- Department of Neurosurgery, Henry Ford Health System, Detroit, Michigan, USA
Department of Neurosurgery, Henry Ford Health System, Detroit, Michigan, USA
DOI:10.4103/sni.sni_234_17Copyright: © 2018 Surgical Neurology International This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Karam Asmaro, Aqueel H. Pabaney, Richard Rammo, Rizwan Tahir, Max K. Kole. Man-in-the-barrel syndrome: Case report of ventral epidural abscess and review of the literature. 16-Jan-2018;9:8
How to cite this URL: Karam Asmaro, Aqueel H. Pabaney, Richard Rammo, Rizwan Tahir, Max K. Kole. Man-in-the-barrel syndrome: Case report of ventral epidural abscess and review of the literature. 16-Jan-2018;9:8. Available from: http://surgicalneurologyint.com/surgicalint-articles/man%e2%80%91in%e2%80%91the%e2%80%91barrel-syndrome-case-report-of-ventral-epidural-abscess-and-review-of-the-literature/
Background:Man-in-the-barrel syndrome (MBS) is an uncommon clinical condition for which patients present with bilateral brachial diplegia but intact lower extremity strength. This syndrome is typically attributed to a cranial/cortical injury rather than a spinal pathology.
Case Description:A 62-year-old diabetic male presented with bilateral upper extremity paresis attributed to a ventral cervical epidural abscess diagnosed on magnetic resonance imaging. Emergent cervical decompression resulted in slight improvement of upper extremity strength. However, he later expired due to sepsis and respiratory compromise.
Conclusion:Establishing the correct diagnosis via clinical examination and proceeding with appropriate management of MBS attributed to a cervical epidural abscess is critical to achieve a good outcome.
Keywords: Brachial diplegia, man-in-a-barrel syndrome, ventral cervical epidural abscess
First reported by Mohr et al. in 1969, man-in-the-barrel syndrome (MBS) is uncommon and characterized by bilateral brachial diplegia but intact lower extremity function.[
A 62-year-old diabetic male presented with left upper extremity weakness. Two days after admission, he developed proximal bilateral upper extremity motor deficits but intact lower extremity strength. Motor compromise was characterized by shoulder abductors 1/5; elbow flexors 1/5; elbow extensors 3/5; wrist extensors 4+/5; finger flexors; and abductors 4+/5 (Medical Research Council grading scale) bilaterally. Deep tendon reflexes were 1+ in both upper and lower extremities, and were accompanied by bilateral Babinski's and Hoffmann's signs without clonus. He also complained of and demonstrated focal tenderness in the posterior cervicothoracic spine.
Laboratory and radiographic evaluation of cervical epidural abscess
Laboratory examination showed leukocytosis (24.8 K/uL), elevated C-reactive protein (38.4 mg/dL), and an elevated erythrocyte sedimentation rate (106 mm/h). The magnetic resonance image (MRI) of the cervical spine showed a ventral cervical epidural abscess extending from C2 to C6 [
(a) Axial MRI cervical spine T1 with contrast showing a contrast-enhancing ventral epidural mass consistent with an abscess or phlegmon. There are no subacute or chronic intramedullary changes in the cervical cord that would otherwise suggest a chronic pathology. (b) Sagittal MRI cervical spine T1 with contrast showing a ring-enhancing ventral epidural mass from C3–T1 consistent with abscess or phlegmon. The ventral epidural mass exhibits mass effect on the cervical cord with effacement of the dura
Postoperatively, the patient exhibited slight improvement in his proximal upper extremity motor strength; 2/5 in the shoulder abductors and elbow flexors. Surgical cultures identified: Streptococus anginosus, S. constellatus, and Staphylococcus epidermidis (methicillin sensitive). The initial broad-spectrum antibiotic therapy was narrowed down to intravenous cefazolin as recommended by the infectious disease team for a total of 6 weeks. Unfortunately, the patient became hemodynamically unstable due to sepsis/aspiration pneumonia on postoperative day 9 and expired.
Three clinical syndromes describe isolated upper extremity weakness; cruciate paralysis of Bell, MBS, and anterior spinal artery (ASA) syndrome. In each of these syndromes, the symptoms are largely permanent and irreversible.
The mechanism of neurological injury varies according to the underlying pathology. ASA syndrome and cruciate paralysis of Bell are typically related to vascular and cervical cord injuries.[
There are less than 10 cases of MBS reported due to spinal cord pathology [
In the case presented here, MBS was caused by a cervical spinal epidural abscess (SEA). The pathophysiology resulting in MBS deficit included both compressive trauma and vascular ischemia. On the other hand, most patients with spinal MBS have vascular dissection, thromboembolism, or spondylosis (e.g., as noted here). Although most patients with spinal MBS do well, our patient expired from pneumonia/sepsis.
Our treatment rationale was focused on the goal of rapid cord decompression along with source control and reducing the infectious burden. We elected instrumented fusion given the multilevel laminectomy, which was concerning for mechanical instability and delayed spinal cord injury through loss of the posterior tension band and resultant kyphosis of the cervical spine. The use of metal in vertebral osteomyelitis and nonsterile spinal procedures is controversial. In the past decade, increasing research on the subject of the biocompatibility of metallic implants in the setting of pyogenic vertebral infections have led to increased use where it has been considered as a safe option.[
Here, we reported the case of a 62-year-old male who presented with MBS secondary to a ventral cervical epidural abscess. Despite prompt intervention, the patient died due to medical comorbidities.
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1. Ben Sassi S, El Euch G, Regaieg A, Mabrouk T, Zouari M, Ben Romdhane N. Man-in-the-barrel syndrome with combination of infarctions in the anterior spinal artery and posterior inferior cerebellar artery territories. Cerebrovasc Dis. 2009. 27: 201-2
2. Berg D, Müllges W, Koltzenburg M, Bendszus M, Reiners K. Man-in-the-barrel syndrome caused by cervical spinal cord infarction. Acta Neurol Scand. 1998. 97: 417-9
3. Chen W, Jiang L, Dai L. Surgical treatment of pyogenic vertebral osteomyelitis with spinal instrumentation. Eur Spine J. 2007. 16: 1307-16
4. Renard JF, Massardier E, Iasci L, Onnient Y, Miret N, Callonnec F. Brachial diplegia caused by cervical spinal cord ischemia. A case. Rev Neurol (Paris). 1997. 153: 690-3
5. Rodríguez-Vico JS, Berrocal-Izquierdo N, Duarte J. Cervical spine myelopathy as a cause of man-in-the-barrel syndrome. Rev Neurol. 2010. 51: 701-3
6. Sage JI, Van Uitert RL. Man-in-the-barrel syndrome. Neurology. 1986. 36: 1102-3
7. Soubrier M, Demarquilly F, Urosevic Z, Zbadi K, Dubost JJ, Risotri JM. Cervical epidural infection. Four case-reports. Rev Rhum Engl Ed. 1995. 62: 29-34
8. Strupp M, Brückmann H, Hamann GF, Brüning R, Brandt T. Simultaneous brachial diplegia and rotational vertigo due to combined spinal anterior and vertebrobasilar embolism. Eur Neurol. 2000. 43: 240-2
9. Yoon B, Juk Kim J, Ha DH. A Case of Man-in-the-Barrel Syndrome Induced by Cervical Spinal Cord Ischemia. J Clin Neurophysiol. 2013. 15: 59-62