Case report
International Journal of STD & AIDS
Hemichorea in a patient with
HIV-associated central nervous
system histoplasmosis
Ingrid Estrada-Bellmann, Carlos R Camara-Lemarroy,
Hazael Flores-Cantu, Hector J Calderon-Hernandez and
Hector J Villareal-Velazquez
Abstract
Central nervous system histoplasmosis is a rare opportunistic infection with a heterogeneous clinical presentation. We
describe the first case of human immunodeficiency virus-associated cerebral histoplasmosis presenting with hemichorea.
The patient recovered after treatment with conventional amphotericin B and itraconazole.
Keywords
AIDS, chorea, histoplasmosis, HIV, opportunistic infection, neurological, Histoplasma capsulatum
Date received: 24 September 2014; accepted: 21 November 2014
Introduction
Opportunistic central nervous system (CNS) infections
in patients with acquired immunodeficiency syndrome
(AIDS) have multiple clinical presentations.
Hemichorea and other movement disorders suggest
the diagnosis of a space occupying mass involving the
basal ganglia, usually due to toxoplasmosis.
Histoplasmosis is an uncommon cause of CNS infec-
tion that has never been associated with movement dis-
orders in patients with AIDS.
Case report
We admitted a 26-year-old Mexican man who has sex
with men (MSM) with a 10-day history of fever, malaise
and right-sided paraesthesia. He had recently visited
cave formations in the region. On clinical examination,
he had a right-sided hemiparesis (4/5), hyperreflexia and
a right-sided Babinski reflex. Cranial nerves examin-
ation was normal. A magnetic resonance imaging
(MRI) scan was ordered, which showed multiple con-
trast-enhancing lesions, surrounded by hyperintensities
on T2 FLAIR (suggestive of vasogenic oedema), affect-
ing multiple supra- and infra-tentorial regions including
the mesencephalus, cerebellum, basal ganglia and left
frontoparietal cortex (Figure 1). An enzyme-linked
immunosorbent assay for human immunodeficiency
2016, Vol. 27(1) 75–77
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DOI: 10.1177/0956462414564608
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virus (HIV) was positive, but CD4 count and HIV
viral load were not available. We suspected an infectious
aetiology and started empirical treatment with isoniazid,
rifampicin, pyrazinamide, co-trimoxazole, fluconazole
and dexamethasone. Cerebrospinal fluid (CSF) analysis
was unremarkable (normal lymphocyte count, protein,
lactate and glucose). CSF polymerase chain reaction for
tuberculosis and toxoplasmosis was negative and CSF
microscopy was negative for cryptococcus. Routine
blood tests showed a lymphopaenia (450 cells/mL),
hyponatraemia (127 mmol/L) and AST was raised to
two times the upper limit of normal. Other routine
blood investigations were normal. After three days, the
patient developed altered consciousness and left-sided
choreoathetosic movements. He had a brain biopsy of
the right temporal lesion which was culture positive for
Histoplasma capsulatum. There was no evidence of dis-
seminated disease. His treatment was rationalised to
conventional amphotericin B (50 mg/day for 30 days),
Servicio de Neurologia, Hospital Universitario Dr. José E. González,
Universidad Autónoma de Nuevo León, Madero y Gonzalitos S/N,
Monterrey, México
Corresponding author:
Carlos R Cámara-Lemarroy, Servicio de Neurologia, Hospital
Universitario Dr. José E. González, Universidad Autónoma de Nuevo
León, Madero y Gonzalitos S/N, Monterrey, NL 64460, México.
Email: crcamara83@hotmail.com
76 International Journal of STD & AIDS 27(1)

Figure 1. Brain MRI with contrast shows multiple cortical and deep subcortical ring enhancing lesions (a, b). On T2 FLAIR sequence,
the lesions were associated with vasogenic oedema, with severe involvement of the left basal ganglia (c). Marked radiological
improvement after six months (d).

itraconazole (400 mg/day, indefinitely) and dexametha-

sone (24 mg/day). His mental status and fever resolved.
His brain lesions subsequently showed radiological
improvement. At 30 days, he continued to show left-
sided choreoathetosic movements, and he was started
on haloperidol, diazepam and amantadine. We started
antiretroviral drugs six weeks after completing ampho-
tericin B treatment, and the patient showed no signs of
immune reconstitution syndrome. His abnormal move-
ments subsided after three months, and there has been
no evidence of disease recurrence.
Discussion
Involvement of the CNS is rare in patients with disse-
minated histoplasmosis, recognised in only 5–10% of
all patients, commonly affecting those with impaired
cellular immunity.1 In HIV-infected subjects with
AIDS, histoplasmosis is thought to be a very rare dis-
ease-defining infection, but the true burden of HIV-
associated histoplasmosis is unknown, as it is widely
mistaken for multidrug-resistant tuberculosis, leading
to numerous avoidable deaths.2
Estrada-Bellmann et al.
Clinically relevant movement disorders are rarely
identified in patients with HIV infection seen at tertiary
referral centres.3 Hemiballismus and hemichorea are
the most common hyperkinesias identified, and the
cause usually is a parenchymal lesion due to an oppor-
tunistic infection, particularly, toxoplasmosis. In a case
series of five HIV-infected patients with chorea, chorea
was associated with cerebral toxoplasmosis in two
patients, progressive multifocal leukoencephalopathy
in one, subacute HIV encephalopathy in another and
the last was probably iatrogenic.4 In another series of
50 patients with AIDS and cerebral toxoplasmosis,
only three were found to have chorea.5 To the best of
our knowledge, ours is the first case of histoplasmosis-
associated hemichorea in either immunocompetent or
immunocompromised patients. The lesions involving
the basal ganglia in our patient could explain the
observed symptoms.
The diagnosis of cerebral histoplasmosis requires a
high index of suspicion when there are no extraneural
signs or symptoms. Although there are no clinical trials
comparing different treatment regimens, expert opinion
favours an initial course of amphotericin B, followed by
at least one year of itraconazole.1 We used conventional
amphotericin B for 30 days (1.5 g total dose) and itra-
conazole with good results.
77
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this
article.
Funding
The authors received no financial support for the research,
authorship, and/or publication of this article.
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