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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 54  |  Issue : 2  |  Page : 70-74

Non-dysraphism intradural cervical lipoma


1 Department of Surgery, Division of Neurosurgery, Cathay General Hospital, Taipei City, Taiwan
2 Department of Surgery, Division of Neurosurgery, Cathay General Hospital, Taipei City; Department of Medicine, School of Medicine, Fu Jen Catholic University, New Taipei City; Department of Neurological Surgery, National Defense Medical Center, Tri-Service General Hospital, Taipei City, Taiwan
3 Department of Neurological Surgery, National Defense Medical Center, Tri-Service General Hospital, Taipei City; Department of Surgery, Ditmanson Medical Foundation, Chia-Yi Christian Hospital, Chia Yi City; Department of Biotechnology, Asia University, Taichung City, Taiwan
4 Department of Surgery, Division of Neurosurgery, Cathay General Hospital, Taipei City; Department of Medicine, School of Medicine, Fu Jen Catholic University, New Taipei City, Taiwan

Date of Submission21-May-2020
Date of Decision03-Aug-2020
Date of Acceptance18-Nov-2020
Date of Web Publication20-Mar-2021

Correspondence Address:
Chih-Ta Huang
Department of Surgery, Division of Neurosurgery, Cathay General Hospital, No. 280, Section 4, Ren'ai Rd., Da'an Dist., Taipei City 106
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_82_20

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  Abstract 


Intradural spinal lipomas not associated with spinal dysraphism account are rare entity. The pure cervical intradural lipomas are rarer and only eleven cases have been reported in the literature. The median age at diagnosis was 35 (8m-52y) years old and the ratio of men to female was 1.4:1. The possible risk factor included weight increase or rapid metabolic change. Only one case had recurrent condition during follow-up period. Here, we reported a 32-year-old male who presented with posterior neck pain and radiating numbness from neck to bilateral upper extremities. The radiograph of cervical spine showed a widening spinal canal diameter. Further study confirmed a pure cervical intradural intramedullary lipoma without spinal dysraphism. For preventing the neurologic deficit, early diagnosis and adequately surgical decompression were needed. Subtotal removal with cavitational ultrasonic surgical aspirator has been widely use as it minimizes any traction on already compromised neural tissues.

Keywords: Cervical spine, intradural-intramedullary, lipoma, spinal neoplasm


How to cite this article:
Liu ZQ, Hsieh CT, Sun JM, Huang CT. Non-dysraphism intradural cervical lipoma. Formos J Surg 2021;54:70-4

How to cite this URL:
Liu ZQ, Hsieh CT, Sun JM, Huang CT. Non-dysraphism intradural cervical lipoma. Formos J Surg [serial online] 2021 [cited 2021 Apr 13];54:70-4. Available from: https://www.e-fjs.org/text.asp?2021/54/2/70/311591




  Introduction Top


Spinal intradural lipoma is a benign tumor, accounting for approximately about 1% of all spinal neoplasms.[1],[2] The majority of spinal intradural lipoma locates in the low thoracic and lumbosacral region. Cervical intradural lipoma is rarer, accounting for approximately about 13% of spinal lipomas.[3] To the best of knowledge, only 11 cases with pure cervical intradural lipomas have been reported in the literatures. Here, we report a case of cervical intradural lipoma without spinal dysraphism, presenting with neck pain with radiculopathy.


  Case Report Top


A case of 32-year-old male presented with posterior neck pain and radiating numbness from neck to bilateral upper extremities 6 months before admission. He denied any medical history and did not taking any medication. His body mass index was 20. On physical examination, there were no palpable lumps, masses nor other skin lesions at the posterior neck region. Neurologic examination revealed the increasing deep tendon reflexes and the presence of right Hoffman's sign. Touch and joint position sensation impaired. The muscle powers of the four limbs were normal. Laboratory studies were within the normal limits.

The radiograph of the cervical spine showed a widening spinal canal diameter [Figure 1]. Computed tomographic scan of the cervical spine revealed a 2 cm × 8 cm mass in the right posterior aspect of the spinal canal over C2–C7 levels [Figure 2]. The magnetic resonance imaging (MRI) showed a huge intradural mass [Figure 3]a compressed the spinal cord. In a T2-weighted image, there is adequate contrast resolution in the cerebrospinal fluid (CSF) space, the mass can be localized within the dura. The mass appeared as a high intensity on T1-weighted [Figure 3]b and T2-weighted images [Figure 3]c and the suppressed intensity on a short T1 inversion-recovery image [Figure 3]d. In general, an intradural mass lesion is demonstrated within the spinal canal, sometimes with extension into the neural foramina and extradural paraspinal region. When there is adequate contrast resolution of the CSF space, the mass can be localized within the dura but outside the spinal cord.
Figure 1: The radiograph of the cervical spine revealed the increased diameter of the spinal canal (white arrow)

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Figure 2: Computed tomographic scan of the cervical spine revealed a 2 cm × 8 cm mass over C2 to C7 level

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Figure 3: The magnetic resonance images of the cervical spine without contrast. (a) Axial view of T1-weighted image revealed a high-intensity mass (white arrow) located in the right dorsal aspect of the spinal canal with severely compressing the spinal cord (white asterisk). (b) Sagittal view of the T1-weighted image revealed the hyperintensity mass (white arrow) located from C2 to C7 level. (c) Sagittal view of the T2-weighted image revealed the mass (white arrow) with high intensity. (d) Sagittal view of short tau inversion recovery revealed the mass (white arrow) with a suppressing the high signal

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Based on these findings, intradural cervical spine lipoma was highly impressed. The other possible differential diagnosis including epidermoid cyst, lipomyelomeningocele, sarcoidosis, or even hematoma (less likely).

At operation, the mass, located in the intradural region, was found with yellowish in color and soft in content [Figure 4]. The dorsal site margin seemed clear, but the ventral site's demarcation was not clear between the spinal cord, roots, and the mass. Our goal of surgery is to reduce the size of the lipoma, not total removal of the fat subtotal tumor excision was performed under microscope and CUSA assisted. Then, we had watertight closure of the cervical dura. We also performed the laminoplasty from C2 to C7. The patient had a well postoperative condition and could discharge on the 7th postoperative day without neurologic deficit.
Figure 4: Intraoperative microscopic view revealed a huge and yellowish lipomatous mass (white arrow) following durotomy

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Histological examination revealed fibrous tissue in the margin and rich in reticulin and composed of mature adipocytes. The pathological examination confirmed the diagnosis of cervical spine lipoma. Postoperatively, the patient's spasticity and neck pain recovered gradually. There was no specific neurologic deficit noted after 1-year follow-up. The subsequent MRI done 2 months after surgery revealed the residual lipoma without compressing the spinal cord [Figure 5].
Figure 5: The T1-weighted magnetic resonance image of the cervical spine without contrast 2 months after surgery revealed a residual mass (white arrow) in the dorsal aspect of the spinal canal without compression of the spinal cord

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  Discussion Top


Of all the primary intraspinal tumors, intradural lipomas account for 1% of cases. Majority of spinal lipoma was found in the thoracic and lumbar regions. Most of these lipomas are found in the posterior part of the spinal cord, and they usually distort and expand the spinal cord.[4] Pure cervical spine lipoma without vertebral dysraphism is very rare and only 11 cases have been reported in the literature [Table 1].[1],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12]. Including our case, there were seven male and five female, and the age ranged from 8 months to 52 years.
Table 1: The summary of published cases with pure cervical intradural lipomas

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Spinal lipoma has been considered as a developmental defect whereby remnants of embryonic fat are included inside the neural tube during its formation.[13] In dysraphism reason, spinal lipomas are generally thought to occur as a result of failed primary neurulation and it usually happened at the lumbosacral level. For nondysraphism reason, most commonly these rare lesions are located within the thoracic spinal cord. The reason why there are less cases of nondysraphism cervical lipoma is still unclear after reviewing related articles.[14]

Metabolically, the fat in these tumors behaves in the same way as normal fat in the rest of the body. In some cases, these lipomas have been observed to increase in size when the patient's weight increase, or simultaneously with metabolic changes, such as pregnancy.[15] However, rapid lipoma growth despite meticulous diet control contradicts these findings.[16]

The number of nondysraphic spinal cord lipomas is less than that of dysraphic spinal cord lipomas, and they are usually located intradurally. Thus, they usually do not present as subcutaneous lesions, like our patient. Among these 11 cases with cervical intradural lipomas, the presentation is often with an ascend spastic gait affecting one or both legs and the neck pain radiation to upper limbs. Most patients are symptomatic for more half-year before seeking aggressive medical advice.

Early diagnosis is important to avoid prolonged cervical cord compression with permanent and irreversible neurological injury.[17] The best diagnostic tool was MRI, usually demonstrates a T1 hyperintense and T2 hypointense intradural mass with an inversion of intensity at short T1 inversion recovery sequences. A similar lesion was seen in the overlying subcutaneous tissue, supporting the intradural mass indicate the adipose tissue.

Surgical intervention is the standard care of the spinal lipoma. Due to the close adhesion of the lipoma to the spinal cords makes complete tumor excision challenging, the appropriate surgical management of intraspinal lipomas is adequate decompression laminectomy for preventing neurologic deficit, especially intramedullary lipoma. On the other hand, subtotal removal with CUSA, has also been widely used as it minimizes any traction on already compromised neural tissues.[18] Song et al. reported their experience with a regrowth of pure cervical intradural lipoma without dysraphism who underwent initial surgery at 14 years ago, and they suggested that the slowly grow lipoma could be managed by appropriate debulking surgery without further neurologic deficit. Azzai et al. advised the duraplasty to prevent the complication of CSF leakage. However, there was no similar complication regarding to the other published cases. Otherwise, Panagopoulos et al.[12] showed spinal fixation of the bilateral lateral mass screws to avoid postoperative cervical kyphosis. However, spinal fixation may limit postoperative neck motion and induce the posterior neck muscle atrophy. In our case, laminoplasty was performed because this procedure had less blood loss and a less long-term complication rate. In cohorts of matched follow-up cervical sagittal alignment, pain outcomes were similar between both groups.[19] Laminoplasty could also avoid the fixation-associated complication than laminectomy with posterior fusion. However, in the limited published cases of intradural lipomas, the difference between laminoplasty and laminectomy or even laminectomy with fusion could not be concluded.[20]

In conclusion, we present a very rare case of cervical spine lipoma without vertebral dysraphism. Early diagnosis and surgical intervention are the best option for these patients with clinical neurological deficits.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Iwatsuki K. Intradural cervical lipoma with parenchymal marginal fibrous tissue: Case report. Neurosurgery 2006;59:E208.  Back to cited text no. 1
    
2.
Giuffrè R. Intradural spinal lipomas. Review of the literature (99 cases) and report of an additional case. Acta Neurochir (Wien) 1966;14:69-95.  Back to cited text no. 2
    
3.
Khurana V, Kishore D, Gambhir IS. High cervical intradural lipoma. Postgrad Med J 2010;86:251-2.  Back to cited text no. 3
    
4.
Son DK, Son DW, Choi CH, Song GS. Regrowth of cervical intradural lipoma without spinal dysraphism. J Korean Neurosurg Soc 2014;56:157-61.  Back to cited text no. 4
    
5.
Fujiwara F, Tamaki N, Nagashima T, Nakamura M. Intradural spinal lipomas not associated with spinal dysraphism: A report of four cases. Neurosurgery 1995;37:1212-5.  Back to cited text no. 5
    
6.
Castilla JM, Velasco MV, Salazar RA. Lipoma cervical intradural sin compromiso neurológico; A propósito de un caso. Neurocirugía 2002;13:54-8.  Back to cited text no. 6
    
7.
Moghaddam AM, Tanriöver N, Ulu MO, Muhammedrezai S, Akar Z. Cervical intradural lipoma with associated hemivertebra formation at C6 level: A case report. Turk Neurosurg 2008;18:187-90.  Back to cited text no. 7
    
8.
Azzazi A, Sedik M, Sakr S. Surgical management for intradural spinal lipoma in adult patients without spinal dysraphism. Neurosurgery-quarterly 2010;20:130-35.  Back to cited text no. 8
    
9.
Arora A, Kumar Puri S, Upreti L, Kapur A, Gupta R. Non-dysraphic cervical cord lipoma. Eurorad 2010 Case 8344.  Back to cited text no. 9
    
10.
Menzilcioglu MS, Citil S, Sahin T. Nondysraphic cervical intradural lipoma. Spine J 2014;14:2515.  Back to cited text no. 10
    
11.
Carrasco S, Camacho A, Franco A, Burriel L, Hernández A. Cervical intradural extramedullary lipoma: A rare differential diagnosis of nontraumatic limping in an adult patient. Neurol Clin Neurosci 2018;6:107-09.  Back to cited text no. 11
    
12.
Panagopoulos D, Markogiannakis G, Koutzoglou M. Intradural-extramedullary cervical cord lipoma: Case report and literature review. World Neurosurg 2018;110:162-8.  Back to cited text no. 12
    
13.
Meher SK, Tripathy LN, Jain H, Basu S. Nondysraphic cervicomedullary intramedullary lipoma. J Craniovertebr Junction Spine 2017;8:271-4.  Back to cited text no. 13
    
14.
Jones V, Wykes V, Cohen N, Thompson D, Jacques TS. The pathology of lumbosacral lipomas: Macroscopic and microscopic disparity have implications for embryogenesis and mode of clinical deterioration. Histopathology 2018;72:1136-44.  Back to cited text no. 14
    
15.
Teneyuque N, Porter KB, Pennington T. Cervicothoracic intradural spinal lipoma without dyraphism diagnosed postpartum. Mil Med 2015;180:e149-51.  Back to cited text no. 15
    
16.
Aoki N. Rapid growth of intraspinal lipoma demonstrated by magnetic resonance imaging. Surg Neurol 1990;34:107-10.  Back to cited text no. 16
    
17.
Lee M, Rezai AR, Abbott R, Coelho DH, Epstein FJ. Intramedullary spinal cord lipomas. J Neurosurg 1995;82:394-400.  Back to cited text no. 17
    
18.
Klekamp J, Fusco M, Samii M. Thoracic intradural extramedullary lipomas. Report of three cases and review of the literature. Acta Neurochir (Wien) 2001;143:767-73.  Back to cited text no. 18
    
19.
Lau D, Winkler EA, Than KD, Chou D, Mummaneni PV. Laminoplasty versus laminectomy with posterior spinal fusion for multilevel cervical spondylotic myelopathy: Influence of cervical alignment on outcomes. J Neurosurg Spine 2017;27:508-17.  Back to cited text no. 19
    
20.
Lao L, Zhong G, Li X, Qian L, Liu Z. Laminoplasty versus laminectomy for multi-level cervical spondylotic myelopathy: A systematic review of the literature. J Orthop Surg Res 2013;8:45.  Back to cited text no. 20
    


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