The Brief Case: Central Nervous System Sparganosis in a 53-Year-Old Thai Man

Abstract

A 53-year-old Thai man with a history of pemphigus vulgaris on chronic prednisolone (30 mg/day) presented to a hospital in Thailand with a 5-month history of lower back pain. He had initially been treated with tramadol, amitriptyline, and gabapentin without relief. Two months prior to presentation, he had developed weakness of the right leg, and he presented when weakness in his right foot made it difficult for him to keep his sandal on. He denied numbness, paresthesia, urinary retention, or bowel incontinence. On physical exam, he appeared well, with a temperature of 36.6°C, a blood pressure of 141/72 mm Hg, and a pulse of 74 beats per minute. Neurologic exam showed a knee flexion score of 4/5 and a foot dorsiflexion score of 3/5 on the right and a knee flexion score of 4/5 and foot dorsiflexion on the left. Sensation was intact bilaterally. Patellar reflexes were 1ϩ bilaterally, with downward plantar reflexes. Initial laboratory investigation showed a white blood cell count of 11,000 cells/mm 3 (normal, 4,000 to 11,000 cells/mm 3 ), with 81.1% neutrophils and 0.3% eosinophils (absolute eosinophil count, 33 cells/mm 3 ). Three serial stool specimens sent for microscopic ova and parasite identification were negative.Gadolinium-enhanced magnetic resonance imaging (MRI) of the lumbosacral spine was performed (Fig. 1A), demonstrating arachnoiditis with a nonenhancing, loculated cystic lesion attached to the left aspect of the cauda equina. Based on this appearance, a parasitic infection was suspected, and neurosurgical consultation was requested. The patient was taken to the operating room for removal of the structure. The cystic lesion was identified in the intradural space, and within was found a macroscopic white helminth (Fig. 1B). The exact length of the specimen could not be determined due to fragmentation during extraction but was greater than 3 cm. Gross pathology showed a helminth with pseudosegmentation evidenced by various circumferences, while microscopic specimens demonstrated a tegumental brush border, calcareous bodies, and a lack of organoid structures (Fig. 1C). With the combination of clinical presentation and pathological findings, a diagnosis of sparganosis was made. Subsequent MRI of the brain also showed evidence of cerebral and cerebellar involvement, with white matter enhancement and serpiginous tunneling (Fig. 1D). On further history, the patient acknowledged that he frequently consumed both raw frog and raw snake meat. He again denied any other neurologic symptoms apart from those mentioned previously, and there was no evidence of cerebellar or cerebral dysfunction on exam. DISCUSSIONSparganosis is a zoonotic infection caused by cestodes of the genera Spirometra and Sparganum, members of the Diphyllobothriidae family (1; DPDx, sparganosis [Centers

Epidemiology, Diagnosis, and Prevention of Sparganosis in Asia

“…Accounting for about 25% of clinical cases, cerebral sparganosis involving the CNS manifests as seizure (20%), headache (12%), altered mental functions (e.g., confusion, memory loss; 11%), hemiparesis (6.8%), motor weakness (4%), cerebral hemorrhage, and fatigue; that involving the spinal cord exhibits voiding difficulty (38%), recurrent back pain (38%), and paresis (23%); and that involving both the CNS and spinal cord causes seizure, convulsion, increased intracranial pressure, facial palsy, hearing loss, voiding difficulty, and paresis of the lower extremities. Cerebral sparganosis appears to affect more men (76%) than women (24%), and infected patients are often in their thirties (20%), forties (23%), fifties (20%), and sixties (20%) [ 21 , 22 ].…”

Cestodiasis in 2 Puerto Rican crested anoles

“…Spargana may cause skin and soft tissue swelling and urticaria, followed by CNS lesions, leading to seizures and hemiparesis. 3,4,21 Sparganosis has been reported worldwide; however, the disease is most frequently diagnosed in east Asia, particularly in China, Japan, Korea, Thailand, and Vietnam, with most cases arising in Japan. 7,10,22 Infection with adult stages of the parasite in people has also been reported in 5 cases.…”

“…Spargana may cause skin and soft tissue swelling and urticaria, followed by CNS lesions, leading to seizures and hemiparesis. 3,4,21…”