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CASE REPORT Table of Contents  
Ahead of print publication
Small-cell lung carcinoma diagnosed only after cervical intradural metastasis caused hemiparesis


1 Department of Neurosurgery, Southern TOHOKU Research Institute for Neurosciences, Koriyama, Fukushima, Japan
2 Department of Respiratory Medicine, Southern TOHOKU Research Institute for Neurosciences, Koriyama, Fukushima, Japan
3 Department of Pathology, Southern TOHOKU Research Institute for Neurosciences, Koriyama, Fukushima, Japan

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Date of Submission25-Jul-2020
Date of Acceptance24-Aug-2020
Date of Web Publication14-Sep-2020
 

  Abstract 


We present a rare case of intradural extramedullary metastasis of small-cell lung carcinoma in the cervical spine. Optimal treatment strategies, including the indication for decompressive surgery, are discussed. A 70-year-old male who first presented with nuchal region pain suddenly developed right hemiparesis and was referred to our emergency service. Magnetic resonance imaging of his brain was unremarkable, but additional scanning of the cervical spine revealed an intradural tumor at the right C1–C3 levels. Urgent decompressive surgery was carried out, and gross total removal of the tumor was achieved. The histopathological diagnosis was metastasis of small-cell lung carcinoma. Computed tomography of the lung and abdomen revealed a primary tumor in his left lung, with multiple metastases to the mediastinal lymph nodes, liver, and adrenal gland. The patient was being considered for chemotherapy after some improvement of his hemiparesis and pain, but died on the 13th day due to sudden aggravation of the general condition. There was probably no surgical indication for the present case. Careful assessment and management of the primary disease should be considered the top priority even in patients with emergent neuronal insult.

Keywords: Optimal treatment, small-cell lung carcinoma, spinal intradural extramedullary metastasis, surgical decompression


How to cite this URL:
Hirano Y, Fujimori D, Omi F, Masawa M, Sakuma H, Watanabe K. Small-cell lung carcinoma diagnosed only after cervical intradural metastasis caused hemiparesis. J Spinal Surg [Epub ahead of print] [cited 2020 Nov 27]. Available from: http://www.jossworld.org/preprintarticle.asp?id=295010



  Introduction Top


Spinal intradural extramedullary metastasis is an extremely rare clinical manifestation, with an incidence of 0.9%–2.1% in autopsy series of cancer patients.[1],[2],[3] We present a rare case of intradural extramedullary tumor in the cervical spine, which was diagnosed as metastasis of small-cell lung carcinoma only after removal. Although the patient presented with hemiparesis and urinary retention on admission, and was referred to us without a past history of lung cancer, we probably should not have operated on this patient. Optimal treatment strategies for spinal intradural metastases, including the indications for decompressive surgery, are discussed.


  Case Report Top


A 70-year-old male with hypertension and obesity presented with progressive pain in the nuchal region persisting for 2 months. He had visited a local general practitioner to have analgesics prescribed. He had suffered from pneumothorax 3 years ago, and had chronic obstructive pulmonary disease, based on his smoking history of forty cigarettes per day for 47 years (Brinkman index, 1880), but he had never been diagnosed with cancer of the lung or any other organs. He suddenly developed right hemiparesis (3/V) and was referred to our emergency service. Magnetic resonance (MR) imaging of the brain detected no remarkable lesion to explain his right hemiparesis, but additional scanning of the cervical spine revealed intradural extramedullary tumor at the right C1–C3 levels with significant spinal cord compression and intramedullary T2-weighted hyperintensity [Figure 1].
Figure 1: T2-weighted magnetic resonance images (a-c) and gadolinium.enhanced T1.weighted magnetic resonance images (d-f) showing an intradural extramedullary tumor at the right C1–C3 levels, causing severe compression of the spinal cord and associated with intramedullary signal intensity changes. The sagittal images (a and d) are obtained on the midline and the axial images at the levels of C2 (b and e) and C3 (c and f)

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Urgent decompressive surgery was decided because of his hemiparesis and urinary retention. The time limitation did not allow consultation with the tumor board of our hospital prior to the operation. Right-sided C1–C3 recapping hemilaminoplasty using the ultrasonic bone scalpel [4] was performed to widely expose the tense dural surface. White jelly-like soft tumor exuded spontaneously as soon as the dural sac was opened [Figure 2]. The tumor was located between the dura and the arachnoid surface, exactly as described previously,[5] and gross total removal was achieved. The dural sac was primarily closed with vascular closure staple clips,[6] and the recapped right C1–C3 laminae were secured with titanium miniplates and screws.[4] Transcranial motor-evoked potential was monitored throughout the operation,[7] which detected no reduction of the amplitude in all extremities. The patient was admitted to our intensive care unit (ICU) postoperatively because of his unstable respiratory status, which improved on the next day.
Figure 2: Intraoperative photograph showing the white jelly-like soft tumor which exuded spontaneously as soon as the dural sac was opened, which was easily removed with forceps

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Blood assay for tumor markers revealed significant elevation of neuron-specific enolase (NSE) to the level of 134.0 ng/mL compared to the normal range of <16.3 ng/mL. Slight elevations of carcinoembryonic antigen (CEA), CYFRA (cytokeratin 19 fragment), and soluble interleukin 2 receptor were also noted. The immunohistopathological diagnosis was neuroendocrine carcinoma, compatible with small-cell lung carcinoma [Figure 3], based on the positive findings for epithelial membrane antigen, NSE, chromogranin A, cluster of differentiation 56, and synaptophysin and negative findings for CEA, cytokeratin 5/6, cytokeratin 7, S-100 protein, and neurofilament. Computed tomography of the lung and abdomen revealed the primary tumor in the lingular segment of his left lung [Figure 4]a, with multiple metastases to the mediastinal lymph nodes [Figure 4]b, right hepatic lobe, and left adrenal gland [Figure 4]c.
Figure 3: Histopathological study of the tumor revealing neuroendocrine carcinoma, compatible with small-cell lung carcinoma. H and E staining (a) showing monotonous tumor cells with high nuclear-cytoplasmic ratio, densely proliferating in an alveolar pattern, as well as mitosis and apoptosis. Immunostaining with neuron-specific enolase (b) and synaptophysin (c) showing strongly positive reaction

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Figure 4: Computed tomography scans showing the primary tumor in the lingular segment of the left lung (a: black arrow), with multiple metastases to the mediastinal lymph nodes (b: white arrow), right hepatic lobe, and left adrenal gland (c: white arrows)

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Rehabilitation and food intake was started on the 3rd postoperative day, with some improvement of his right hemiparesis and nuchal region pain, so the patient left the ICU and was referred to our respiratory physicians for possible chemotherapy for the primary disease. He showed slightly more improvement in his motor function and respiratory status within a week. Contrary to our expectations for recovery, the patient suddenly developed severe respiratory distress and liver dysfunction, and became unconscious on the 10th postoperative day. He died of multiple organ failure on the 13th postoperative day. Autopsy permission was refused by his family.


  Discussion Top


Intradural spinal metastases are extremely rare, with the reported incidence of 0.9%–2.1% of cancer patients in autopsy series,[1],[2],[3] and little is known about its surgical indications and outcomes.[8] However, recent improvements in cancer therapy have prolonged the life expectancy of the patients, so formerly rare metastases like this have become a growing medical problem. Optimal management for this clinical entity is still controversial due to the very scarce literature.[9] A review of previous cases of intradural spinal metastases found that the most common histopathological finding was adenocarcinoma and the most frequent site of origin was the breast.[10] A series of ten cases reported in 1982 included only thoracic and lumbar lesions,[11] but more recent studies [8],[9],[10] revealed widespread distribution from the craniovertebral junction to the lumbosacral levels. Surgical treatment is intended to obtain the histopathological diagnosis, decompress the neural elements, increase functional capability, and relieve pain.[12] Certainly, invasive treatment for terminally ill patients with short life expectancy should be avoided.

Stereotactic radiotherapy is a potential option, which can be applied without the risks of general anesthesia and postoperative functional worsening. Stereotactic radiosurgery was applied very successfully in a case of intradural metastasis of small-cell lung carcinoma.[13] The MR imaging findings were similar in that case and our present case, but the previous patient had no neurological symptom except for severe neck pain. Another difference was that small-cell lung carcinoma had been confirmed in the patient before the symptom developed, and the efficacy of radiotherapy was expected. Our present patient presented with sudden onset of right hemiparesis, and radiotherapy could not be considered because of the absence of information such as medical history and histopathological findings about the primary disease.

Consequently, we deeply regret to conclude that there was probably no surgical indication for the present patient, despite the various difficulties experienced in the decision-making process, as well as the current controversies on treatment strategies for spinal intradural metastases. However, refusal of surgical treatment for emergency patients with paralysis of sudden onset due to significant spinal cord compression is extremely difficult from the neurosurgical point of view. The benefit of obtaining the histopathological diagnosis also cannot be ignored. Clearly, the decision to operate depends on the extent of the primary disease, systemic disease progression, and the life expectancy,[12] even in patients with neuronal insult. Surgical treatment for this clinical entity is only palliative,[14] and careful systemic assessment and management of the primary disease should be considered the top priority. The expertise of a tumor board available even for the emergency cases on weekends would be valuable to resolve such clinical problems.

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 his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Chason JL, Walker FB, Landers JW. Metastatic carcinoma in the central nervous system and dorsal root ganglia. A prospective autopsy study. Cancer 1963;16:781-7.  Back to cited text no. 1
    
2.
Costigan DA, Winkelman MD. Intramedullary spinal cord metastasis. A clinicopathological study of 13 cases. J Neurosurg 1985;62:227-33.  Back to cited text no. 2
    
3.
Okamoto H, Shinkai T, Matsuno Y, Saijo N. Intradural parenchymal involvement in the spinal subarachnoid space associated with primary lung cancer. Cancer 1993;72:2583-8.  Back to cited text no. 3
    
4.
Matsuoka H, Itoh Y, Numazawa S, Tomii M, Watanabe K, Hirano Y, et al. Recapping hemilaminoplasty for spinal surgical disorders using ultrasonic bone curette. Surg Neurol Int 2012;3:70.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Borovich B, Keret D, Ben-Arie J, Grushkiewicz I, Peyser E. Spinal intradural metastases of extraneural origin. Acta Neurochir (Wien) 1981;56:99-105.  Back to cited text no. 5
    
6.
Sugawara T, Itoh Y, Hirano Y, Higashiyama N, Shimada Y, Kinouchi H, et al. Novel dural closure technique using polyglactin acid sheet prevents cerebrospinal fluid leakage after spinal surgery. Neurosurgery 2005;57:290-4.  Back to cited text no. 6
    
7.
Takeda M, Mizuno J, Hirano Y, Tomii M, Matsushima T, Watanabe K, et al. Intraoperative neurophysiological monitoring during spine surgery. Jpn J Neurosurg (Tokyo) 2012;21:103-10.  Back to cited text no. 7
    
8.
Hoover JM, Krauss WE, Lanzino G. Intradural spinal metastases: A surgical series of 15 patients. Acta Neurochir (Wien) 2012;154:871-7.  Back to cited text no. 8
    
9.
Wostrack M, Pape H, Kreutser J, Ringel F, Meyer B, Stoffel M, et al. Surgical treatment of spinal intradural carcinoma metastasis. Acta Neurochir (Wien) 2012;154:349-57.  Back to cited text no. 9
    
10.
Chow TS, McCutcheon IE. The surgical treatment of metastatic spinal tumors within the intradural extramedullary compartment. J Neurosurg 1996;85:225-30.  Back to cited text no. 10
    
11.
Perrin RG, Livingston KE, Aarabi B. Intradural extramedullary spinal metastasis. A report of 10 cases. J Neurosurg 1982;56:835-7.  Back to cited text no. 11
    
12.
Alves OL, Hoover JM, Krauss WE, Lanzino G. Intradural spinal metastases: A surgical series of 15 patients. Acta Neurochir (Wien) 2012;154:877.  Back to cited text no. 12
    
13.
Bennett EE, Berriochoa C, Habboub G, Brigeman S, Chao ST, Angelov L. Rapid and complete radiological resolution of an intradural cervical cord lung cancer metastasis treated with spinal stereotactic radiosurgery: Case report. Neurosurg Focus 2017;42:E10.  Back to cited text no. 13
    
14.
Hentschel SJ, Mendel E, Singh S, Rhines LD. Metastatic prostate carcinoma to the intradural extramedullary spinal compartment. Case report. J Neurosurg 2004;100:375-7.  Back to cited text no. 14
    

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Correspondence Address:
Yoshitaka Hirano,
Department of Neurosurgery, Southern TOHOKU Research Institute for Neurosciences, 7-115 Yatsuyamada, Koriyama, Fukushima 963-8563
Japan
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/joss.joss_5_20



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