Table 1
Summary of published cases* of chorea and ballism associated with cerebral toxoplasmosis and intracranial tuberculosis.
Abbreviations: ART, antiretroviral therapy; BG, basal ganglia; CSF, cerebrospinal fluid; CT, computed tomography; CXR, chest radiograph; d, day/s; F, female; HB, hemiballismus; HC, hemichorea; HCHB, hemichorea-hemiballismus; HIV, human immunodeficiency virus; HR, isoniazid, rifampicin; HRZE, isoniazid, rifampicin, pyrazinamide, ethambutol; M, male; mo, month/s; MRI, magnetic resonance imaging; NR, not reported; PCR, polymerase chain reaction; PPD, purified protein derivative; Pt, patient; Pyr, pyrimethamine; STN, subthalamic nucleus; T. gondii, Toxoplasma gondii; TB, tuberculosis; TMP/SMX, trimethoprim/sulfamethoxazole; w, week/s; y, year/s.
Legends:
* Included are all relevant articles with either English full text or abstract with sufficient patient data for review; specific data is included when available.
† CD4 count in cells/mL in parentheses when available.
§ Died due to other causes.
** Definite TB meningitis is based on autopsy, positive cerebrospinal fluid smear or culture for acid-fast bacilli while probable TB meningitis is based on clinical and CSF findings, evidence of TB in extraneural sites, positive CSF enzyme-linked immunosorbent assay or adenosine deaminase activity or if at least two of the following were positive (Mantoux test, CXR, CT or MRI, and a history of TB).
†† Included patients had clinical or laboratory evidence of central nervous system tuberculosis.
| AUTHOR & YEAR | NO. OF CASES | AGE IN YEARS/SEX | HIV STATUS (CD4 COUNT)† | MODE OF ONSET AND SITE | INVESTIGATIONS | TREATMENT | CLINICAL OUTCOME | RADIOLOGIC RESPONSE |
|---|---|---|---|---|---|---|---|---|
| Cerebral toxoplasmosis (Total number of cases = 29) | ||||||||
| Navia [5] 1986 | 2 | Pt 1: NR/M | + | Chronic, chorea of four limbs | CT: bilateral BG hypodense lesions, positive T. gondii serology, autopsy revealed T. gondii tachyzoites on multiple brain lesions | Pyr, sulfadiazine | Death | NR |
| Pt 2: NR/M | + | Chronic, left choreoathetosis | CT: left internal capsule and right occipital abscess, positive T. gondii serology, autopsy revealed T. gondii tachyzoites on multiple brain lesions including BG and thalamus | Pyr, sulfadiazine | Death | NR | ||
| Nath [6] 1987 | 3 | Pt 1: 26/F | + | NR, right HCHB | CT: left BG and frontal abscess, positive T. gondii titers in CSF, positive CSF cryptococcal antigen | NR | Improved | Improved |
| Pt 2: 56/M | + | NR, right HB | CT: normal, positive T. gondii titers in CSF, autopsy showed toxoplasmosis abscess on left STN | NR | Death | NR | ||
| Pt 3: 47/M | + | NR, right HCHB | CT: left BG and cerebral hemisphere abscess, positive T. gondii serology | NR | NR | NR | ||
| Dewey [7] 1989 | 2 | Pt 1: 26/F | + | Acute, right HCHB, right face | CT or MRI: left BG or frontal lobe Toxoplasma abscess | Haloperidol | Partial recovery | NR |
| Pt 2: 47/M | + | Acute, right HCHB | CT or MRI: left BG or cerebral hemisphere Toxoplasma abscess | Pyr sulfate | Partial recovery | NR | ||
| Sanchez-Ramos [8] 1989 | 1 | 33/F | + | Subacute, left HCHB | CT: right STN, thalamus, cerebellum and left caudate abscess, positive T. gondii serology | Pyr, sulfadiazine | Complete recovery at 2 mo§ | Marked resolution at 20 d |
| Awada [9] 1993 | 1 | 33/F | + | Subacute, right HB | CT: left thalamus and STN lesion, negative T. gondii serology | Pyr, sulfadiazine | Complete recovery at 6 w | Complete resolution at 6 w |
| Nath [10] 1993 | 1 | 32/M | + | Chronic, left HCHB, left face | CT: right globus pallidus, bilateral cerebral hemisphere abscess, positive T. gondii serology | Pyr, sulfadiazine, haloperidol | Partial recovery at 10 d§ | Partial resolution at 10 d |
| Garretto [11] 1995 | 1 | 26/M | + (218) | Subacute, left HCHB | MRI: right cerebral peduncle, frontal and left temporo-occipital abscess, positive T. gondii serology | Pyr, sulfadiazine, haloperidol | Minimal recovery | Partial resolution after 6 w |
| Manji [12] 1995 | 1 | 28/F | + | Subacute, right choreoathetosis | CT: bilateral lentiform nuclei, thalami, cerebral hemisphere lesions, positive T. gondii serology | Pyr, sulfadiazine | Partial recovery at 5 w | Complete resolution at 5 mo |
| Krauss [13] 1996 | 1 | 32/M | + | Subacute, right HB | CT or MRI: abscess in left STN from toxoplasmosis | Antitoxoplasmosis; underwent cervical ventrolateral chordotomy | No recovery with medication; complete recovery after surgery at 6 mo | Complete resolution |
| Maggi [14] 1996 | 3 | Pt 1: 27/M | + (50) | NR, left arm choreoathetosis | MRI: right thalamic, bilateral hemisphere abscess from toxoplasmosis | Pyr, sulfadiazine | Complete recovery at 1 mo | NR |
| Pt 2: 31/M | + (30) | NR, left HCHB | MRI: right caudate, STN abscess from toxoplasmosis | Pyr, sulfadiazine | Complete recovery at 1 mo | Complete resolution at 4 mo | ||
| Pt 3: 32/M | + (30) | NR, left choreoathetosis | MRI: right STN, midbrain and bilateral hemisphere abscess from toxoplasmosis | Pyr, sulfadiazine | Complete recovery at 10 d | Complete resolution at 3 mo | ||
| Piccolo [15] 1999 | 2 | Pt 1: 27/M | + | Acute, right HC | CT and MRI: abscess in left STN, positive T. gondii serology | Pyr and sulfamethopyrazine | Complete recovery at 2 mo | Complete resolution at 1 mo |
| Pt 2: 35/M | + (10) | Acute, chorea of four limbs, face and mouth | MRI: abscess in right cerebral peduncle and BG, left occipital, temporal thalamocapsular and frontal operculum, autopsy revealed T. gondii tachyzoites on circular brain lesions | NR | Death | NR | ||
| De Mattos [16] 2002 | 6 | Mean age 32.5 (Range: 27 to 40)/all male | + | Acute, left HCHB in 5 cases, right HCHB in 1 case | Ring enhancing lesions in striatum or in the frontal lobe suggestive of toxoplasmosis | NR | NR | NR |
| Piccolo [17] 2003 | 2 | Pt 1: 27/NR | + | NR, hemichorea | Toxoplasma abscess in contralateral STN | NR | Complete recovery at 6 mo | NR |
| Pt 2: 35/NR | + | NR, generalized chorea | Pathologically confirmed toxoplasmosis in BG | NR | Death | NR | ||
| Rabhi [18] 2011 | 1 | 59/F | + (91) | Chronic, left HCHB | MRI: right capsulothalamic abscess, positive T. gondii serology | TMP/SMX | Complete recovery at 2 w | Marked resolution |
| Sta. Maria [19] 2012 | 1 | 30/M | + | Acute, left HC | MRI: Abscess in cerebral peduncle and frontal lobe, positive T. gondii serology | TMP/SMX | NR | NR |
| Moccia [20] 2013 | 1 | 32/F | + (250) | Subacute, right HCHB | MRI: ring enhancing lesion in left caudate to midbrain, T. gondii IgG positive 25 UI/mL | Pyr, sulfadiazine, haloperidol, ART | Complete recovery at 3 mo | Small residual lesion in the midbrain at 3 mo |
| Intracranial tuberculosis (Total number of cases = 23) | ||||||||
| Bedwell [21] 1960 | 1 | 39/M | NR | Subacute, left HC | Autopsy revealed tuberculoma in the right STN | Anti-TB | Death | NR |
| Riela [22] 1982 | 1 | 11 mo/M | NR | Subacute, left choreoathetosis | CT: hypodensity in right BG, positive M. tuberculosis CSF culture, positive PPD | HR | Complete recovery | NR |
| Delaporte [23] 1983 | 1 | 33/NR | NR | NR, left HB | CT: tuberculoma in right STN | Anti-TB | Complete recovery at 1 y | Marked resolution at 1 y |
| Babikian [24] 1985 | 1 | 2.5/M | NR | Acute, right HCHB | CT: bilateral BG infarct, hydrocephalus, positive M. tuberculosis CSF culture | HR, streptomycin, diphenhydramine | Partial recovery at 1 y | NR |
| Alarcón [25] 2000 | 7** | Range: 0.5 to 27/4 male, 3 female | NR | Acute to subacute, 3 HC, 4 generalized chorea | 5 cases of definite TB meningitis, 2 probable TB meningitis, HC cases had infarcts in contralateral thalamus, caudate or internal capsule; generalized chorea cases had cortico-subcortical atrophy, hydrocephalus or bilateral caudo-capsular infarct | Anti-TB | 2 with complete recovery, 3 with partial recovery, 2 died at 2 y | NR |
| Kalita [26] 2003 | 1 | 16/F | – (191) | Subacute, left HC, left face | CT: tuberculoma in right caudate and putamen, hydrocephalus and meningeal enhancement, positive CSF acid-fast bacillus smear, right upper fibrocavitary lesion on CXR | HRZE | Marked improvement at 1 w, complete recovery at 3 mo | NR |
| Ozer [27] 2006 | 1 | 78/M | – | Chronic, right HCHB | MRI: tuberculoma in the left thalamus, STN and midbrain, positive PPD | HRZE | Partial recovery at 6 mo, complete recovery at 2 y | Partial resolution at 6 mo |
| Alarcón [28] 2011 | 8†† | Pt 1: 9/F | NR | Subacute, left HC | CT or MRI: tuberculoma in frontal region and right thalamus, hydrocephalus | Anti-TB | Complete recovery at 1 y | NR |
| Pt 2: 16/F | NR | Subacute, generalized chorea | CT or MRI: tuberculoma in left thalamus, hydrocephalus | Anti-TB | Complete recovery at 1 y | NR | ||
| Pt 3: 28/F | NR | Subacute, right HC | CT or MRI: tuberculoma in left thalamus and frontoparietal region, hydrocephalus | Anti-TB | Complete recovery at 1 y | NR | ||
| Pt 4: 22/M | NR | Subacute, right HC | CT or MRI: tuberculoma in left caudate and thalamus, hydrocephalus | Anti-TB | Partial recovery at 1 y | NR | ||
| Pt 5: 8/F | NR | Subacute, right HC | CT or MRI: tuberculoma in left thalamus and parietal region, right cerebellum | Anti-TB | Complete recovery at 1 y | NR | ||
| Pt 6: 17/F | NR | Subacute, right arm chorea | CT or MRI: left frontotemporal tuberculoma | Anti-TB | Partial recovery at 1 y | NR | ||
| Pt 7: 42/F | NR | Subacute, right arm chorea | CT or MRI: right cerebellar tuberculoma | Anti-TB | Death | NR | ||
| Pt 8: 27/F | NR | Subacute, right HC | CT or MRI: tuberculoma in left frontotemporal region and thalamus | Anti-TB | Partial recovery at 1 y | NR | ||
| Pinto [29] 2013 | 1 | 14/F | – | Acute, right HC | MRI: left caudate and lenticulo-capsular infarct, diagnosed miliary TB (meningitis and pulmonary disease) | HRZE | NR | NR |
| Rubio-Hernandez [30] 2020 | 1 | 33/F | – | Subacute, left HCHB | MRI: multiple tuberculomas in right thalamus, STN, cerebral peduncle, cerebellum and subcortical white matter, positive CSF M. tuberculosis PCR, CXR showed miliary pattern and left nodular lesion | HRZE, olanzapine | Complete resolution at 6 mo | Partial resolution at 1 mo |
Video
Hemichorea-hemiballismus (HCHB). Segment 1. Patient at presentation three days after symptom onset showing random and occasionally ballistic movements of the left upper and lower limbs. Segment 2. Post-treatment at nine months after symptom onset showing marked reduction in amplitude and intensity of the HCHB and residual mild choreoathetoid movements of the left upper limb.
Figure 1
Cranial magnetic resonance imaging of the patient at presentation. A non-enhancing hypointense focus (white arrow) on postcontrast T1 weighted imaging (A) is seen on the right subthalamic region and cerebral peduncle appearing hyperintense on axial (B), coronal (C) and sagittal (D) T2 weighted images, representing focal cerebritis. Also seen is an abscess formation in the left frontal region (white arrowhead) with a slightly hyperintense rim on noncontrast T1 weighted image (E) suggestive of tuberculoma. On postcontrast axial T1 weighted image (F), it appears rim-enhancing with a nodular area of enhancement along its medial wall possibly an “eccentric target sign”, with a hypointense core and surrounding moderate vasogenic edema on axial T2 weighted (G) and diffusion weighted (H) imaging. Considerations for this space-occupying lesion include a tuberculoma and cerebral toxoplasmosis.
Figure 2
Timeline of events. This timeline highlights the chronology of the patient’s symptoms, work-up, management, and status on follow-up. Abbreviations: ART, antiretroviral therapy; CSF, cerebrospinal fluid; CT, computed tomography; HIV, human immunodeficiency virus; HRZE, isoniazid/rifampicin/pyrazinamide/ethambutol; PCR, polymerase chain reaction; PTB, pulmonary tuberculosis; TMP/SMX, trimethoprim/sulfamethoxazole.