Spontaneous idiopathic acute spinal subdural hematomas are highly exceptional. Neurological symptoms are usually severe, and rapid diagnosis with MRI is mandatory. Surgical evacuation has frequently been used therapeutically; however, spontaneous recovery in mild cases has also been reported. We present a case of spontaneous recovery from severe paraparesis after spontaneous acute SSDH, and review the English-speaking literatur

Agosti, Reto

Payer, Michael

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CASE REPORTSpontaneous acute spinal subdural hematoma: spontaneousrecovery from severe paraparesis—case report and reviewMichael Payer & Reto AgostiReceived: 4 June 2010 /Accepted: 23 July 2010 /Published online: 11 August 2010# Springer-Verlag 2010Abstract Spontaneous idiopathic acute spinal subduralhematomas are highly exceptional. Neurological symptomsare usually severe, and rapid diagnosis with MRI ismandatory. Surgical evacuation has frequently been usedtherapeutically; however, spontaneous recovery in mildcases has also been reported. We present a case ofspontaneous recovery from severe paraparesis after sponta-neous acute SSDH, and review the English-speakingliterature.Keywords Spinal . Subdural hematoma . Spontaneous .ParaparesisIntroductionSpontaneous idiopathic acute spinal subdural hematomas(SSDHs) are highly exceptional. As neurological symptomsare usually severe, rapid diagnosis with MRI is mandatory[2, 9]. In therapeutic management, the main question iswhether surgical evacuation is necessary or not. We presenta case of spontaneous subtotal recovery from severeparaparesis after spontaneous acute SSDH, and review theEnglish speaking literature.Case reportA 59-year-old man, under acetylic salicylic acid andclopidrogrel after cardiac stenting, presented with sponta-neous acute left-dominant paraparesis sub T8 with anaverage muscle strength of M3 and severe sphincterdysfunction. Gadolinium-enhanced MRI revealed an acuteSSDH from T2-9 anterior to the spinal cord (Fig. 1). Nounderlying tumor or vascular malformation was seen.Because of the extensive cranio-caudal hematoma exten-sion and poor medical condition, particularly in the fear ofcausing postoperative recurrent subdural or epidural hema-toma because of acetylic salicylic acid and clopidogrel,conservative treatment was chosen under constant readinessfor surgery in case of progressive worsening. Clopidogrelwas discontinued in accordance with the treating cardiolo-gist, while aspirine was continued. Under initial bed restand steroids, leg strength fluctuated between grades M 1–3for 2 days and improved thereafter. Full muscle strengthwas regained at 1 year; ataxia, temperature/pain sensation,and urge incontinence had well improved. MRI at 5 weeksshowed hematoma resorption (Fig. 2), and at 16 weeks,cystic arachnoiditis was observed with spinal cord defor-mity and without further re-bleedings (Fig. 3).DiscussionSSDHs are uncommon and often result from major orminor spine trauma or from spine puncture including spinalanesthesia. “Spontaneous” acute SSDHs are even more rareM. Payer (*)Neurosurgery Klinik Hirslanden,Witellikerstrasse 40,8032 Zürich, Switzerlande-mail: mpayer@hotmail.comM. PayerDepartment of Neurosurgery, University Hospital of Geneva,Geneva, SwitzerlandR. AgostiKopfwehzentrum Hirslanden,Zürich, SwitzerlandActa Neurochir (2010) 152:1981–1984DOI 10.1007/s00701-010-0758-7and have mostly been observed in conjunction withcoagulopathy or anticoagulant therapy, intraspinal tumor,and vascular anomaly such as an aneurysm or duralarteriovenous fistula [3, 9, 12]. Spontaneous SSDHswithout detectable structural lesion or anticoagulation areless numerous again, and we detected 16 properly docu-mented cases in the readily available English-speakingliterature (Table 1).SSDHs are predominantly found in the thoracic spine,usually extending over several vertebral levels [2, 3, 6, 9].Severe acute local back pain with sensorimotor deficits andsphincter disturbance are the typical clinical manifestation[3, 9, 12]. MRI is the imaging of choice; it shows thehematoma location in relation to the dura and spinal cord,its cranio-caudal extension, and usually shows or rules outan underlying tumor or AVM [9, 11, 12]. Acute SSDHs(1–3 days) are isointense on T1 and mixed on T2 images; inthe subacute stage (4–7 days), they show high intensity onboth T1 and T2, and diffuse high signal is seen in thechronic stage after weeks to months [9, 11]. While epiduralhematomas are usually found posterior to the spinal cord ina lentiform shape, SSDHs are commonly found anterior-lateral to and around the spinal cord in a semi-circularpattern with compression and little displacement [3, 9, 11].Fig. 3 Sagittal T2-weighted MRI at 16 weeks after the acutebleeding, showing cystic arachnoiditis with spinal cord deformityand without further re-bleedingsFig. 2 Sagittal T2-weighted MRI at 5 weeks after the acute bleeding,showing resorption of the thoracic SSDHFig. 1 Sagittal T2- and T1-weighted MRI showing acute SSDHprimarily anterior to the spinal cord from T2–91982 Acta Neurochir (2010) 152:1981–1984Angiography has been used inconsistently to rule out AVF,AVM, or aneurysm, and was performed in six of the 16retrieved cases (Table 1). While some authors recommendspinal angiography in the presence of spontaneous SSDHwhenever available [3, 12, 15], others judge that gadolin-ium injected MRI, particularly if repeated, should showvascular malformations directly or by flow void signals [7,17]. Many papers do not discuss the indication for spinalangiography [1, 6, 9–11, 14], and it remains a case to casedecision based on availability, degree of neurosurgicalemergency, and suspicion level of vascular malformation.The pathomechanism in spontaneous idiopathic SSDHsis little understood [2, 11]. It is commonly thought thateither the hematoma originates from rupture of valvelessradiculomedullary veins in the subarachnoid space afterincreased intra-abdominal or intra-thoracic pressure (cough-ing or straining) or from minor trauma and that itsubsequently breaks through the thin arachnoid into thesubdural space [9, 12, 15]. This hypothesis could explainclinical signs of SAH in many patients with SSDH, thereported combination of SSDH and SAH, and the potentialdilution of such hematomas by the CSF [9, 12]. Conversely,SSDH has been thought to arise from the few thin, delicateextra-archnoidal vessels located on the inner dural surfaceand then breaking through the arachnoid into the subarach-noid space, and it is usually impossible to determine theorigin [3, 7, 13]. In either case, the diluting effect of theCSF prevents clot formation, unless the hematoma issufficiently large to block CSF flow [8, 12].In idiopathic SSDHs, therapy is limited to the hematomamanagement, as there is no underlying pathology to addresssurgically. Platelet dysfunction has been shown to beassociated with SSDH not only in our case but also in acases with polycythemia vera [4] and essential thrombocy-themia [10]. Discontinuation of anti-aggregating medica-tion, however, must be weighed against potentialthrombotic complications, and depends on the individualindication of such a treatment. Patients with mild symptomsTable 1 Published cases of idiopathic spontaneous acute SSDHArticle Year Age, sex Hematoma location Bleeding cause, risk factors Preop neurodeficitAngio Treatment(spinal)RecoverySwann [16] 1984 46, f(case 1)TL junction Unknown Transient mildparaparesisYes LumbarpunctureCompleterecoveryKalina [4] 1995 60, f T7-S2, anterior Unknown,polycythemia veraMild paraparesis No Conservative CompleterecoveryKang [5] 2000 49, f T5-L3, anterior Unknown Transient mildparaparesisNo Conservative CompleterecoveryKüker [11] 2000 81, m(case 4)Mid T spine Unknown Paraparesis(M3/5)No Surgery Completerecovery56, f(case 6)Thoraco-lumbar,anteriorUnknown Paraparesis(M1–3/5)Yes Surgery Good recoveryKirsch [9] 2000 47, m(case 1)T4-L5, antero-lateral Unknown Paraparesis Yes Lami T11-L1Improved42, m(case 2)CCJ–L3, around SC Unknown Paraplegia No Lami T2–5 No recovery34, m(case 3)T1–4, around SC Unknown Only pain andparesthesiaYes Conservative CompleterecoveryYamada[17]2003 38, f T1–7, anterior Unknown Mild paraparesis Yes Conservative CompleterecoveryKonitsiotis[10]2003 60, f T3-L5,anterior-lateralUnknown, essentialthrombocythaemiaOnly pain No Conservative Pain subsidedCha [1] 2005 72, f T3–6,posterior-lateralUnknown; aspirin+lowmolecular heparinParaplegia Yes Lami T3–5 No relevantrecoveryKyriakides[12]2007 44,m T2–6, anterior Unknown Paraplegia No Lami SubtotalrecoveryKim SD [6] 2008 48, f T1–4, mainly anterior Unknown, FMD Paraplegia No Lami T1–4 No recoveryOzdemir[15]2008 50, m T4–8, anterior Unknown Paraparesis(M3–4)No Lami T4–6 CompleterecoveryKakitsubata[3]2009 66, m T11/12, anterior-lateralUnknown Only pain No Conservative Pain subsidedOh [14] 2009 59, f C3–6, posterior-lateral left sideUnknown Left-sidedhemiparesisNo Conservative CompleterecoveryLami laminectomy, SC spinal cord, FMD fibromuscular dysplasiaActa Neurochir (2010) 152:1981–1984 1983or in poor general conditions should be treated conserva-tively [3, 9, 15]. There is an ongoing controversy, however,whether surgical or conservative treatment should bepreferred in severe neurological deficit. While most authorsadvocate rapid surgical evacuation via laminectomy, poste-rior dural opening, and hematoma removal in such cases [6,9, 11, 12, 15], several spontaneous recoveries have beenreported in cases with moderate neurological deficits [4, 14,17], and surgical evacuation in a large review of 106 caseswith all types of SSDHs has yielded a poor outcome in 58%[2]. Kyriakides in his review concluded that “patients withincomplete neurologic deficit tend to recover with orwithout surgery. Patients who exhibit signs of deteriorationare usually likely to benefit from percutaneous drainage orsurgery” [12]. Our current case shows that substantialneurological recovery can occur even in severe paraparesis;however, we do not conclude that conservative treatmentshould generally be chosen in severe neurological deficitwith “difficult” medical conditions. Initial conservativetreatment and close observation can be a treatment optionas has been shown previously for “mild” paraparesis andhemi-paresis [4, 14, 17]. Indeed, the current authors wereinitially not sure whether they should go for surgery in theircase, but were very reluctant to do so primarily because ofacetylic salicylic acid and clopidogrel.We agree to the hypothesis that spontaneous recoveryfrom SSDH may be explained by a progressive cranio-caudal distribution and pressure drop of the hematomawithin the subdural space on one hand and dilution of thehematoma by CSF in the presence of arachnoidal tearing onthe other hand [8, 12, 17]. The current case further confirmsthat sphincter disturbance is the least likely to be improved,as has been found before [11]. Paraplegia carries a poorprognosis even with surgery [6, 12].In conclusion, there is a general agreement that in mildsymptoms, conservative treatment is preferred in spontaneousidiopathic acute SSDH. A clear trend for surgical evacuationvia laminectomy and posterior dural opening is found in theliterature in cases of a severe neurological deficit; however,the current case demonstrates that good neurological recoverycan occur even in severe paraparesis. Patients with paraplegiaand sphincter disturbance have the poorest prognosis regard-less of surgical or conservative treatment.Conflicts of interest NoneReferences1. Cha YH, Chi JH, Barbaro NM (2005) Spontaneous spinalsubdural hematoma associated with low-molecular-weight hepa-rin. Case report. J Neurosurg Spine 2(5):612–6132. Domenicucci M, Ramieri A, Ciappetta P (1999) Nontraumaticacute spinal subdural hematoma: report of five cases and reviewof the literature. J Neurosurg 91(1 Suppl):65–733. Kakitsubata Y, Theodorou SJ, Theodorou DJ, Miyata Y, Ito Y,Yuki Y, Honbu K, Maehara T (2010) Spontaneous spinalsubarachnoid hemorrhage associated with subdural hematoma atdifferent spinal levels. Emerg Radiol 17(1):69–724. Kalina P, Drehobl KE, Black K, Woldenberg R, Sapan M (1995)Spinal cord compression by spontaneous spinal subdural haema-toma in polycythemia vera. Postgrad Med J 71(836):378–3795. Kang HS, Chung CK, Kim HJ (2000) Spontaneous spinalsubdural hematoma with spontaneous resolution. Spinal Cord 38(3):192–1966. Kim SD, Park JO, Kim SH, Lee YH, Lim DJ, Park JY (2008)Spontaneous thoracic spinal subdural hematoma associated withfibromuscular dysplasia. J Neurosurg Spine 8(5):478–4817. Kim JS, Lee SH (2009) Spontaneous spinal subarachnoidhemorrhage with spontaneous resolution. J Korean NeurosurgSoc 45(4):253–2558. Kim YH, Cho KT, Chung CK, Kim HJ (2004) Idiopathicspontaneous spinal subarachnoid hemorrhage. Spinal Cord 42(9):545–5479. Kirsch EC, Khangure MS, Holthouse D, McAuliffe W (2000)Acute spontaneous spinal subdural haematoma: MRI features.Neuroradiology 42(8):586–59010. Konitsiotis S, Glantzouni A, Argyropoulou MI, Tsapoga T, ElisafM, Efremidis SC (2003) Acute spontaneous spinal subduralhaematomas in a patient with essential thrombocythaemia. JNeurol 250(9):1109–111111. Küker W, Thiex R, Friese S, Freudenstein D, Reinges MH,Ernemann U, Kringes T, Skalej M (2000) Spinal subdural andepidural haematomas: diagnostic and therapeutic aspects in acuteand subacute cases. Acta Neurochir (Wien) 142(7):777–78512. Kyriakides AE, Lalam RK, El Masry WS (2007) Acutespontaneous spinal subdural hematoma presenting as paraplegia:a rare case. Spine 32(21):619–62213. Morandi X, Carsin-Nicol B, Brassier G, Scarabin JM (1998) MRdemonstration of spontaneous acute spinal subdural hematoma. JNeuroradiol 25(1):46–4814. Oh SH, Han IB, Koo YH, Kim OJ (2009) Acute spinal subduralhematoma presenting with spontaneously resolving hemiplegia. JKorean Neurosurg Soc 45(6):390–39315. Ozdemir O, Calisaneller T, Yildirim E, Caner H, Altinors N(2008) Acute spontaneous spinal subdural hematoma in a patientwith bilateral incarcerated inguinal hernia. Joint Bone Spine 75(3):345–34716. Swann KW, Ropper AH, New PF, Poletti CE (1984) Spontaneousspinal subarachnoid hemorrhage and subdural hematoma. Reportof two cases. J Neurosurg 61(5):975–98017. Yamada K, Nakahara T, Yamamato K, Muranaka T, Ushio Y(2003) Nontraumatic spinal subdural haematoma occurring in apostpartum period. Acta Neurochir (Wien) 145(2):151–1551984 Acta Neurochir (2010) 152:1981–1984

Spontaneous acute spinal subdural hematoma: spontaneous recovery from severe paraparesis—case report and review

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Spontaneous acute spinal subdural hematoma: spontaneous recovery from severe paraparesis—case report and review

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