Two Cousins with Acute Hemichorea after BBIBP-CorV (Sinopharm) COVID-19 Vaccine.

The recent COVID‐19 pandemic has affected the world in several ways and caused many challenges. COVID‐19 vaccines have variable degrees of immunogenicity and safety.

Here we report on two adolescents who developed hemichorea after receiving BBIBP‐CorV (Sinopharm), an inactivated virus COVID‐19 vaccine.

A 13‐year‐old boy presented to our Movement Disorders clinic 7 days after receiving his first dose of BBIBP‐CorV (Sinopharm) inactivated virus. He began to experience left‐sided involuntary movements. His past medical history was unremarkable, and he was otherwise healthy. He did not experience fever, cough, sore throat, or other symptoms. He was born full‐term by vaginal delivery to nonconsanguineous parents and did not have any pre‐ and postnatal complications. His family history was unremarkable.

On examination, the boy had large‐amplitude choreic movements affecting the right side of his body that affected his gait (Video S1‐A). He did not have chorea in the face, abnormal tongue movements, or speech impairment. The remainder of the examination was unremarkable.

A paternal cousin of the first, who was an 18‐years‐old man, received BBIBP‐CorV (Sinopharm) 1 week after case 1 and after 7 days developed left‐sided hemichorea. His past medical history was negative, and he was also born to nonconsanguineous parents. On examination, he had choreic movements that mainly affected the left upper limb and shoulder (Video S2) but also involved the left lower limb. His face was not affected, and he did not have abnormal tongue movements or speech impairment. The remainder of the examination was normal.

Extensive laboratory investigations of the blood and cerebrospinal fluid were carried out in both patients and were unremarkable (Table 1).

TABLE 1

Laboratory investing to exclude secondary causes

Laboratory results	Investigations	Case 1	Case 2
CSF	Cell count, differential, protein, glucose, oligoclonal bands, culture, Sars‐Cov‐2 PCR	0 RBC, 0 WBC, protein 51 (g/L), glucose 56 (mg/dL) Otherwise negative	3 RBC, 4 WBC, protein 34 (g/L), glucose 64 (mg/dL) Otherwise negative
Autoimmune encephalitis/paraneoplastic (serum and CSF)	Anti‐NMDAR, anti‐CASPR2, anti‐LGI‐1, anti‐GABA‐B, anti‐DPPX, anti‐VGCC, anti‐Yo, ANNA‐1, ANNA‐2, amphiphysin antibodies, anti‐Ma2, anti‐AMPAR, anti‐GAD	Negative	Negative
Vasculitis/autoimmune (serum)	ANAs, ENAs, ANCAs, anti‐RFs, anti‐CCPs, dsDNAs, C3, C4, cryoglobulins, anticardiolipin antibody (IgM and IgG), lupus anticoagulant, B2 glycoprotein antibody, PLP	Negative	Negative
Routine lab tests	Full blood count, electrolytes, calcium, magnesium, sodium, potassium, phosphate, liver function tests, INR, serum glucose, HBA1c, CRP, ESR, TSH, T4, BUN, creatinine, cupper, ceruloplasmin	All in normal ranges (BS: 101)	All in normal ranges (BS: 111)

Abbreviations: CSF, cerebrospinal fluid; PCR, polymerase chain reaction; NMDAR, N‐methyl‐d‐aspartate receptor; VGCC, voltage‐gated calcium channel antibody; ANA, antinuclear antibody; AMPAR, AMPA receptor; GAD, glutamic acid decarboxylase; ENA, extractable nuclear antigen antibody; ANCA, anti‐neutrophil cytoplasmic antibody; anti‐RF, rheumatoid factor antibody; anti‐CCP, cyclic‐citrullinated peptide antibody; dsDNA, double‐stranded DNA antibody; C3, C4, complement; INR, international normalized ratio; HBA1c, glycosylated hemoglobin; CRP, C‐reactive protein; ESR, erythrocyte sedimentation rate; TSH, thyroid‐stimulating hormone; BUN, blood urea nitrogen.

Brain MRI (magnetic resonance imaging) of case 1 showed multiple white matter lesions, one of them enhanced with gadolinium (Fig. 1). Patient 2 had few nonspecific white matter lesions (not shown).

FIG 1

Sagittal fluid‐attenuated inversion recovery magnetic resonance imaging (FLAIR MRI) imaging (top image) shows multiple white matter lesions, and axial T1 with gadolinium (bottom image) demonstrates a juxtacortical gadolinium‐enhancing lesion. [Color figure can be viewed at wileyonlinelibrary.com]

Both were treated with intravenous methylprednisolone (1 g/d) for 3 days followed by oral prednisolone (50 mg/d) and tetrabenazine (25 mg/d), which caused moderate improvement after a 2‐week follow‐up (Video S1‐B). At 1‐month follow‐up, patient 1 improved, and patient 2 still had mild choreic movements.

This is a description of hemichorea in two adolescents after Sinopharm vaccination, which is one of the most widely used COVID‐19 vaccines globally.

Three cases with hemichorea have been reported previously, 2 after AZD1222 vaccination and 1 after Pfizer‐BioNTech COVID‐19 vaccination, but all reported patients were in their 80s.2, 3

The pathophysiology of hemichorea in these cases is not clear, but several theories have been described for hemichorea after COVID‐19 vaccination. These include focal immune‐mediated endotheliopathy induced by the spike protein and functional disturbance in contralateral thalamus, which was confirmed by single‐photon emission computed tomography.

On the contrary, Decio et al reported an 11‐year‐old girl who developed chorea after the human papillomavirus vaccine. She responded to steroids, which supported an autoimmune mechanism as the pathophysiology.

In addition, a 62‐year‐old man has been reported with acute chorea after getting a Sars‐Cov‐2 infection, and the authors concluded that inflammation may have played a role in the development of chorea in this patient.

Presentation of hemichorea in our patients suggests an inflammatory mechanism, as the first patient had inflammatory white matter changes on brain MRI, but the occurrence in the two cousins may indicate the possibility of a genetic predisposition as a factor for developing this phenomenon.

Although COVID‐19 vaccines are generally safe, and neurological complications are rare and self‐limited, these 2 cases suggest that movement disorders can be a complication of vaccines.

Author Roles

(1) Research project: A. Conception, B. Organization, C. Execution; (2) Manuscript: A. Writing of the first draft, B. Review and critique.

MS: 1A, 1B, 1C, 2A

ME: 1A, 1B, 1C, 2B

Ethical conduct and informed consent

This study was approved by the ethical committee of Shahid Beheshti University of Medical Science (IR.SBMU.RETECH.REC.1400.387). We hereby confirm that the present study conforms to the ethical standards and guidelines of the journal. The patient has given written and informed consent for online publication of his videos.

Full financial disclosures for the previous 12 months

None.

Video S1. (A) Case 1: a 13‐year‐old boy presented 7 days after the first dose of BBIBP‐CorV (Sinopharm) inactivated virus with choreic movements on the right side. The video shows continuous, nonpatterned large‐amplitude choreic movements in the right arm and leg, which affected his gait (as the mask covered his face and is not clear in the video, it should be clarified that the face was not affected).

(B) Case 1: 2 weeks after treatment, choreic movement improved dramatically, and his gait improved secondary to lessened chorea in the leg, but persistence of mild choreic movements can be observed mainly in the hand.

Click here for additional data file.

Video S2. Case 2: an 18‐year‐old man who presented with choreic movement 1 week after the first dose of BBIBP‐CorV (Sinopharm) inactivated virus. As shown in the video, the chorea affected his left side and mainly his shoulder, but it was milder in his leg (not shown in the video).

Click here for additional data file.