentiform and caudate nuclei (The initial hyperdense area is shown by red arrows, which has resolved by day 10)
Fig. 2
Fig. 2
a: MRI on day 3 showing slightly low signal intensity of left side basal ganglia on T2- weighted images. b: MRI on day 3 showing slightly high signal intensity of left side basal ganglia on T1- weighted images
See this image and copyright information in PMC

References

    1. Oh SH, 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 Neurol Sci. 2002;200(1–2):57–62. doi: 10.1016/S0022-510X(02)00133-8. - DOI - PubMed
    1. Chang KH, et al. Temporal features of magnetic resonance imaging and spectroscopy in non-ketotic hyperglycemic chorea-ballism patients. Eur J Neurol. 2010;17(4):589–593. doi: 10.1111/j.1468-1331.2009.02867.x. - DOI - PubMed
    1. Lai PH, et al. Chorea-ballismus with nonketotic hyperglycemia in primary diabetes mellitus. AJNR Am J Neuroradiol. 1996;17(6):1057–1064. - PMC - PubMed
    1. Cherian A, et al. Concepts and controversies in nonketotic hyperglycemia-induced hemichorea: further evidence from susceptibility-weighted MR imaging. J Magn Reson Imaging. 2009;29(3):699–703. doi: 10.1002/jmri.21672. - DOI - PubMed
    1. Lin JJ, et al. Presentation of striatal hyperintensity on T1-weighted MRI in patients with hemiballism-hemichorea caused by non-ketotic hyperglycemia: report of seven new cases and a review of literature. J Neurol. 2001;248(9):750–755. doi: 10.1007/s004150170089. - DOI - PubMed

Publication types