Bogousslavsky, J.

Kuntzer, Th

Qanadli, S.

Vingerhoets, F.

Von Segesser, L.-K

Wider, Ch

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J Neurol (2006) 253 : 809–810DOI 10.1007/s00415-006-0083-5Christian WiderThierry KuntzerLudwig-Karl Von SegesserSalah Dine QanadliJulien BogousslavskyFrançois VingerhoetsBilateral compressivelumbosacral plexopathydue to internal iliac arteryaneurysmsReceived: 16 August 2005Received in revised form: 19 October 2005Accepted: 7 November 2005Published online: 7 February 2006Sirs: A 75-year-old man was re-ferred to our outpatient clinic forprogressive walking difficulties for1 year, with lightning pain andtingling in both legs. Thirteenyears earlier, he had presented witha right parietal spontaneous brainhaemorrhage, with mild residualleft hemisyndrome.On examination, a left-predomi-nant bilateral distal leg weaknesswas found. Ankle reflexes wereabsent, patellar reflexes were weak.There was partial sensory loss forall modalities under the knee witha left predominance. Stance andgait were on a broad base,Romberg’s sign was positive. Gaitwas with irregular steps andmarked instability. Upper limbexamination was normal. Both feetwere pulseless.Nerve conduction study re-vealed normal motor and sensoryparameters in the median andulnar nerves. In the lower limbs,the compound muscle actionpotentials (CMAPs) amplitude wasseverely reduced following stimula-tion of the peroneal and tibialnerves. The sural and peronealsensory nerve action potentialswere absent. Lumbar punctureshowed elevated protein level(504 mg/l, normal less than460 mg/l) and a normal cell count.Blood tests comprising full bloodcount, liver and kidney tests, sedi-mentation rate, several antibodies(rheumatoid factor, antinuclearantibodies, ANCA, anti-Hu, anti-Yoand anti-Ri), protein electrophore-sis and vitamins were unremar-kable. Anti-GM1 IgG antibodiestitre was found to be as high as12,582 U (normal less than 800 U).Abdominal CT revealed bilateralpartially thrombosed giant internaliliac artery aneurysms (see Fig.).Surgical aneurysmorrhaphy wasperformed; the left aneurysm wasremoved, and the right one wasexcluded from systemic blood flow.Three months later, the patientcould again walk as he did beforethe operation, had no more pain,and anti-GM1 antibody titre hadfallen to 6,001 U. Six months afterthe operation, the patient couldwalk for 20 minutes without help,and neurological examinationshowed improvement in the senso-rimotor deficit and in balance andgait stability.Two years after surgery, hiscondition had remained stable,and anti-GM1 antibodies were nolonger detectable.Our patient presented withbilateral lumbosacral plexopathyassociated with partially throm-bosed giant aneurysms of bothLETTER TO THE EDITORSJON 2083Fig. Multi-slice CT scan after IV iodinated contrast material injection. Axial transverse image (top) showing bi-lateral partially thrombosed giant internal iliac artery aneurysms (), the left one with a 6.7cm diameter and a4.2cm thrombus, and the right one (actually located at the beginning of the obturator artery) with sacral boneerosion (arrows). Coronal (left) and sagittal (right) reconstructions showing sacral bone erosion by aneurismalmasses (arrows) (bottom)809_810_Wider_JON_2083  22.06.2006  8:02 Uhr  Seite 809810internal iliac arteries. This associa-tion suggests an unusual mecha-nism of plexus nerves damage bydirect compression, which isstrongly supported by the tightcontact between aneurysms andnerves, along with the eroded as-pect of the sacral bone (see Fig.).Unilateral lumbosacral plexuscompression has been exception-ally described in aneurysms of thecommon iliac artery [6] and ofthe internal iliac artery [4], in apseudo-aneurysm following renaltransplantation [8], and in a fewmore cases involving the internaliliac [2, 5] or gluteal arteries [10].Although nerves are ratherresistant to ischaemia and bloodsupply to the lumbosacral plexus is richly anastomosed, ischaemiamight have played an additionalrole, through the combination ofoccluded internal iliac arterybranches and direct mass effect,the latter including secondary vasanervorum compression. Anatomi-cally, along with the deep iliaccircumflex and the lumbar seg-mental arteries, the internal iliacartery plays a major role in theblood supply of the lumbar andsacral plexuses, mainly through theiliolumbar, lateral sacral, and supe-rior and inferior gluteal arteries[3]. Chronic ischaemic plexopathycan occur with aortic occlusion,and in stenosis and occlusion ofiliac arteries. Acute lumbosacralplexopathies may be encounteredas a rare complication of aorto-iliac surgery, and in aortic dissec-tion.Alternate diagnoses in our casecomprise polyradiculopathy, othercauses of plexopathy and asymmet-ric axonal polyneuropathy thatwould have incidentally revealedthe aneurysms. However, investiga-tions clearly pointed to lum-bosacral plexopathy, for whichusual causes were reasonablyexcluded.High anti-GM1 IgG antibodiestitres are reported in lower motorneuron diseases, in axonal Guil-lain-Barré syndrome, and in senso-rimotor neuropathy of undeter-mined origin [1, 7, 9]. In our casethey were thought to reflect pe-ripheral nerve damage, becauseclinical improvement was corre-lated to the fall in their serum titresafter surgery.Our patient presented with anunusual cause of bilateral lum-bosacral plexopathy that wastreated by decompression, andwould have been potentially lethalif missed.References1. Adams D, Kuntzer T, Burger D, Chof-flon M, Magistris MR, Regli F, Steck AJ(1991) Predictive value of anti-GM1ganglioside antibodies in neuromus-cular diseases: a study of 180 sera. JNeuroimmunol 32:223–2302. al Asadi AD, le Fur R, al Amri A (1995)Motor deficit complicating an isolatedaneurysm of the internal iliac artery. JChir (Paris) 132:361–3633. Day MH (1964) The blood supply ofthe lumbar and sacral plexuses in thehuman foetus. J Anat 98:104–1164. Delgado-Garcia F, Lopez-DominguezJM, Casado-Chocan JL, Blanco-OlleroA, Robledo-Strauss A, Mendez-SangilB, Diaz-Espejo C (1999) Lumbosacralplexopathy as a form of presentationof an aneurysm of the iliac artery. RevNeurol (Paris) 28:1072–10745. Eekhof JL, Ansink BJ, Davies GE (1990)Iliac aneurysms. J Neurol NeurosurgPsychiatry 53:9276. Giraldi C, Merlini V, Mosca F, Ferrari M(1984) Compression of the lum-bosacral plexus in a case of volumi-nous aneurysm of the common iliacartery. Riv Neurol (Paris) 54:399–4047. Kornberg AJ (2000) Anti-GM1 ganglio-side antibodies: their role in the diag-nosis and pathogenesis of immune-mediated motor neuropathies. J ClinNeurosci 7:191–1948. Luzzio CC, Waclawik AJ, Gallagher CL,Knechtle SJ (1999) Iliac arterypseudoaneurysm following renaltransplantation presenting as lum-bosacral plexopathy. Transplantation67:1077–10789. McCombe PA, Wilson R, Prentice RL(2000) Results of testing for anti-GM1antibodies. J Clin Neurosci 7:209–21210. Yurtseven T, Zileli M, Goker EN,Tavmergen E, Hoscoskun C, Parildar M(2002) Gluteal artery pseudoaneurysm,a rare cause of sciatic pain: case reportand literature review. J Spinal DisordTech 15:330–333Ch. Wider, MD () · Th. Kuntzer, MD · J. Bogousslavsky, MD · F. Vingerhoets, MDService de NeurologieCHUV1011 Lausanne, SwitzerlandTel.: +41-21/3141111Fax: +41-21/3141244E-Mail: christian.wider@chuv.chL.-K. Von Segesser, MDCardiovascular Surgery DepartmentCentre Hospitalier Universitaire VaudoisRue du Bugnon1011 Lausanne, SwitzerlandS. D. Qanadli, MD, PhDRadiology DepartmentCentre Hospitalier Universitaire VaudoisRue du Bugnon1011 Lausanne, Switzerland809_810_Wider_JON_2083  22.06.2006  8:02 Uhr  Seite 810

Bilateral compressive lumbosacral plexopathy due to internal iliac artery aneurysms

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Bilateral compressive lumbosacral plexopathy due to internal iliac artery aneurysms

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