MOJ Clinical & Medical Case Reports

Intradural-Extramedullary Metastatic Tumor from Lung
Cancer-A Case Report and Review
Case Report

Abstract
A 62-year-old male presented with a thoracic intradural-extramedullarymetastasis from lung cancer. He complained of walking difficulty and bilaterallower leg numbness. He had a past history of lung adenocarcinoma three years
prior. Thoracic spine magnetic resonance imaging showed that the tumor
occupied the right subarachnoid space and spinal nerve roots had shifted to
the left side at Th12. He was diagnosed with cauda equina syndrome by an
intradural-extramedullary tumor. Surgery was performed (Th12 laminectomy)
and we opened the dura mater and arachnoid membrane. The tumor was in
the right side and was removed. After the operation, his neurological condition
soon improved and he could walk with a cane. By four moths postoperatively,
a metastatic brain tumor occurred and he had to use a wheel chair. He died
one year after surgery. Immunohistochemical staining was positive for thyroidtranscription factor-1 in both the lung adenocarcinoma and intraduralextramedullary tumor. We diagnosed a metastatic tumor. Including the present
case, 102 cases of a metastatic intradural-extramedullary tumor have beenreported and, thus, the condition may not be rare. In more than twenty years
there have only been two case studies, which recommended both operative andnon-operative methods. We reviewed the validity of an operation base on the
prognosis and surgical results of previous reports. The average life expectancy
was 8.6 months. The functional improvement rate was 78%. We recommend
an operation for an intradural-extramedullary metastatic tumor because it
improves the quality of a patients’ short life.

Volume 6 Issue 1 - 2017

Department of Orthopedic Surgery, Osaki Citizen Hospital,Japan
2
Department of Orthopedic Surgery, Tokyo west Tokushukai
hospital, Japan
1*Corresponding author: Tatsuro Sasaji, Department ofOrthopedic Surgery, Osaki Citizen Hospital 3-8-1, Furukawa
Honami, Osaki-shi, Miyagi, Japan, Tel: +81-229-23-3311; Fax:
+81-229-23-5380; Email:Received: December 19, 2016 | Published: January 18,
2017

Keywords: Metastatic tumor; Intradural-extramedullary tumor; Lung cancer;
Cauda equina syndrome; Laminectomy

Abbreviations: IEMT: Intradural-Extramedurally Metastatic

Tumor; JOA: Japanese Orthopaedic Association; MRI: Magnetic
Resonance Imaging; TTF-1: Thyroid Transcription Factor-1

Introduction

Metastases are by far the most common skeletal tumors
seen by orthopedists, and the spine is the most common site ofskeletal involvement [1]. A spinal epidural metastasis is the mostcommon neoplastic myelopathy [2]. In a total of 1215 cases ofprimary lung cancer in an autopsy study, 20 (1.65%) revealed
intradural parenchymal involvement [3]. Among 200 patientstreated for symptomatic spinal metastases, a localized intradural
extramedullary tumor was discovered in 10 cases [4]. Among136 patients who underwent surgery for spinal metastases,
an intradural extramedullary tumor was observed in two cases(1.5%) [5]. To our knowledge, Rogers et al. [6] were the first toreport on an intradural-extramedurally metastatic tumor in 1958
since then, 101 cases have been reported in 66 articles [4–69].
Including our case, the total number of reported cases is now
102. Many authors have described an intradural-extramedullarymetastasis as a rare metastatic condition, however, we do notconsider it to be rare given the number of reported cases.
Two types of treatment have been reported for an intraduralextramedullary tumor, the recommended operative and nonoperative methods [4,18,31,38,45,63]. In two case studies, Perrin
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et al. [4] recommended a non-operative method and Chow et al.
[30] recommended an operation. Both reports had a small numberof cases (n=10). There have been no reviews of many cases thatwere not limited to primary tumors and in more than twenty years
since the two case studies were reported, medical technology has
advanced. Surgery for a metastatic tumor is palliative and cannot
extend the life expectancy. So, we consider that there is a need toreview previous reports and examine the validity of surgery for an
intradural-extramedullary metastatic tumor.

In this article, we report on a case of a thoracic intraduralextramedullary metastatic tumor from lung cancer. We further
review previous reports and our case from the point of view of
postoperative life expectancy and surgical results. The patientand his family gave consent to submit data for publication.
The protocol for case report and review was approved by the
Ethics Committee at Osaki Citizen Hospital (Osaki, Japan) (H28Rinri-18).

Case Presentation

In 2015, a 62-year-old female presented to our hospital withgait disturbance and numbness in the bilateral lower extremities.
There was a past history of adenocarcinoma in the apical portionof left lung that had been treated with chemotherapy and radiationin 2012 (Figure 1). In 2013, a brain metastatic tumor from lungcancer occurred that was treated with an operation and gamma
knife.
MOJ Clin Med Case Rep 2017, 6(1): 00147

Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case Report andReview

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Figure 1: Computed tomographic image of the chest.
The tumor can be seen in the apex of the left lung (black arrow).

Neurological examination

Radiological Findings

Upon neurological examination, the iliopsoas muscle strength
was zero and quadriceps muscle and tibialis anterior muscle
strengths were three (fair) in the right lower extremity using amanual muscle test. Muscle strength was graded on a 0 (zero)
to 5 (normal) scale (arbitrary units) [70]. The patellar tendon
and Achilles tendon reflexes could not be elicited. Sensory
disturbance was detected in the right anterior femur. The patient
was diagnosed with cauda equina syndrome. The Japanese
Orthopaedic Association (JOA) score for lumbar disease was 7 outof 29 [71]. All laboratory findings were within normal limits.

Preoperative sagittal magnetic resonance imaging (MRI)
showed that the tumor was located in the dura mater at Th12
(Figure 2A–C). Compared with the spinal cord, the tumor had
an iso-intensity signal in a T1-weighted MRI and high-intensity
signal in a T2-weighted MRI (Figure 2A,B). The tumor wasenhanced by Gadolinium with diethlenetriaminepentacetate (GdDTPA) (Figure 2C,D). The spinal nerve roots were compressed by
the tumor, as observed on an enhanced T1-weighted axial image
with Gd-DTPA (Figure 2D). The patient was diagnosed as having
an intradural-extramedullary tumor and tumor resection surgery
was performed to decompress the nerves.

Figure 2: Magnetic resonance image of the thoracic spine.
Figure 2A: T1-weighted sagittal imaging.
Figure 2B: T2-weighted sagittal imaging.
Figure 2C: Gadolinium with diethlenetriaminepentacetate (Gd-DTPA) enhanced
sagittal imaging.
Figure 2D: Gd-DTPA enhanced axial imaging.
An intradural-extramedullary tumor can be seen at the 12th thoracic vertebral body level. The tumor has an iso intensity in T1-weighted imaging (A)and high intensity in T2-weighted imaging (B) compared with the spinal cord (white arrow). The tumor was enhanced by Gd-DTPA (white arrow)(C, D).

Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case Report andReview

Operation
The thoracic spine was explored through a straight posterior
midline approach. A laminectomy at Th12 revealed no extradural
tumor. The dura mater and arachnoid membrane were exposed

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and the spinal nerve roots and conus medullaris were compressedby the tumor (Figure 3A). We scraped out and removed the tumor
after which the spinal nerve roots and conus medullaris were
decompressed (Figure 3B).

Figure 3: Intraoperative photograph.
Figure 3A: Before tumor resection.
Figure 3B: After tumor resection.
Once the dura matter was opened, the tumor was can be seen in the left dura mater. The tumor adhered to spinal nerve roots (A). Following a tumor
resection, the conus medulallis (black arrow) and spinal nerve roots were decompressed (B).

Postoperative Course

Muscle weakness in the right lower extremities improved soonafter surgery. It was irradiated at 34 Gy after the operation. Threemonths later, the patient complained of numbness in the rightlower extremity. His muscle strength improved to normal and hecould walk with a cane. The JOA score was 22 out of 29 [71]. Fourmonths later, the brain metastatic tumor recurred and he could
no longer walk and used a wheel chair. The patient died one year
after surgery.

Pathological Findings

Lung cancer stained by Hematoxilin-eosin (HE) showed low
differentiated adenocarcinoma (Figure 4A). The intraduralextramedullary tumor HE stained showed a moderately
differentiated adenocarcinoma (Figure 4B). Both the lungadenocarcinoma
andintradural-extramedullarytumor
were positive by immunohistochemical staining for thyroidtranscription factor-1 (TTF-1) (Figure 4C,D). TTF-1 is a substancethat is a specific stain for lung adenocarcinoma and thyroid
cancer and is recognized as useful for the diagnosis of lungadenocarcinoma [72,73]. According to the above pathologicalfindings, the pathological diagnosis was an intraduralextramedullary metastatic tumor from lung adenocarcinoma.

Discussion

According to previous reports, the main primary tumors of
intradural-extramedullary metastasis were lung cancer (24cases

including ours), breast cancer (18cases) and renal cell carcinoma(15cases) (Figure 5) [4-69]. The average life expectancy of an
intradural-extramedullary metastasis was 110 days in lung cancer
and more than one year in renal cell carcinoma [3,62]. These
reports were limited to each primary tumor. However, the primarytumor cannot be diagnosed before surgery and we need thegeneral life expectancy for treatment. The general life expectancy
of an unfamiliar condition, intradural-extramedullary metastasis,is still unknown. Therefore, we examined the postoperative life
expectancy of all 65 cases, 64 cases previously reported plus thepresent case [5,6,8,10-14,16-18, 22, 24, 26-32,34,36-38,41,4446,48,50-52,54,55,58,60-65,67-69]. We excluded articles that
had no description or a vague description about life expectancy.
In cases where patients were alive at the last examination,
we calculated as over the time until the last examination. We
calculated one month as four weeks. The average life expectancy
was more than 8.6 months (1week ~ more than 63 months)(Figure 6). The life expectancy of an intradural-extramedullarymetastasis was considered to be not so long.
Most reports about an intradural-extramedullary metastasis
were case reports. Postoperative functions were reported invarious ways and were not uniform so the general surgical results
were unknown. According to Chow et al. [31], we evaluated thesurgical results of each report by simple three grades: improved,
unchanged, or worse. We defined the percentage of functional
improvement as the number of improved cases divided by the
total number of cases × 100. We reviewed a total of 70 patients:

Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case Report andReview

69 cases with descriptions about the postoperative function plus
the present case [5,6,8,10,11,13,14,16-18,20,21, 23-36, 28-35,3749,51, 52,54,55,57,60-62,64-66,68,69]. A total of 55 cases were
improved (Figure 7). The overall functional improvement rate
was 78%. An intradural-extramedullary metastatic tumor was
expected to improve following surgery regardless of the primarytumor.
For the treatment of an intradural-extramedullary metastatic
tumor, some reports recommended surgery [18,31,45,63] andsome recommended non-surgical methods [4,38]. Of 102 cases,
including ours, 94 received surgery. Non-surgical methods
were used in eight patients because of a poor general condition[19,22,26,34]. According to our review of an intradural-

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extramedullary metastasis, the life expectancy was more than
8.6 months and a functional improvement was expected. So, we
recommended an operation for the treatment of an intraduralextramedullary metastatic tumor to improve the quality of life,
even though the life expectancy may be short.

This review has some limitations. This review included
both old and recent reports that were under different medical
conditions. Some reports had an inadequate or no descriptionof the postoperative survival duration. All reports describedpostoperative function in different ways and the postoperativeevaluation of this review was, therefore, inadequate. The
functional improved duration was not described.

Figure 4: Histopathologic examination.
Figure 4A: Lung cancer (Hematoxilin and eosino stain).
Figure 4B: Intradural extramedullary tumor (Hematoxilin and eosino stain).
Figure 4C: Lung cancer (Thyroid transcription factor-1 stain).
Figure 4D: Intradural extramedullary tumor (Thyroid transcription factor-1 stain).
Lung cancer in the Hematoxilin-eosin (HE) stain showed a proliferation of atypical cells composed of a high nucleo-cytoplasmic ratio. This was low
differentiated adenocarcinoma (A). An intradural-extramedullary tumor in the HE stain showed that atypical epithelial cells proliferated. This wasmoderately differentiated adenocarcinoma (B). The antibody to thyroid transcription factor-1 (TTF-1) is a specific marker for lung adenocarcinoma
and thyroid cancer. Both lung cancer and THE intradural-extramedullary tumor showed positive immunohistochemical staining for TTF-1 (C, D).
The intradural-extramedullary tumor was diagnosed as a metastatic tumor from lung adenocarcinoma. Magnified 400 times.

Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case Report andReview

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Figure 5: Reported primary tumors including present case.
Lung cancer was 24 cases [4,5,9,15,22,31,51,55,56,59,63,67, present case]. Breast cancer was 18 cases [4, 5,7,8,11,13,14,16,22,24,41,57,61]. Renal
cell cancer (including Wilm’s tumor and Grawitz tumor) was 15 cases [25,27,35,38,43,45-47,49,52,54,58,64-66]. Occult primary cancer was 9 cases[10,13,14,16,17,22, 24,26,31]. Ovarian cancer was 4 cases [19,31,32,39] Prostate cancer was 4 cases [26,34,44,68]. Malignant melanoma was threecases [4,18]. Carcinoid was three cases [23,33,48]. Chondrosarcoma was two cases [28,53]. Uterine malignancy was two cases [4,20]. Nasopharynx
was two cases [21,31]. Colorectal cancer was two cases [12,22]. Neuroblastoma was two cases [13,60]. There was once case each of adrenal glandcancer [6], gastric cancer [29], anal cancer [30], glioblastoma [36], sinonasal carcinoma [37], germinoma [40], submandibular gland carcinoma [82],papillary carcinoma of the thyroid [62], muscle [31], lymphoreticular system [31], lacrimal gland [31], and a pancreatic neuroendocrine tumor [69].

Figure 6: Postoperative life expectancy.
There were 10 cases of <1 month, 18 cases between 1-3 months, 12 cases between 3-6 months, 15 cases between 6-12 months, six cases between
1-2 years, three between 2-3 years, and one case between 3-6 years. The average life expectancy was 8.6 months and 84.6% patients died within
one year.

Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case Report andReview

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Figure 7: Postoperative functional results.
There were a total of 70 cases of which 55 were improved [5,6,8,10,11,13,14,16-18,20,23-25,28-33,35,37-41,44-49,52,54-59,61-69], eight remained
unchanged [8,21,26,31,51], and seven were worse [13,31,34, 42,43,60]. The postoperative functional improvement rate was 78%.

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Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

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Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147

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Citation: Sasaji T, Imaizumi H, Takano H, Saitoh H, Murakami T, et al. (2017) Intradural-Extramedullary Metastatic Tumor from Lung Cancer-A Case
Report and Review. MOJ Clin Med Case Rep 6(1): 00147. DOI: 10.15406/mojcr.2017.06.00147