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Year : 2021  |  Volume : 4  |  Issue : 2  |  Page : 255-259

A rare case of primary high cervical intramedullary cysticercosis: Uncomplicated surgery but a preoperative diagnostic predicament

Indraprastha Apollo Hospital, Sarita Vihar, New Delhi, India

Date of Submission22-Sep-2020
Date of Acceptance08-Apr-2021
Date of Web Publication16-Jul-2021

Correspondence Address:
Sunit Mediratta
Consultant Neurosurgeon, Indraprastha Apollo Hospital, Sarita Vihar, Mathura Road, New Delhi.
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ISJ.ISJ_75_20

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Neurocysticercosis (NCC) is the most common parasitic infection involving the central nervous system. The involvement of spine by this disease is extremely uncommon. When the spine does get affected, it generally develops as a synchronous infection with an existing cerebral cysticercosis and usually involves the intradural extramedullary space. Primary intramedullary involvement is rare. A case of primary high cervical intramedullary cysticercosis with non-progressive symptoms is described. In this case, a pre-operative diagnosis could not be ascertained. The patient underwent total surgical resection of the lesion and made excellent recovery. Post-operative evaluation did not reveal disease at other sites. This case highlights the safety and ease of surgical resection in intramedullary NCC.

Keywords: Cervical, intramedullary, neurocysticercosis, primary

How to cite this article:
Mediratta S. A rare case of primary high cervical intramedullary cysticercosis: Uncomplicated surgery but a preoperative diagnostic predicament. Indian Spine J 2021;4:255-9

How to cite this URL:
Mediratta S. A rare case of primary high cervical intramedullary cysticercosis: Uncomplicated surgery but a preoperative diagnostic predicament. Indian Spine J [serial online] 2021 [cited 2022 Jan 24];4:255-9. Available from: https://www.isjonline.com/text.asp?2021/4/2/255/321581

  Introduction Top

The parasitic infection most commonly affecting the human central nervous system is cysticercosis.[1],[2],[3],[4],[5] However, it is extremely uncommon for the spine to be involved by cysticercosis, unless and until there is a pre-existing infection of the brain,[1],[4] even then most often the infection is limited to the intradural extramedullary space.[1],[4]

Primary spinal intramedullary involvement is rare and only 63 cases were reported till 2017,[1] with a few sporadic reports in the last 3 years.[3],[4] Without the appearance of a scolex, the radiological findings can be non-specific and the disease creates a preoperative diagnostic predicament.[4],[6],[7] This is a report of one such case wherein the diagnosis could not be ascertained preoperatively. The patient showed near complete recovery after surgical excision of the lesion.

  Case Report Top

A 27-year-old man presented with history of neck pain with paraesthesia involving bilateral upper limbs of 1-month duration without any limb weakness. He had no history of fever, sensory loss, bladder, or bowel involvement. His neurological examination revealed normal muscle tone in bilateral upper and lower limbs. The motor examination revealed normal strength in the right upper and bilateral lower limbs. Left shoulder flexion and abduction along with elbow flexion was 4+/5. Power in the left wrist and digits were normal. The deep tendon reflexes were brisk in the left upper limb along with a positive Babinski sign on the left. He had decreased pain sensation below the C2 dermatome on the right. The magnetic resonance imaging (MRI) of the cervical spine revealed an intramedullary lesion in the cervical cord at the C2 level, which was iso- to hypointense on T1-weighted (wt) [Figure 1] and hyperintense on T2-wt images [Figure 2] with well-defined bright peripheral enhancement [Figure 3]A and B]. Significant perilesional edema extending from the medulla to the D1 level could be appreciated on the T2-wt images [Figure 2]. The hematological investigations were unremarkable with normal total leukocyte count and differential count along with normal erythrocyte sedimentation rate (ESR). Chest X-ray was normal. With the radiological imaging, a differential diagnosis of tuberculoma, abscess, ependymoma, and cystic astrocytoma was considered and the patient was advised surgical intervention. The patient was started on intravenous dexamethasone 4 mg/8 hourly preoperatively.
Figure 1: MRI cervical spine, T1-wt sagittal images showing mildly hypo- to isointense intramedullary lesion at the level of C2 vertebra

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Figure 2: T2-wt sagittal MRI image showing hyperintense intramedullary lesion at the C2 level with significant perilesional edema extending from the medulla to dorsal D1

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Figure 3: A. Postcontrast MRI axial images showing intramedullary peripheral ring-enhancing lesions at C2. B. Postcontrast MRI sagittal images showing intramedullary peripheral ring-enhancing lesion at C2

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In prone position, the patient underwent a C2, partial C3 laminectomy and total excision of the lesion. On opening the dura, the lesion was seen as a slight pale bulge on the surface of the cord posterolaterally toward the left of the midline. The myelotomy was made directly over the pale avascular area just adjacent to the midline toward the left. The lesion was well defined with a plane of cleavage between the cyst wall and parenchyma. It appeared as a thin transparent capsule containing clear fluid. One of the larger cysts ruptured intraoperatively before removal, whereas the other could be removed in toto [Figure 4]. This was achieved using gentle traction on the cyst wall with cupped forceps, separating the cord parenchyma with a small cotton patty. The operative field was irrigated with 3% hypertonic saline. Postoperatively, the patient was extubated; he had transient weakness of the left shoulder, elbow, and wrist and improved to the preoperative levels within 2 weeks along with complete resolution of pain and paraesthesia. Dexamethsone was tapered off after 1 week of postoperative period. Albendazole was prescribed at 15 mg/kg/day for a period of 4 weeks in the postoperative period. An ultrasound abdomen along with an MRI brain was done postoperatively, which did not reveal any other lesions. The histopathology of the lesion was reported as neurocysticercosis (NCC) [Figure 5]. At 6 months follow-up, the patient is asymptomatic; however, his left shoulder and elbow flexion is 4+/5.
Figure 4: One of the translucent cysts removed in toto

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Figure 5: Hematoxylin–eosin-stained section at 20× magnification showing the larva with invaginated scolex

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  Discussion Top

The most common parasitic infection of the central nervous system affecting people all around the world is NCC.[2],[3],[4] It is caused by the larval stage of the tapeworm, Taenia solium (pork tapeworm).[1],[2] The disease is more prevalent in the developing countries of Southeast Asia, sub-Saharan Africa, Eastern Europe, and South and Central America.[1],[4] In almost 90% of the cases, the age group affected is between 20 and 45 years.[5] The disease commonly affects the intracranial compartment, and the spine is an extremely uncommon site of cysticerci infection.[1],[4] The incidence of spinal NCC is 1.5–3% and almost all being contemporaneous infections with coexisting cerebral involvement.[1],[3] Even when a de novo infection of the spine does take place, it generally affects the intradural extramedullary space.[1],[5] Primary intramedullary spinal involvement by NCC is extremely rare with just about 64 cases reported worldwide.[1],[4]

Humans are accidental dead-end hosts of the parasitic cycle.[2],[4]T. solium eggs are shed in the stool of a human tapeworm carrier.[2] Accidental ingestion of the eggs occurs by consumption of uncooked pork and raw unclean vegetables.[2],[4] The eggs thereafter release the embryos in the intestine where they penetrate the intestinal mucosa to gain access to the vasculature, thereby reaching the peripheral tissues and the central nervous system, and develop into cysticerci.[2],[6] Primary spinal involvement most often has been reported in the lower thoracic cord.[1],[5] This is postulated and assumed to be due to the fact that the dorsal spine receives relatively greater blood flow,[1],[5] also contributing to it is the prominent artery of Adamkiewicz, which often arises directly from the aorta and enters the cord between the D9 and D11 regions.[8] The clinical features in a patient with intramedullary NCC can be varied and non-specific.[4] It usually depends on the location and size of the cyst along with the presence of any associated inflammatory reaction.[4] Larval degeneration results in evoking an immune response causing inflammation and perilesional edema producing focal deficits.[1],[4],[7] Although most patients have gradual onset of symptoms, an acute inflammatory response may result in sudden worsening.[5] The disease may remain asymptomatic for years without eliciting an immune response.[1]

In order to radiologically confirm the diagnosis of intramedullary NCC, MRI is by far the best imaging modality.[1],[5],[8],[9] However, in the absence of an identifiable scolex within the cyst, the imaging can be non-specific[4],[7] and invariably shows up as a cystic ring enhancing lesion with perilesional edema,[8],[9] as seen in our case.

In such a scenario in which a scolex is not identifiable, one would have to consider other inflammatory, granulomatous, neoplastic, or developmental cystic lesions in the differential diagnosis, viz., abscess, tuberculoma, cystic ependymoma or astrocytoma, neurenteric cysts.[1],[4],[7],[8]

Concomitant intracranial lesions can help with corroborative evidence to establish a diagnosis in suspected cases as about 30% of the patients may harbor them.[8] Additional evidence toward the diagnosis may be provided by cerebrospinal fluid evaluation in suspected cases as cysticerci antibody detection by enzyme-linked immune sorbent assay or by enzyme-linked immune electrotransfer blot assay in the serum has a very high sensitivity and specificity.[1],[3] The antibody detection tests were not done in this case due to low index of suspicion.

On T1-wt images of MRI, in the vesicular stage of NCC, the cyst appears as a well-defined hypointense lesion, with a hyperintense scolex visualized as an eccentric dot within the cyst.[5],[8] This is considered a pathognomonic neuroimaging finding in cysticercal lesions[8],[9]and is seen in about 40% of the cases.[8] In the colloidal stage, the thickened cyst wall and its contents appear hyperintense on T1-wt image,[5] the capsule appears hypointense on T2-wt image,[5] and the hyperintense cyst contents, therefore, obscure the scolex on T1-wt images.[5],[10],[11]

A preoperative diagnosis of NCC in our case was difficult to envisage. This was because the lesion appeared in an uncommon location in the high cervical cord; furthermore, on T1-wt image, it was isointense along with non-visualization of a hyperintense scolex. The blood eosinophil count which is occasionally high in helminthic infections was also normal.

In patients in whom intramedullary NCC has been diagnosed preoperatively and their clinical signs and symptoms remain mild and non-progressive, they can be managed conservatively with anthelminthic therapy using albendazole 15 mg/kg/day for 4–6 weeks along with dexamethasone.[1],[3] Oral steroids are known to increase the blood concentration levels of albendazole and also mitigate the inflammatory response during treatment.[1],[5],[12],[13]

Cases have been reported wherein there has been clinical and radiological resolution of the disease with medical therapy alone when used for primary intramedullary NCC.[1],[5]

Surgical excision remains the treatment of choice for all cases in which the diagnosis is in doubt or patient has progressive neurological worsening.[1],[3] This is to be followed by anthelminthic therapy using albendazole 15 mg/kg/day for 4–6 weeks as NCC is considered to be a focal involvement of a systemic disease.[1],[12] Even though many have reported difficulty in excising these cysts owing to the inflammatory process causing adhesions around the cyst,[1],[3],[14] no such issues were encountered in this particular case, despite the inflammation and extensive perilesional edema as seen on the MRI [Figure 2]. A few authors have reported abandoning the procedure or being able to achieve a subtotal resection owing to the intense inflammatory adhesions between the cyst wall and the intramedullary parenchyma.[1],[15] A total excision was possible in this case, and one of the cysts could be removed in toto. This may be attributed to the lesion reaching almost up to the surface of the cord posterolaterally which allowed an initial early access to the plane of cleavage without much intervening neural tissue. Slight traction on the cyst wall with cupped forceps along with a gentle advancement of a small cotton patty in the plane between the cyst wall and cord allows delivery of the cyst in toto.

  Conclusion Top

Isolated intramedullary NCC is a rare condition and a preoperative diagnosis is challenging without the appearance of a scolex on MRI. All cases with suspicious intramedullary NCC should undergo cranial MRI as an aid to diagnosis and to rule out concomitant intracranial pathology. Surgical excision results in excellent outcomes.

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Conflicts of interest

There are no conflicts of interest.

  References Top

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Blaizot R, Melot B, Schepers K, Nicolas M, Gaumond S, Poullain P, et al. Report of three imported cases of neurocysticercosis in Guadeloupe. BMC Infect Dis 2017;17:106.  Back to cited text no. 2
Maste PS, Lokanath YK, Mahantshetti SS, Soumya S. Isolated intramedullary spinal cysticercosis: A case report with review of literature of a rare presentation. Asian J Neurosurg 2018;13:154-6.  Back to cited text no. 3
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Chhiber SS, Singh B, Bansal P, Pandita KK, Razdan S, Singh J. Intramedullary spinal cysticercosis cured with medical therapy: Case report and review of literature. Surg Neurol 2009;72:765-8; discussion 768-9.  Back to cited text no. 12
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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]


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