Beck, Bernhard

Buck, Alfred

Gonzenbach, Roman

Kong, Yoon

Semmler, Alexander

Weller, Michael

RERO DOC Digital Library

LETTER TO THE EDITORSHigh-dose praziquantel therapy for cerebral sparganosisRoman R. Gonzenbach • Yoon Kong •Bernhard Beck • Alfred Buck • Michael Weller •Alexander SemmlerReceived: 22 January 2013 / Revised: 18 March 2013 / Accepted: 20 March 2013 / Published online: 2 April 2013 Springer-Verlag Berlin Heidelberg 2013Dear Sirs,A 39-year-old Bangladeshi man was admitted to our hos-pital with a first generalized epileptic seizure. Brain MRIshowed multiple ring-enhancing lesions with perifocaledema (Fig. 1a and g, February 2008). CSF analysisshowed 8 cells/ll and normal protein, glucose and lactatelevels. Serum and CSF serological tests were negative for abroad range of parasites, fungi and bacteria.The patient was suspected to have neurocysticercosisand was treated with albendazole (400 mg bi-daily for30 days). However, follow up MRI showed new, adjacentlesions with ring- and tunnel-shaped enhancement; andpunctate calcifications in the left parieto-occipital area inthe CT scan (Fig. 1b, h and m, December 2009), suggestingthat the treatment had been ineffective. We therefore per-formed a brain biopsy (with prior MRI for the localizationof the sparganum, see Fig. 1c and i, February 2010), whichrevealed a degenerated cyst containing membrane-shapedstructures with multiple foci of calcification. PCR waspositive for Spirometra erinaceieuropaei, leading to thediagnosis of cerebral sparganosis.Surgical removal of the parasite is the treatment ofchoice as standard anti-helmintic therapy is consideredineffective in sparganosis [1–3]. The repeated MRI indi-cated that the lesion had been wandering again (Fig. 1d andj). An ethylcholine PET showed a defined region withincreased tracer uptake, most likely indicating the currentlocation of the sparganum (Fig. 1n). However, the patientrefused the recommended stereotactic surgery. We there-fore treated the patient with a high-dose regimen of pra-ziquantel (3 9 25 mg/kg body weight daily) for 7 dayscombined with cimetidine (3 9 400 mg daily) and a highcarbohydrate diet to increase plasma levels of praziquantel[4]. The treatment was well-tolerated.Follow-up MRIs 3 and 11 months later showed drasticimprovement and the ethylcholine PET had normalized(Fig. 1e, f, k, l, o and p). Anti-sparganum antibody levelswere determined from preserved CSF and serum samplesby a specific ELISA [5]. Before therapy (Fig. 1, December2009), anti-sparganum antibody levels were elevated in theserum (0.29) and in the CSF (0.55 normal value in serumand CSF \ 0.22). After therapy (Fig. 1, June 2011), anti-body levels had dropped to 0.09 in serum (a lumbarpuncture was not repeated after therapy). Seizures dis-continued and anticonvulsive treatment was slowlytapered.Sparganosis is a rare parasitic disease caused by infes-tation with the larval cestode of Spirometra spp. Most caseshave been reported in Southeast and Eastern Asia. Humansare infected by drinking unfiltered, contaminated water orthrough consumption of raw snakes and frogs. When theparasite invades the central nervous system, epileptic sei-zures and headache are common symptoms [2]. Charac-teristic MRI findings of cerebral sparganosis include aR. R. Gonzenbach (&)  M. Weller  A. SemmlerDepartment of Neurology, University Hospital Zurich,Frauenklinikstrasse 26, 8091 Zurich, Switzerlande-mail: roman.gonzenbach@usz.chY. KongDepartment of Molecular Parasitology, School of Medicine,Sungkyunkwan University, Seoul, KoreaB. BeckDepartment of Infectiology, University Hospital, Zurich,Zu¨rich, SwitzerlandA. BuckDepartment of Radiology, University Hospital, Zurich,Zu¨rich, Switzerland123J Neurol (2013) 260:1423–1425DOI 10.1007/s00415-013-6901-7‘tunnel sign’, and ‘wandering lesions’ on successive ima-ges representing the track of motion of the parasite [6, 7].The treatment of choice for cerebral sparganosis issurgical removal because treatment with anti-helminthics,including praziquantel, has been described as not effective[1–3]. In our patient the clinical improvement, and thenormalization of brain imaging and of anti-sparganumantibody levels suggest that the parasite was eradicated byhigh-dose praziquantel therapy. Although the close tem-poral correlation of therapy and presumed death of theparasite suggests a causal relationship we cannot rule outthe possibility that this was, in fact, due to a coinciding,natural death of the parasite and not due to the chemo-therapeutics. There is no information regarding the ces-tode’s life expectancy in human tissue in the literature.Cerebral sparganosis should be suspected in patientsfrom endemic areas with indications of cerebral parasiticinfection and ‘‘wandering lesions’’ in the MRI. High-dosepraziquantel treatment may be considered in inoperablecases of cerebral sparganosis. Ethylcholine PET can beused preoperatively for the precise localization of thesparganum. This should be particularly valuable if theprecise location is not known due to the often multifocallesions in MRI.Conflicts of interest On behalf of all authors, the correspondingauthor declares no conflict of interest.References1. Deng L, Xiong P, Qian S (2011) Diagnosis and stereotacticaspiration treatment of cerebral sparganosis: summary of 11 cases.J Neurosurg 114(5):1421–14252. Kim DG, Paek SH, Chang KH, Wang KC, Jung HW, Kim HJ, ChiJG, Choi KS, Han DH (1996) Cerebral sparganosis: clinicalmanifestations, treatment, and outcome. J Neurosurg 85(6):1066–10713. Torres JR, Noya OO, Noya BA, Mouliniere R, Martinez E (1981)Treatment of proliferative sparganosis with mebendazole andpraziquantel. Trans R Soc Trop Med Hyg 75(6):846–8474. Sotelo J, Jung H (1998) Pharmacokinetic optimisation of thetreatment of neurocysticercosis. Clin Pharmacokinet 34(6):503–515Fig. 1 Radiology findings. The upper and middle row show axialbrain MRIs in chronological order. The upper row shows contrast-enhanced horizontal T1-weighted sections (a–f). The middle rowshows the corresponding T2 sections (g–l). Characteristic punctatecalcifications in the CT in the affected region are shown in (m).Repeated ethylcholine PET scans before and 3 and 11 months afterpraziquantel therapy are shown in (n–p). The relative amount oftracer uptake is color-coded with arbitrary units1424 J Neurol (2013) 260:1423–14251235. Kong Y, Kang SY, Cho SY (1991) Single step purification ofpotent antigenic protein from sparganum by gelatin-affinitychromatography. Kisaengchunghak Chapchi 29(1):1–76. Song T, Wang WS, Zhou BR, Mai WW, Li ZZ, Guo HC, Zhou F(2007) CT and MR characteristics of cerebral sparganosis. AJNRAm J Neuroradiol 28(9):1700–17057. Chang KH, Chi JG, Cho SY, Han MH, Han DH, Han MC (1992)Cerebral sparganosis: analysis of 34 cases with emphasis on CTfeatures. Neuroradiology 34(1):1–8J Neurol (2013) 260:1423–1425 1425123

High-dose praziquantel therapy for cerebral sparganosis

http://doc.rero.ch/record/319752/files/415_2013_Article_6901.pdf

High-dose praziquantel therapy for cerebral sparganosis

Abstract

Similar works

Full text

Available Versions

RERO DOC Digital Library