14 - review diastematomielia cervical associada a lipoma e

Case Report

J Bras Neurocirur 31(4): 378-385, 2020Gonçalves DB, Falcão MGVG, Cabral LCR, Santos MIA, Silva HTF, et al. - Diastematomielia Cervical Associada a Lipoma e Hipotrofia de Membros Superiores: relato de caso e revisão da literatura

Diastematomielia Cervical Associada a Lipoma e Hipotrofia de Membros Superiores: relato de caso e revisão da literatura Cervical Diastematomyelia Associated With Lipoma And Superior Limbs Hipotrophy: report case and review of literature

Daniel Buzaglo Gonçalves1

Marcos Gabriel do Vale Gomes Falcão2

Lucas de Cristo Rojas Cabral1

Maria Izabel Andrade dos Santos1

Henzo Theodoro Fonseca Silva1

Louise Makarem Oliveira1

Stephanie Ramos3

Rodrigo Viana3

Gabriela Campos da Silva Coutinho1

Moyses Cohen4

Franklin Reis5

Robson Amorim4,6

ABSTRACTIntroduction: Diastematomyelia is a hidden congenital spine dysraphism (HSD) first described in 1837 and represents 3.8% of all spinal dysraphisms. Few cases of cervical cord diastematomyelia have been reported since the condition’s main location is low-thoracic, conus medullaris. In this paper, we performed a literature review and present a rare case of cervical diastematomyelia associated with lipoma and superior limbs hypotrophy. Case presentation: A 48-year-old woman presented at our outpatient clinic complaining that two years before she had noticed the onset of right-hand atrophy, which evolved to the contralateral side after six months. In the intraoperative period, the dural membrane was opened, and a yellow lesion was found, reinforcing the suggestive diagnosis of lipoma. After removal, the splitted spinal cord became clear. Discussion: To our knowledge, this is the first case of diastematomyelia associated with lipoma in the cervical region. Since 1985, only 23 adult patients diagnosed using computed tomography or MRI were treated surgically. It is possible to divide spinal cord malformation (SCM) patients into two groups. One group of patients diagnosed in the first years of life and with remaining symptoms and another group formed by those diagnosed only in adulthood. Our patient is in the second group.

Keywords: Cervical diastematomyelia; Spinal cord malformation; Hidden spine dysraphism

RESUMOIntrodução: Diastematomielia é um disrafismo espinhal oculto (HSD), descrito pela primeira vez em 1837 e representa 3,8% de todos os HSD. Poucos casos de diastematomielia cervical foram relatados, uma vez que a localização principal da doença é a coluna torácica baixa, no cone medular. Neste artigo, fizemos uma revisão da literatura e descrevemos um caso raro de diastematomielia cervical associada a lipoma e hipotrofia de membros superiores. Relato do caso: Uma mulher de 48 anos apresentou-se em nosso ambulatório com queixa de atrofia da porção distal do membro superior direito, que evoluiu para o lado contralateral após seis meses. No intraoperatório, a membrana epidural foi aberta, sendo encontrada uma lesão amarelada, reforçando o diagnóstico sugestivo de lipoma. Após a remoção, a medula espinhal dividida tornou-se clara. Discussão: Segundo nossa pesquisa, este é o primeiro caso de lipoma associado a diastematomielia na região cervical. Desde 1985, apenas 23 pacientes adultos foram diagnosticados com exames de imagem e tratados cirurgicamente. É possível dividir os pacientes com malformação da medula espinhal (SCM) em dois grupos.


Case Report

Diastematomyelia is a hidden congenital spine dysraphism (HSD) first described by Olivier1, in 1837, and represents 3,8% of all spinal dysraphisms. It is characterized by a longitudinal division of the spinal cord into two parallel portions with a fibrocartilaginous or bone septum1-4. These malformations are due to failure in closing the neurenteric channel. Two different forms of diastematomyelia have been described1: type 1 with two dural tubes, and type 2 with only one dural tube. There is a close relationship with such malformation and abnormalities of the adjacent spine and skin2,3. The prevalence is higher in females, and symptoms occur more frequently in children3,5. Diastematomyelia is often underdiagnosed, but the diagnostic rate in adults has been increasing since MRI for spinal complaints investigation was introduced6.

Few cases of cervical cord diastematomyelia have been reported since the main location is low-thoracic, conus medullaris2-4. Conti et al. (2010)6 performed a literature review, aiming to find case reports of diastematomyelia and spinal cord lesions simultaneously. From 1951 to 2009, 15 cases were reported, and one lipoma was found in a 53-year-old patient at L3-L4. Clinical presentation varies. Intermittent pain associated with progressive weakness, gait disturbance, lower limb sensory symptoms, and autonomic disorders are frequently observed. After a literature search between 1980 and 2020, only one case of concomitant lipoma and HSD

was noticed (lumbar area)7. No cases of cervical lipoma and diastematomyelia were found1,8.

In this article, we perform a literature review and present a rare case of cervical diastematomyelia associated with lipoma and superior limbs hypotrophy.

A literature review was performed from 1980 to January 2020 in the MedLine database. The literature review was performed crossing the following keywords: “adult” AND “diastematomyelia” OR “split cord” OR “tethered cord”. The inclusion criteria were case report or case series of adult patients sustaining diastematomyelia.

A 48-year-old woman presented at our outpatient clinic complaining that two years before, she had noticed the onset of right-hand atrophy, which evolved to the contralateral side after six months. Shortly after, she noticed the sensation of numbness in her fingertips, ultimately comprising the elbow (lateral and medial face). She also mentioned weakness during daily activities performance. Throughout the last year, she has

INTRODUCTION

METHODS

Um grupo de pacientes diagnosticados nos primeiros anos de vida com sintomas remanescentes e outro grupo formado por aqueles diagnosticados na idade adulta. Nosso paciente encontra-se no segundo grupo.

Palavras-chave: Diastematomielia cervical; Malformação medular; Disrafismo espinhal oculto

1 Medical student, Federal University of Amazonas (UFAM), Manaus, AM, Brazil2 Medical student, Universidade do Estado do Amazonas (UEA) , Manaus, AM, Brazil3 MD, MR, Medical Residency in Neurosurgery, Hospital Universitário Getúlio Vargas (HUGV), Manaus, AM, Brazil4 Neurosurgeon at Hospital Universitário Getúlio Vargas (HUGV) , Manaus, AM, Brazil5 Professor of Fundamentals of Pain, Faculdade Metropolitana de Manaus (FAMETRO) , Manaus, AM, Brazil6 Department of Neurology, University of Sao Paulo (USP), Sao Paulo, SP

Received Jan 29, 2021Corrected Feb 10, 2021Accepted Jun 15, 2021

CASE PRESENTATION

Case Report

J Bras Neurocirur 31(4): 378-385, 2020Gonçalves DB, Falcao MGVG, Cabral LCR, Santos MIA, Silva HTF, et al. - Diastematomielia Cervical Associada a Lipoma e Hipotrofi a de Membros Superiores: relato de caso e revisão da literatura

been progressively worsening of her arm strength, losing the ability to do ordinary activities such as raising a cooking pan and preparing a fi sh.

Initial neurological examination revealed hypoesthesia in the C5 to T1 segments, bilateral tenar region hypotrophy (more severe in the left hand), left upper limb paresis (grade 4 strength), hypoactive and symmetrical refl exes in the upper limb (1+) and absent Hoff man sign bilaterally.

Imaging exams included magnetic resonance imaging (MRI) that showed a well-defi ned intradural expansive lesion at C5-T1 level. Th e lesion had signal intensity similar to fat, displaying no signifi cant contrast enhancement. It was associated with a suspicious splitted spinal cord (Figure 1a and 1b).

Th e patient was referred to our Neurosurgery service for evaluation. Our pre-operative hypothesis was an intradural lipoma.

In the intraoperative period, the dural membrane was opened, and a yellowish lesion was found, reinforcing the suggestive diagnosis of lipoma. Th e lesion was carefully dissected, and a gross total resection was performed. Aft er removal, the splitted spinal cord became clear. An osteocartilaginous membrane divided the organ, characterizing the diastematomyelia (Figure 2). Th e histopathological examination confi rmed the hypothesis of lipoma, and postoperative MRI showed complete resection (Figure 3).

Literature ReviewTh e literature search retrieved 26 articles. Sixty-fi ve were excluded because they were not related to the inclusion criteria. Th ere were just 92 papers published. All were case reports or case series (Table 1). From 27 patients: 6 patients were submitted to surgery due to tethered spinal cord; 2 patients were submitted to surgery due to spondylolisthesis; 2 patients were submitted to surgery due to arachnoid cysts; 1 patient was submitted to surgery due to a lipoma; 6 patients were submitted to surgery due to teratomas; 2 patients was submitted to surgery due to arachnoid cysts; 2 patients were submitted to surgery due to vertebral body compressions; 1 patient was submitted to surgery due to a neurenteric cyst;

and 1 patient was submitted to surgery due to a spinal stenosis. Additional spinal malformation in the lumbar region was found in 18 patients. Only 1 patient had a lipoma associated.

To our knowledge, this is the fi rst case of diastematomyelia associated with lipoma in the cervical region. Although being a rare but well-described disorder in children, diastematomyelia or split cord malformation (SCM) is not seldomly underdiagnosed in adulthood. Since 1985, only 23

Figure 2. A. Intraoperative aspect of the cervical limoma. B. Splitted spinal cord after removal of the lipoma.

17

18

Figure 1. A. MRI, non-contrast T1 sequence depicting hyperintense lesion at C6-T1. B. Pre-operative MRI in T2 sequence showing the

a suspicious splitted spinal cord

Figure 2. A. Intraoperative aspect of the cervical lipoma. B. Splitted spinal cord after removal of the lipoma.

DISCUSSION


Case Report

Table 1. Case reports of diastematomyelia and associated lesions.

adult patients diagnosed using computed tomography or MRI were treated surgically9-12. The preoperative status, surgical procedures, and results are summarized (Table 1), and the data from our patient was also included. Patients were followed for an average of 16.6 months (range 0.3-66). For those who were initially symptomatic, in the postoperative period, 100% showed improvement in arm pain, 75% had reduced back pain, 75% reduced leg pain, 83% improved motor weakness, 50% experienced sensory improvement, and 50% improved bowel or bladder dysfunction.

Most patients had the SCM at the lumbar spine level, validating previous data from other published studies2-4. Our patient had a rare presentation of the diastematomyelia and a lipoma producing the weakness symptoms.

Our literature search found one single case of adulthood diastematomyelia and a lipoma between 1985 and 20206,7. However, the lesion was located at the lumbar spine.

Karim Ahmed et al.13 reported that in a cohort in adults, the following body segments were the most common sites: lumbar

spine (51.9%), lumbosacral spine (16.9%), and thoracic spine (13.2%). He also reported that the most common symptoms were neck or back pain (68.5%). Another typical finding is scoliosis, not observed in our patient 14-15. The prevalence of SCM was calculated at 16% within a group of 251 patients with congenital scoliosis, by McMaster et al.12.

It is possible to divide SCM patients into two groups. One group of patients was diagnosed in the first years of life and with remaining symptoms, and the other group formed by those diagnosed only in adulthood. Our patient is in the second group. During the investigation of the symptoms, most complaints were related to other concurrent diseases16,17. In most cases, no surgical intervention is aimed at the distematomyelia, but to these other malformations or diseases.

The location of SCM was predominant in the lumbar spine. Only one case was found in the literature review using the methods previously described, located in the lumbar spine (Table 1). All patients underwent surgical procedures with success in most cases, improving or even eliminating preoperative symptoms.

Table 1. Case reports of diastematomyelia and associated lesions.

Authors Age of diagnosis at RCVS

Sex Additional neurologic symptoms

Localization of the diastematomyelia

Additional lesions/malformations

Surgery (surgical indication)

Starnoni et al.

(2017)7

47 M Back pain radiating to

both legs

Lumbar spine Lipoma Yes (lipoma)

Davanzo et al.

(2016)8

43 M Lower thoracic and

lumbar spine pain.

Atrophy of the left calf

with some plantar

flexion weakness of the

left foot

Lumbar spine Grade 2

spondylolisthesis of the

L5–S1 space

Yes

(spondylolisthesis)

Viswanathan et

al. (2018)10

67 F Lower back and right-

sided L5 distribution

radicular leg pain

Lumbar spine Tethered spinal cord

and a non-rigid septum

at L4

Yes (tethered spinal

cord)

Viswanathan et

al. (2018)10

53 F Low back pain and leg

weakness and

numbness

Lumbar spine Type 1 Spinal Cord

Malformation (SCM) at

L1-L2 with a midline

osseous spur and low-

lying conus at L3

Yes (tethered spinal

cord)

11

Case Report


Pallatroni et al.

(2004)11

78 F Progressive

equinovarus foot

deformity, back pain, leg

weakness and bladder

dysfunction

Lumbar spine Tethered spinal cord No (Age)

Patankar

(2019)12

30 F Left lower limb

weakness

Lumbar spine Cystic lesion at T12-L1 Yes (arachnoid cyst)

Sack et al.

(2016)14

29 F No neurological defcits Thoraco-lumbar

junction

Spina bifida occulta Not mentioned

Skalej et al.

(1999)16

48 F Incomplete monoparesis

of the left leg

Thoracic spine Syringomyelia and

hydromyelia

Yes (bone spur of

the

diastematomyelia)

Hudson et al.

(2016)18

49 F Occasional mild neck

pain

Thoracic spine Vertebral fusions from

upper cervical spine to

upper thoracic spinE

No

Abu-Bonsrah

et al. (2016)19

40 M Left lower extremity pain Lumbar spine Spina bifida Yes (Right hemicord

epidermoid cyst)

Babu et al.

(2014)20

66 F Lower extremity

stiffness and weakness

over a 1year period

Lumbar spine Teratoma Yes (Teratoma)

König et al. 26 F Severe low back pain, Lumbar spine Occult spina bifida and Yes

12

(2012)21 and bilateral L5/S1

sciatica after a fall

tethered cord syndrome (Spondylolisthesis)

Méndez et al.

(2009)22

88 F Low back pain while

attempting to get out of

bed

Lumbar spine Collapse of the L4

vertebral body

Yes (Collapse of the

L4 vertebral body)

Callari et al.

(2008)23

80 F Pain in the dorsal spine

that was resistant to

medical treatment

Lumbar spine T7—T8 vertebral body

compression fractures,

and syringomyelia

Yes (Vertebral body

compression)

Izci et al.

(2007)24

20 M Aplasia cutis and an

abnormal hairy patch

around the skin defect in

the lumbar region

Lumbar spine Aplasia cutis No

Izci et al.

(2007)24

21 M Aplasia cutis and an

abnormal hairy patch

around the skin defect in

the throracic region

Thoracic spine Aplasia cutis No

13

Table 1. Case reports of diastematomyelia and associated lesions.(continued)


Case Report

Porensky et al.

(2007)25

54 F Progressive left lower-

extremity paresis and

back pain that radiated

into the left leg

Thoraco-lumbar

junction

Intradural

extramedullary mass,

consistent with an

epidermoid tumor.

Dextroscoliosis

Yes (epidermoid

tumor)

Tsitsopoulos et

al. (2006)26

44 F Progressive lower limb

muscle weakness, gait

disturbances and

dysesthesias in the

trunk and lower

extremity


Becker et al.

(2005)27

59 F Recurrent sebaceous

cyst on the back of her

neck

Cervical spine Benign neurenteric cyst Yes (Neurenteric

cyst)

Soni et al.

(2004)28

30 F Occasional back pain,

tingling and numbness

in left lower limb, with

involvement of the right

lower limb.

Thoracic spine Intradural extra

medullary dorso-medial

cyst compressing the

left hemicord below the

bony spur

Yes (Cyst

compressing the left

hemicord)

Quinones- 73 F Worsening low back Thoracolumbar Tethered spinal cord No (Age)

14

Hinojosa et al.

(2004)29

pain and progressive

difficulty walking

junction

Lewandrowski

et al. (2004)30

44 F Significant loss of

function in her right leg,

increasing bilateral leg

pain and right leg

weakness with gross

atrophy

Lumbar spine Tethered spinal cord,

spinal stenosis

Yes (Spinal

stenosis)

Sheehan et al.

(2002)31

38 F Lower-extremity pain

and weakness on the

right side. Sensorimotor

deficits, hyperreflexia,

and a positive Babinski

reflex in the right lower

extremity were

demonstrated on

examination

Thoracic spine Cervical syrinx,

epidermoid spinal cord

tumor

Yes (Epidermoid

tumor)

Mastroyianni et

al. (2002)32

2 F Inability to walk and

neurodevelopmental

delay.

Lumbar spine Tethered spinal cord,

Angelman syndrome

Yes (Tethered cord)

15


Case Report


Elmaci et al.

(2001)33

42 M Pain and distal left-leg

weakness as well as

neurogenic claudication

for 1 month


Kaminker et al.

(2000)34

38 M Bilateral leg pain into

both thighs and

occasionally into his feet

Lumbar spine Tethered spinal cord Yes (Tethered

spinal cord)

D'Agostino et

al. (2019)35

57 F Back and left leg pain,

difficulty with

ambulation, and

intermittent urinary

incontinence

Lumbar spine Spina bifida occulta,

tethred cord

Yes (tethered cord)

FIGURES

Figure 1. A. MRI, non-contrast T1 sequence depicting hyperintense lesion at C6-T1. B. Pre-operative MRI in T2 sequence showing

the a suspicious splitted spinal cord

16


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Daniel Buzaglo GonçalvesFaculty of MedicineFederal University of Amazonas (UFAM), Manaus, AM, Brazil Manaus-Amazonas, BrasilE-mail: [email protected]

Institution: Federal University of Amazonas (UFAM)

CORRESPONDING AUTHOR

14 - review diastematomielia cervical associada a lipoma e

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