Disseminated Human Subarachnoid Coenurosis

Abstract

Background: Traditionally, human coenurosis has been ascribed to Taenia multiceps while neurocysticercosis has been attributed solely to Taenia solium infection. Historically, however, the identification and differentiation of cestodal infection was primarily based on inaccurate morphological criteria. With the increasing availability of molecular methods, the accuracy of identification of the larval cestode species has improved, and cestodal species not typically associated with central nervous system (CNS) infection are now being identified as aetiological agents. Case report: We present a case of a 5-year-old male patient who presented with acute hydrocephalus. Initial MRI revealed multiple cysts in the cerebrospinal fluid (CSF) spaces with a predominance of clumped grape-like cysts in the basal cisterns with resultant acute obstructive hydrocephalus. The child underwent an emergency ventriculo-peritoneal (VP) shunt. A presumptive diagnosis of neurocysticercosis racemosus was made and the child was started on empiric albendazole (15 mg/kg/day) and praziquantel (30 mg/kg/day) treatment, along with concomitant prednisone (1 mg/kg) treatment. Despite prolonged anti-helminthic therapy, the child continued to deteriorate, and endoscopic removal of the 4th ventricular cysts was required. Post-operative MRI revealed radiological improvements, with a reduction in the number and size of cysts, especially in the basal cisterns, with no cysts visualized in the fourth ventricle. DNA was extracted from CSF and cyst tissue using the QiAMP DNA mini kit (Qiagen). The PCR performed on the extracted DNA displayed a band of 275 bp on an agarose gel. The consensus sequence had 97.68% similarity to Taenia serialis 12S ribosomal RNA gene. The child, unfortunately, continued to do poorly, requiring multiple VP shunt revisions for repeated blockage of the VP shunt system, and ultimately demised, despite the ‘successful’ surgical intervention and continued maximal medical management. Discussion and conclusions: There have been approximately 40 reported cases of human CNS coenurosis, with the assumed etiological agent being confined to T. multiceps. In 2020, the first case of human CNS coenurosis caused by T. serialis was reported. This case involved a single parenchymal lesion in the occipital lobe, which, following complete surgical excision, was confirmed to be T. serialis by mitochondrial gene sequencing. The case we present is the first case of disseminated subarachnoid coenurosis caused by T. serialis. It appears that T. serialis infection can mimic either of the two basic pathological forms of neurocysticercosis, namely, cysticercosis cellulosae or cysticercosis racemosus. We postulate that the term coenurosis racemosus is applicable if CNS T. serialis infection presents with extensive, multiple grape-like bladders proliferating within the subarachnoid space.

Keywords: Taenia serialis; Taenia solium; coenurosis; neurocysticercosis.

Figure 1

(a) T2 axial MRI brain demonstrates dilated lateral ventricles (arrows) with transependymal oedema in keeping with acute hydrocephalus. (b) T2 axial MRI and (c–f) FLAIR axial MRI brain demonstrates multiple cysts (arrows) in the basal cisterns, cerebellomedullary cistern, pontomedullary cistern, fourth ventricle and left posterior horn of lateral ventricle. Some cysts demonstrated a central dot in keeping with a scolex of NCC (arrowhead).

Figure 2

T2 sagittal MRI spine demonstrates (a) multiple cysts in the posterior spinal epidural space extending from lower cervical to the lumbar region with (b) smaller cysts within the subarachnoid space around filum terminale (arrows).

Figure 3

(a) T2 axial MRI and (b–c) FLAIR axial MRI brain demonstrates acute hydrocephalus with a re-distribution of cysts (arrows) within the CSF spaces. (d) GRE axial MRI brain demonstrates a new isolated right frontal lobe lesion (arrowhead) that blooms on GRE, most likely in keeping with a calcified granuloma.

Figure 4

(a) T2 axial MRI brain demonstrates a right subdural collection (arrow) and further improvement in the hydrocephalus. (b–d) FLAIR axial MRI brain shows improvement in number and size of cysts (arrows), especially in the basal cisterns, with cysts no longer visualized in in the 4th ventricle.