Cauda equina syndrome secondary to extramedullary spinal sparganosis

Tzu-Hsieh Hsu; Tzu-Tsao Chung; Yi-Ani Chen; Among-Hsuan Chung

doi:10.4103/fjs.fjs_53_20

CASE REPORT

Year : 2020 | Volume : 53 | Issue : 6 | Page : 230-232

Cauda equina syndrome secondary to extramedullary spinal sparganosis

Tzu-Hsieh Hsu, Tzu-Tsao Chung, Yi-Ani Chen, Among-Hsuan Chung
Department of Neurological Surgery, National Defense Medical Center, Tri-Service General Hospital, Taipei, Taiwan

Date of Submission	21-Apr-2020
Date of Decision	15-May-2020
Date of Acceptance	29-Jun-2020
Date of Web Publication	19-Dec-2020

Correspondence Address:
Tzu-Tsao Chung
Department of Neurological Surgery, Tri-Service General Hospital, No. 325, Section 2, Chenggong Road, Neihu District, Taipei 114
Taiwan

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/fjs.fjs_53_20

Abstract

We report a rare case of sparganosis presented as cauda equina syndrome. Cauda equina syndrome secondary to extramedullary spinal sparganosis infection represents a diagnostic challenge. It should be carefully considered in the differential diagnosis of patients presenting similar characteristics. Although both medical and surgical treatments contribute to the management of this condition, in cases of neurologic deficit due to the mass effect of the lesion, urgent surgical decompression should be attempted to prevent further worsening of the neurological condition.

Keywords: Cauda equina syndrome, sparganosis, Spirometra erinaceieuropaei

How to cite this article:
Hsu TH, Chung TT, Chen YA, Chung AH. Cauda equina syndrome secondary to extramedullary spinal sparganosis. Formos J Surg 2020;53:230-2

How to cite this URL:
Hsu TH, Chung TT, Chen YA, Chung AH. Cauda equina syndrome secondary to extramedullary spinal sparganosis. Formos J Surg [serial online] 2020 [cited 2021 Jan 27];53:230-2. Available from: https://www.e-fjs.org/text.asp?2020/53/6/230/304021

Introduction

Sparganosis is a rare parasitic infection, caused by the migration of tapeworm larvae from its first intermediate host, cyclops, to the second intermediate host, including freshwater fish, amphibians, and reptiles.^[1],[2] The definitive hosts are cats, dogs, and other wild carnivores.^[2] Human infection aberrantly occurs by: (1) drinking untreated water containing either the larvae or first intermediate hosts, (2) ingesting raw or inadequately cooked flesh of infected snakes or frogs, (3) applying the flesh of an infected intermediate host as a poultice to a wound.

Sparganosis usually appears as slowly growing and migratory subcutaneous nodules. The parasite may be found anywhere in the body, including the central nervous system (CNS).^[3]

Only in rare cases, sparganosis involves the spinal cord, usually at the thoracic to the lumbar region.^[4] In this report, we describe a case with spinal sparganosis. The diagnosis, treatment, and outcome of patient.

Case Report

The patient, a 45-year-old female, arrived in the neurosurgery department of our hospital with severe low back pain and bilateral lower limb weakness on March, 2016. The patient provided consent to be included in the manuscript. She had previously undergone thoracolumbar spine surgery (total laminectomies, T12-L5), with the diagnosis of sparganum infection in July, 2012 [Figure 1]. She had a history of ingesting inadequately cooked snake. She presented to our hospital complaining of lumbodorsal pain, progressive paraparesis with sensory impairment for 2 months, and bowel/bladder dysfunction for several days. On examination, the patient was found to have a normal body temperature; skin nodules or organomegaly was not observed. Mental status and cranial nerve functions were normal.

Figure 1: (a) the lesion image was not contrast-enhanced; (b) magnetic resonance imaging of the lumbar spine showing a hyper-intense bead-like lesion on a T2-weighted image below the T11 vertebral level (white arrow) (c) image after the first operation

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Neurological examinations revealed reduced motor tone of both legs. Motor strength of the left lower extremity decreased from Grade V to II in the hamstring, iliopsoas, and quadriceps muscles, Grade I–II in the ankle and toe plantar flexor muscles, and Grade I in the ankle dorsiflexor muscles and the extensor hallucis longus muscle. For the right lower extremity, motor strength decreased to Grade II. Sensations below the T11 dermatome were decreased on both sides with perianal paresthesia. The anal sphincter tone was reduced. Clinically, she was diagnosed with cauda equina syndrome.

Laboratory data indicated her white blood cell to be 7.11 × 10³/μl, with a neutrophil count of 61.6%, lymphocyte count of 29.1%, and eosinophil count of 2%. Her C-reactive protein level was 0.14 mg/dl. The results of liver function tests were normal. Stool examination revealed no evidence of parasite infection.

Magnetic resonance imaging of the whole spine showed multiple extramedullary intradural serpiginous lesions in the subarachnoid space, continuous from the T11 region to L5 [Figure 2]. The mass lesion remained presence after the first operation.

Figure 2: Multiple extramedullary intradural serpiginous lesions in the subarachnoid space (a) T2 image (b) T1 with contrast image

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Nerve conduction velocity tests showed prolonged F wave, distal latency, and reduced cyclic adenosine monophosphate amplitudes of bilateral peroneal and tibial nerves. Electromyography revealed: (1) increased insertion activities, such as fibrillation, positive sharp wave, and fasciculation at all sampled muscles; (2) mild-to-moderate polyphasic waves with prolonged duration increased the amplitudes in sampling muscle, such as tensor fasciae latae, and in lower lumbar paraspinal muscle. The result was compatible with motor peroneal and tibial neuropathies superimposed with a sub-acute denervated status of lumbar polyradiculopathy involving left leg; lumbosacral plexopathy with the involvement of motor roots was also suspected.

Laminectomy was performed at T10 and T11 levels to obtain a tissue sample for histopathological examinations. During the operation, we noticed a matted mass involving nerve roots, with remarkable inflammation and multiple cystic lesions. The lesions were found to have multiple ovum [Figure 3] between the nerve roots and were completely removed with partial lysis of the adhesions under nerve root monitoring.

Figure 3: Operative finding: egg of tapeworm and adhesion tissue

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The histological sections were stained with hematoxylin and eosin; histological examinations demonstrated a degenerated solid section of folded cestode larva having irregular folding tegument, which was homogeneously eosinophilic in color. Many calcareous corpuscles [Figure 4], characteristic of a tapeworm larva, were also seen. There were mild inflammatory cell infiltrations, including eosinophils, lymphocytes, few plasma cells, and neutrophils in the larval section [Figure 4]. Definite diagnosis of sparganosis could be made from the histopathological findings.

Figure 4: Histopathological findings of the resected lesion (a) section of a degenerating tapeworm larva with homogenously eosinophilic and calcareous corpuscles, along with inflammatory cell infiltrations; ×100; (b) Calcareous corpuscles (arrows) and mild inflammatory cell infiltrations are seen; ×400

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After laminectomy at T10 and T11 levels, the patient received mebendazole therapy, after which her lumbosacral pain moderately improved. However, she persistently complained of a dull, uncomfortable sensation on both lower limbs and buttocks. Paraparesis with sensory impairment, and difficulty in urination and defecation persisted after series of treatments.

Discussion

Sparganosis is an uncommon disease in humans, caused by the larvae of the tapeworm genus Spirometra, whose definitive hosts include both domestic and wild cats and dogs.^[5],[6] The disease usually involves the subcutaneous tissue or muscle of the chest, abdominal wall, or limbs. The involvement of CNS is rare, and the involvement of the spinal cord is extremely rare.^[3],[7]

The sparganum more commonly involves intradural (70%) rather than extradural sites,^[4] usually at the thoracic level,^[1],[4],[8] followed by that at the cervical level.^[9] There are only few cases of sparganosis involving the conus medullaris and cauda equina.^[6],[10]

In nearly all patients with spinal sparganosis, lesions were detected in the thoracic cord. Our patient was unique in demonstrating long and multiple intradural extramedullary lesions extending from the T12 vertebral levels distally until the L5 level. The patient presented with severe lumbodorsal pain due to a mass compressing the adjacent spinal cord and subsequent spinal cord edema. Pain in the nerve root may occur long before the signs of the spinal cord compression have developed. In our patient, lumbodorsal pain developed before paraparesis and bowel/bladder involvement. Although the route of entry of worms into the spinal cord remains unclear, their hematogenous spread seems likely.^[4]

It is a challenge to diagnose sparganosis and differentiate it from neoplastic and inflammatory disorders or other parasitic visceral larva migrans, since it is rare and has nonspecific manifestations. We could not easily find any specific diagnostic feature from neuroradiological images of these kinds of cases. An immunological approach, using ELISA, is presently used for diagnostic purposes. ELISA for the detection of anti-sparganum IgG antibody is highly sensitive (85.7%–100%) and specific (95.7%). However, it cannot identify the causative worm at the species level. Polymerase chain reaction-based molecular techniques should be considered for the identification of causative pathogens in such infectious diseases.

Conclusion

Although the sparganosis probability of CNS infection is very rare, it can lead to various neurological deficits and should be considered as a differential diagnosis. Therefore, detailed history taking, imaging, and immunoassay can be helpful for the diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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