Review
Isolated brainstem cysticercosis: A review

https://doi.org/10.1016/j.clineuro.2012.12.026Get rights and content

Abstract

Objective

Review of patients with isolated brainstem cysticercus to outline the features of this form of neurocysticercosis.

Methods

MEDLINE and manual search of patients with isolated brainstem cysticercus. Abstracted data included: demographic profile, clinical manifestations, neuroimaging findings, evolutive stage of parasites neurocysticercosis, therapy, and follow-up.

Results

Twenty-nine patients were reviewed. Of these, 22 (76%) came from India. Mean age was 31 years, and 72% were men. Parasites were located in midbrain (16 patients), pons (12 patients), and medulla (one patient). All but three lesions were less than 10 mm in diameter and most were at or near the midline. Most common clinical forms of presentation were isolated paresis of the third cranial nerve, internuclear ophthalmoplegia, and crossed brainstem syndromes. Neuroimaging studies showed colloidal cysticercus in 24 patients, vesicular cysts in four, and a calcification in one. Fourteen patients received cysticidal drugs, eight were treated with steroids alone, and three received no therapy at all. The remaining four patients underwent surgical resection of the lesion. Twenty-seven patients recovered completely and the remaining two were left with mild sequelae. Control neuroimaging studies showed complete or partial resolution of the lesion in the 18 patients in whom they were performed.

Conclusions

Isolated brainstem cysticercosis is rare. Clinical and neuroimaging findings on admission allowed a correct differentiation of this condition from other space-occupying lesions of the brainstem (tuberculomas, abscesses, gliomas) in most patients. The prognosis is benign provided the patients receive prompt therapy.

Introduction

Neurocysticercosis is highly pleomorphic due to individual differences in the number and location of lesions within the CNS [1]. Cysticerci may be located in brain parenchyma, subarachnoid space, ventricular system, or spinal cord. In the former, parasites usually lodge in the cerebral cortex or the subcortical white matter, due to the high vascular supply of these areas [2]. So, parenchymal brain cysticercosis usually presents as recurrent seizures which, in up to 70% of cases, constitutes the sole manifestation of the disease [3], [4], [5]. Brainstem location of parenchymal cysticerci usually happens in the context of disseminated disease with simultaneous involvement of cerebral hemispheres, basal ganglia, or cerebellum [6], [7]. The occurrence of isolated brainstem cysticercosis poses diagnostic and therapeutic challenges as this form neurocysticercosis seems to be rare, may present with clinical and neuroimaging findings indistinguishable from those of other brainstem lesions, and creates concern on the potential risks related to therapy. Here, the literature on isolated brainstem cysticercosis is reviewed to outline its clinical features, and to comment on the diagnostic and therapeutic challenges of this overlooked form of the disease.

Section snippets

Methods

A literature search of case reports or case-series of patients with isolated brainstem cysticercosis was performed using the electronic database of MEDLINE (National Library of Medicine, Bethesda, MD). The search was updated to November 5, 2012, using the key words “cysticercosis” and “neurocysticercosis” were combined with “brainstem”, “midbrain”, “mesencephalon” “pons”, “pontine” “medulla”, and “bulbar”. Limits were not applied to the search. Thereafter, a manual search that included the

Results

The search detected 28 papers reporting 32 patients with isolated brainstem cysticercus [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22], [23], [24], [25], [26], [27], [28], [29], [30], [31], [32], [33], [34], [35], [36]. Three of these papers (all describing single case-reports) were not included in the review; in two cases, the original publication could not be found [34], [35], and in the other, information was incomplete [36]. Some other articles mentioned

Discussion

This review suggests that isolated brainstem cysticercosis is rare. Only 29 cases were found after a thorough search of the literature, and most of them came from India. However, these findings must be interpreted with caution as the nature of a series based on the literature is quite partial, since a number of cases may have been reported in local journals or not reported at all.

That most cases came from India should not be surprising since it is just in this country where the most common

Conflict of interest

Nothing to disclose.

Contributors

Design of the study was done by O.H.D.; review and abstraction of data by O.H.D. and V.J.D.; analysis of data by V.J.D.; and drafting of the manuscript was done by O.H.D. and V.J.D.

References (43)

  • E.T. Kimura-Hayama et al.

    Neurocysticercosis: radiologic–pathologic correlation

    Radiographics

    (2010)
  • O.H. Del Brutto et al.

    Epilepsy due to neurocysticercosis: analysis of 203 patients

    Neurology

    (1992)
  • H. Carabin et al.

    Clinical manifestations associated with neurocysticercosis: a systematic review

    PLOS Neglected Tropical Diseases

    (2011)
  • J.S. Kim et al.

    Third cranial nerve palsy from midbrain neurocysticercosis: repeated exacerbation on tapering corticosteroids

    Journal of Neuro-Ophthalmology

    (2004)
  • O.H. Del Brutto et al.

    Proposed diagnostic criteria for neurocysticercosis

    Neurology

    (2001)
  • K.S. Anand et al.

    Solitary pontine cysticercal cyst presenting as isolated sixth nerve palsy

    Journal, Indian Academy of Clinical Medicine

    (2006)
  • W.O. Arruda et al.

    Brainstem cysticercosis simulating cystic tumor lesion

    Arquivos de Neuro-Psiquiatria

    (1994)
  • F. Barinagarrementeria et al.

    Ataxic hemiparesis from parenchymal brain cysticercosis

    Journal of Neurology

    (1988)
  • R. Bhatia et al.

    Isolated facial myokymia as a presenting feature of pontine neurocysticercosis

    Movement Disorders

    (2008)
  • A.K. Bhattacharya et al.

    Bilateral internuclear ophthalmoplegia. An atypical presentation of neurocysticercosis

    Journal, Indian Academy of Clinical Medicine

    (2006)
  • S.R. Chandran et al.

    Neurocysticercosis presenting as isolated wall-eyed mononuclear internuclear ophthalmoplegia with contraversive ocular tilt reaction

    Journal of Neurosciences in Rural Practice

    (2012)
  • Cited by (13)

    View all citing articles on Scopus
    View full text