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CASE REPORT
Year : 2019  |  Volume : 14  |  Issue : 3  |  Page : 949-951

Giant spinal intradural metastatic adenocarcinoma of unknown primary: A rare case report


Department of Neurosurgery, Institute of Neurosurgery, Madras Medical College, Chennai, Tamil Nadu, India

Date of Web Publication2-Aug-2019

Correspondence Address:
Natarajan Meenakshisundaram
Room No. 16, PG Men's Hostel, Superspeciality Block, Chennai - 600 003, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ajns.AJNS_65_18

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  Abstract 


Giant intradural metastases of nonneurogenic origin involving multiple segments represent an extremely rare manifestation of an unknown primary. The respective literature is very scarce. We present a 45-year-old female with complaints of low back pain for 4 years, involuntary urination for 2 years, and difficulty in using both lower limbs for 1 month. Examination revealed paraparesis with hypotonia. Imaging of lumbosacral spine revealed that expansile lytic destruction of vertebral bodies and posterior elements was noted from D8 to S2 vertebra and a large-sized patchy enhancing heterogeneous intradural extramedullary lesion was noted in D8–S2 level. Decompressive laminectomy from D11 to L4 vertebra and subtotal excision of the lesion were done. There was a marked improvement in the lower limb weakness and low back pain postoperatively. Histopathology revealed metastatic adenocarcinoma. Immunohistochemistry showed epithelial membrane antigen positivity. Accordingly, the aim of the surgery is strictly palliative. The majority of patients benefit with respect to neurological deficit/pain independent of the extent of resection. Thus, decompressive surgery is recommended to increase the quality of life. The occurrence of intradural spinal metastasis is rare. Only few cases of intra dural spinal metastasis involving multiple cord segments and osteolytic bony erosions have been documented. Hence this case is being presented here for its rarity and its uniqueness.

Keywords: Giant intradural metastasis, posterior decompression, unknown primary


How to cite this article:
Meenakshisundaram N, Dhandapani B. Giant spinal intradural metastatic adenocarcinoma of unknown primary: A rare case report. Asian J Neurosurg 2019;14:949-51

How to cite this URL:
Meenakshisundaram N, Dhandapani B. Giant spinal intradural metastatic adenocarcinoma of unknown primary: A rare case report. Asian J Neurosurg [serial online] 2019 [cited 2019 Nov 15];14:949-51. Available from: http://www.asianjns.org/text.asp?2019/14/3/949/258110




  Introduction Top


Giant intradural metastases of nonneurogenic origin involving multiple segments represent an extremely rare manifestation of an unknown primary. The respective literature is very scarce.


  Case Report Top


A 45-year-old female patient presented with complaints of low back pain for 4 years, involuntary urination for 2 years, and difficulty in using both lower limbs for 1 month. Examination revealed paraparesis with hypotonia, absent lower limb reflexes, and decreased anal tone. All sensory modalities decreased from L1. Computed tomography (CT) lumbosacral spine revealed expansile lytic destruction of vertebral bodies and posterior elements from D8 to S2 vertebra [Figure 1] and [Figure 2]. Magnetic resonance imaging (MRI) Lumbo-sacral spine revealed a large patchy hetero intense lesion noted from D8–S2 level (Intra Dural Extra Medullary - IDEM) with erosion of D11–S2 and enhances with contrast [Figure 3] and [Figure 4]. Decompressive laminectomy was carried out from D11 to L4 vertebra, and a highly vascular IDEM exophytic lesion from D8 to S2 grayish-white was noted pushing the cord laterally [Figure 5]. The cord was found to be thinned out, and bony erosions were noted intraoperatively [Figure 6]. Subtotal excision of the lesion was done [Figure 7]. There was a marked improvement in the lower limb weakness and low back pain postoperatively. Histopathology revealed metastatic adenocarcinoma. Immunohistochemistry showed epithelial membrane antigen positivity. CT chest and abdomen was normal. Panendoscopy and colonoscopy were also negative. The patient has completed radiotherapy.
Figure 1: CT LS Spine showing expansile lytic destruction of vertebral bodies and posterior elements from D8 to S2 vertebra

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Figure 2: Computed tomography lumbosacral spine with three-dimensional reconstruction

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Figure 3: MRI LS Spine T2 Sagittal section showing the extent of the lesion from D8 to S2

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Figure 4: MRI LS Spine axial section showing the invasive nature of the lesion

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Figure 5: Intra operative image showing the extensive metastatic lesion

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Figure 6: Intra operative image showing the osteolytic lesions in the vertebra after subtotal excision of the metastatic lesion

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Figure 7: Excised specimen

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  Discussion Top


Spinal metastases can be anatomically classified into (i) intramedullary, (ii) IDEM, and (iii) extradural. Although spinal metastases are common, almost 95% of these are extradural lesions.[1] Although vertebral and epidural metastases are commonly seen to occur in malignancies, intradural metastases are very rare. Intradural lesions occur within the dural sac and can be anatomically classified into (a) IDEM and (b) intradural intramedullary. Extramedullary lesions are most commonly nonneoplastic. The differential diagnoses for IDEM spinal lesions include meningiomas, neurofibromas, and nerve sheath tumors.[2]

Pain is the most common symptom in 90% of cases.[3] Associated neurological deficits manifest based on the location of the lesion. Gadolinium-enhanced MRI is the preferred imaging modality to assess the lesion.

Five possible routes of spread, for the development of intradural spinal metastases from outside the central nervous system, include: (1) through the rich venous plexus, (2) perineural lymphatics, (3) seeding from involved osseous structures to the cerebrospinal fluid (CSF) through the dura, (4) spreading through subarachnoid space, and (5) hematogenous spreading through the arterial system.

IDEM metastases are commonly thought to originate from CSF seeding. Initially, tumor cells are transferred to the brain, and then, they enter the CSF and are transported throughout the nervous system by CSF flow. This can result in either multifocal or diffuse infiltration of the leptomeninges. Finally, metastatic tumors arise as IDEM lesions (drop metastasis).

Decompressive laminectomy was once the primary treatment for malignant spinal cord compression. Although laminectomy allows for a larger posterior space for the spinal cord, most metastatic impingement originates from the vertebral body and leads to primarily ventral pressure.[4],[5]

With the availability of radiation therapy (RT), laminectomy was combined with adjuvant radiation. With the addition of RT, improved results were obtained with approximately 30%–50% of patients remaining ambulatory after treatment.[6],[7],[8],[9] However, retrospective studies suggested that radiation alone was as effective as laminectomy plus postoperative RT in the treatment of malignant spinal cord compression.[10],[11] Nevertheless, combined treatment remained the standard until 1980 when a small randomized trial [12] suggested that RT alone was as effective as laminectomy with adjuvant radiation in the treatment of spinal cord compression.


  Conclusions Top


Intradural metastases are associated with limited survival time. Accordingly, the aim of the surgery is strictly palliative. The majority of patients benefit with respect to neurological deficit/pain independent of the extent of resection. Thus, decompressive surgery is recommended to increase the quality of life. The occurrence of intradural spinal metastasis is rare and that there is insufficient literature to document the involvement of multiple segments of the cord incontiguity and bony erosions from D11 to S2 by the metastatic lesion. Hence, we report a rare case of giant intradural spinal metastatic adenocarcinoma of unknown origin.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Schick U, Marquardt G, Lorenz R. Intradural and extradural spinal metastases. Neurosurg Rev 2001;24:1-5.  Back to cited text no. 1
    
2.
Chamberlain MC, Tredway TL. Adult primary intradural spinal cord tumors: A review. Curr Neurol Neurosci Rep 2011;11:320-8.  Back to cited text no. 2
    
3.
Jacobs WB, Perrin RG. Evaluation and treatment of spinal metastases: An overview. Neurosurg Focus 2001;11:e10.  Back to cited text no. 3
    
4.
Hirabayashi H, Ebara S, Kinoshita T, Yuzawa Y, Nakamura I, Takahashi J, et al. Clinical outcome and survival after palliative surgery for spinal metastases: Palliative surgery in spinal metastases. Cancer 2003;97:476-84.  Back to cited text no. 4
    
5.
Leviov M, Dale J, Stein M, Ben-Shahar M, Ben-Arush M, Milstein D, et al. The management of metastatic spinal cord compression: A radiotherapeutic success ceiling. Int J Radiat Oncol Biol Phys 1993;27:231-4.  Back to cited text no. 5
    
6.
Akeyson EW, McCutcheon IE. Single-stage posterior vertebrectomy and replacement combined with posterior instrumentation for spinal metastasis. J Neurosurg 1996;85:211-20.  Back to cited text no. 6
    
7.
Byrne TN. Spinal cord compression from epidural metastases. N Engl J Med 1992;327:614-9.  Back to cited text no. 7
    
8.
Loblaw DA, Perry J, Chambers A, Laperriere NJ. Systematic review of the diagnosis and management of malignant extradural spinal cord compression: The cancer care Ontario practice guidelines initiative's neuro-oncology disease site group. J Clin Oncol 2005;23:2028-37.  Back to cited text no. 8
    
9.
Marshall LF, Langfitt TW. Combined therapy for metastatic extradural tumors of the spine. Cancer 1977;40:2067-70.  Back to cited text no. 9
    
10.
Wright RL. Malignant tumers in the spinal extradural space: Results of surgical treatment. Ann Surg 1963;157:227-31.  Back to cited text no. 10
    
11.
Gilbert RW, Kim JH, Posner JB. Epidural spinal cord compression from metastatic tumor: Diagnosis and treatment. Ann Neurol 1978;3:40-51.  Back to cited text no. 11
    
12.
Young RF, Post EM, King GA. Treatment of spinal epidural metastases. Randomized prospective comparison of laminectomy and radiotherapy. J Neurosurg 1980;53:741-8.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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