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Coluna/Columna

Print version ISSN 1808-1851

Coluna/Columna vol.10 no.2 São Paulo  2011

http://dx.doi.org/10.1590/S1808-18512011000200015 

RELATO DE CASO CASE REPORT

 

Motor tic disorder and traumatic cervical myelopathy: a case report

 

Transtorno de tique motor e mielopatia cervical traumática: relato de caso

 

Trastorno de tic motor y mielopatía cervical traumática: relato de caso

 

 

Ericson SfredoI; Felipe Martins de Lima CecchiniII; Sérgio Fernando RauppIII; Julia Bertholdo BossardiIV; Asdrubal FalavignaV

IMédico do Departamento de Neurocirurgia do Hospital Moinhos de Vento, Porto Alegre, Brasil
IIMédico do Departamento de Neurocirurgia do Hospital Municipal de Novo Hamburgo,Novo Hamburgo, Brazil
IIIProfessor de Neuroradiologia da Pontifícia Universidade do Rio Grande do Sul (PUCRS), Rio Grande do Sul, Brasil
IVAcadêmica do Curso de Medicina da Universidade de Caxias do Sul, Caxias do Sul, Brasil
VProfessor de Neurocirurgia da Universidade de Caxias do Sul, Caxias do Sul, Brasil, coordenador do curso de Medicina da Universidade de Caxias do Sul

Correspondência

 

 


ABSTRACT

The association between motor tics and cervical myelopathy is rare and not well understood. Only a few papers in the literature reported this disorder until the present date. This is a case report of a cervical myelopathy case secondary to a motor tic disorder. A 23-year-old male with a 10-year history of motor tic disorder, involving sudden forced extension of the head and cervical spine. Disturbed tactile sensation and kinetic posturing that progressed to the Lhermitte sign every time he made the movement were detected over the last six months. Magnetic resonance imaging (MRI) showed hyperintense intramedullary lesion at C2-C3, degeneration at C3-C4, and no signs of spinal cord compression. On sagittal view, functional MRI with head extension showed anterior compression with protrusion of the intervertebral disc and posterior compression of the yellow ligaments causing spinal cord stenosis. Anterior discectomy and fixation of C3-C4 were performed. There were no complications. The patient showed improvement and the motor tics were controlled by haloperidol. The patient remains symptom-free after 2 years of follow-up. Uncontrolled motor tics can compromise spinal cord function. Functional MRI can reproduce the abnormal movements and clarify the physiopathology.

Keywords: Tic disorders; Spinal cord injuries; Tics; Spinal cord compression.


RESUMO

A associação entre tiques motores e mielopatia cervical é rara e não é bem compreendida. Poucos estudos relatando esse distúrbio foram encontrados na literatura até o presente momento. Este é o relato de um caso de mielopatia cervical secundária a tiques motores. Paciente masculino de 23 anos com história de transtorno de tique motor há 10 anos, envolvendo extensão forçada da cabeça e coluna cervical. Deficiência de sensações táteis e postura cinética que progrediram para o sinal de Lhermitte, cada vez que ele se movimentava, foram diagnosticadas nos últimos seis meses. A Ressonância Magnética (RM) revelou lesão intramedular hiperintensa ao nível de C2-C3, degeneração ao nível de C3-C4 e ausência de sinais de compressão medular. Na visão sagital, a RM funcional com extensão da cabeça revelou compressão anterior com protusão do disco intervertebral e compressão posterior dos ligamentos amarelos, causando estenose medular. Foi realizada discectomia anterior e fixação de C3-C4. Não houve complicações. O paciente apresentou melhora e os tiques motores foram controlados pelo haloperidol. Após dois anos de seguimento, ele permanece livre dos sintomas e tiques motores. Tiques motores não controlados podem comprometer a medula espinhal. A ressonância magnética funcional pode reproduzir os movimentos anormais e clarear a patofisiologia.

Descritores: Transtornos de tiques; Traumatismos da medula espinhal; Tiques; Compressão da medula espinhal.


RESUMEN

La asociación entre tics motores y mielopatía cervical es rara y no es bien comprendida. Pocos estudios, que analizan este disturbio, han sido encontrados en la literatura hasta el momento. Este es el relato de un caso de mielopatía cervical secundaria a tics motores. Paciente masculino de 23 años con historial de trastorno de tic motor desde hace diez años, involucrando extensión forzada de la cabeza y columna cervical. En los últimos seis meses, se diagnosticaron deficiencias de sensaciones táctiles y postura cinética que progresaron hacia la señal de Lhermitte, cada vez que el paciente se movía. La Resonancia Magnética (RM) reveló lesión intramedular hiperintensa al nivel de C2-C3, degeneración al nivel de C3-C4, y ausencia de señales de compresión medular. En la visión sagital, la RM funcional con extensión de la cabeza reveló compresión anterior con protrusión del disco intervertebral, y compresión posterior de los ligamentos amarillos, causando estenosis medular. Se realizó discectomía anterior y fijación de C3-C4. No hubo complicaciones. El paciente presentó mejora y los tics motores fueron controlados por haloperidol. Luego de 2 años de acompañamiento, permanecía libre de los síntomas y tics motores. Tics motores no controlados pueden comprometer a la médula espinal. La resonancia magnética funcional puede reproducir los movimientos anormales y aclarar la patofisiología.

Descriptores: Trastornos de tics; Traumatismos de la médula espinal; Tics; Compresión de la médula espinal.


 

 

INTRODUCTION

Cervical myelopathy is a pathological entity that may develop as a result of movement disorders such as motor tics1. Tics are sudden, involuntary, stereotypic, repetitive, but non-rhythmic movements or vocalizations. Their prevalence ranges between 5 to 100 cases per 10,0002. Only a few papers reporting the association between motor tics and cervical myelopathy have been found in the literature1,3-10. We report a case of a cervical motor tic that caused a traumatic cervical myelopathy.

 

CASE REPORT

A 23-year-old-male patient had a 10-year history of motor tic disorder, involving rapid and abrupt movements of forced extension of the head and cervical region (DSM-IV F95.1). Movements were recurrent, arrhythmic, stereotyped, uncontrollable, and would disappear for variable periods of time.

The patient presented with numbness in the lower limbs that rapidly progressed to the upper limbs six months prior to neurological consultation. His symptoms worsened in the week before consultation, with the onset of more intense and uncontrollable tics associated with the sensation of shocks throughout his body (Lhermitte's sign). The patient was investigated by MRI that showed a hyperintense, nonexpansive, intramedullary lesion at C2 with an extension at the C2-C3 level. The intervertebral disk at C3-C4 showed signal loss and mild posterior protrusion (Figure 1). He underwent a functional MRI with cervical extension, which showed posterior spinal cord compression at C3-C4 caused by a cervical stenosis at the yellow ligament.

 

 

An anterior cervical approach with C3-C4 discectomy was performed. An iliac bone graft was placed at the intervertebral space with a C3-C4 anterior fixation (Figure 2). At the 2-year follow-up, the patient was neurologically asymptomatic without myelopathy. The motor tic disorder was controlled by haloperidol.

 

 

DISCUSSION

Tics are defined as stereotyped and repetitive movements or vocalizations11. They are classified into four categories: Tourette's syndrome (TS), defined as the presence of multiple motor tics and one or more vocal tics occurring over the span of at least 12 months; chronic tic disorder (CTD), defined as either motor or vocal tics (but not both) lasting at least 12 months; transient tic disorder, when tics are present for at least 4 weeks but not 12 months; and tic disorder not otherwise specified (NOS), diagnosed when tics are present but do not meet criteria for another tic disorder12. Studies suggested that cervical myelopathy caused by motor tics are caused by violent and frequent neck flexion or twisting and extension movements6,7. This association was usually present in youngsters with the mean interval between the onset of the tic and myelopathy 11.1 years8.

The literature contains only a few reports correlating a motor tic disorder with traumatic cervical myelopathy1,3-10 (Table 1). Nomura et al.7 reported a case of cervical myelopathy with no spondylosis or disk herniation and suggested that the lesion was due to the direct trauma caused by a neck flexion tic. Some studies found tics to be associated with cervical disk herniation and cervical spondylosis3. Fen et al.13 on the other hand, found no alteration at the neurological exam of 58 patients with Tourette's disorder. This patient underwent a functional MRI with cervical extension in order to investigate spinal cord compression, because routine exams did not reveal it.

Cervical myelopathy due to motor tics can be treated with surgery,3,4,6,8 pharmacological therapy1,5, psychotherapy5, or with botulinum toxin (BTX)9-10. In three previously reported cases, patients underwent surgical decompression but their symptoms worsened postoperatively despite treatment, and the tic remained constant in all cases4,6,10. These results probably indicate that segment stabilization is not enough to make myelopathy disappear and that treatment is necessary to control the tics. If involuntary movements persist after surgery, the risk recurrence increases; thus, pharmacologic treatment is as important as neurosurgical intervention8.

 

CONCLUSION

The association between motor tic disorder and myelopathy of the cervical spine is extremely rare and difficult to diagnose. We report a patient who underwent C3-C4 discectomy and anterior fixation and who remained symptom free at 2-year follow up with the administration of haloperidol. When there are no evident signs of compression, especially in the presence of discopathy, functional MRI is an effective method to clarify the physiopathology of the medullar lesion. Treatments must be individualized in order to achieve the best results for each patient.

 

REFERENCES

1. Lin JJ, Wang HS, Wong MC, Wu CT, Lin KL. Tourette's syndrome with cervical disc herniation. Brain Dev. 2007;29(2):61-3.         [ Links ]

2. Steinberg T, Shmuel Baruch S, Harush A, Dar R, Woods D, Piacentini J, Apter A. Tic disorders and the premonitory urge. J Neural Transm. 2010;117(2):277-284.         [ Links ]

3. Brill CB, Hartz WH, Mancall EL. Cervical disc herniation in the Gilles de la Tourette syndrome. Ann Neurol. 1981;9(3):311.         [ Links ]

4. Dobbs M, Berger JR. Cervical myelopathy secondary to violent tics of Tourette's syndrome. Neurology. 2003;60(11):1862-3.         [ Links ]

5. Goetz CG, Klawans HL. Gilles de la Tourette syndrome and compressive neuropathies. Ann Neurol. 1980;8(4):453.         [ Links ]

6. Krauss JK, Jankovic J. Severe motor tics causing cervical myelopathy in Tourette's syndrome. Mov Disord. 1996;11(5):563-6.         [ Links ]

7. Nomura T, Kira J, Yoshimura T, Goto I, Hasuo K. [Flexion myelopathy due to tic of neck]. Rinsho Shinkeigaku. 1989;29(2):177-9.         [ Links ]

8. Muroi A, Matsumura A, Asakawa H, Enomoto T, Iwasaki N, Nose T. Myelopathy caused by tics in an adolescent, associated with T2 signal intensity changes of the spinal cord. Childs Nerv Syst. 2002;18(3-4):191-4.         [ Links ]

9. Aguirregomozcorta M, Pagonabarraga J, Diaz-Manera J, Pascual-Sedano B, Gironell A, Kulisevsky J. Efficacy of botulinum toxin in severe Tourette syndrome with dystonic tics involving the neck. Parkinsonism Relat Disord. 2008;14(5):443-5.         [ Links ]

10. Adler CH, Zimmerman RS, Lyons MK, Simeone F, Brin MF. Perioperative use of botulinum toxin for movement disorder-induced cervical spine disease. Mov Disord. 1996;11(1):79-81.         [ Links ]

11. Diagnostic and statistical manual of mental disorders. 4th edition. Washington, DC: American Psychiatric Association; 2000.         [ Links ]

12. Franklin SA, Walther MR, Woods DW. Behavioral interventions for tic disorders. Psychiatr Clin North Am. 2010;33(3):641-55.         [ Links ]

13. Fen CH, Barbosa ER, Miguel EC. Síndrome de Gilles de la Tourette - Estudo clínico de 58 casos. Arq Neuropsiquiatr. 2001;59(3B):729-32.         [ Links ]

 

 

Correspondência:
Asdrubal Falavigna.
Rua: General Arcy da Rocha Nóbrega, 401 / 602
Caxias do Sul - RS - Brasil - CEP: 95040-290
E-mail asdrubalmd@gmail.com

Recebido em 20/05/2011, aceito em 26/06/2011

 

 

Trabalho realizado na Universidade de Caxias do Sul.

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