Accepted Manuscript Unusual Cause of Cord Compression - A Pressing Issue for Neurosurgeons Kamal Kant Sahu, M.D, Pruthvi Sanamandra, M.D, Preethi Jeyaraman, M.D, Ganesh Kumar, M.D, Gaurav Prakash, M.D, DM Medical Oncology, Narender Kumar, M.D, Pankaj Malhotra, M.D PII:

S1878-8750(15)00494-5

DOI:

10.1016/j.wneu.2015.04.061

Reference:

WNEU 2890

To appear in:

World Neurosurgery

Received Date: 29 April 2015 Accepted Date: 30 April 2015

Please cite this article as: Sahu KK, Sanamandra P, Jeyaraman P, Kumar G, Prakash G, Kumar N, Malhotra P, Unusual Cause of Cord Compression - A Pressing Issue for Neurosurgeons, World Neurosurgery (2015), doi: 10.1016/j.wneu.2015.04.061. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

ACCEPTED MANUSCRIPT TITLE – UNUSUAL CAUSE OF CORD COMPRESSION - A PRESSING ISSUE FOR NEUROSURGEONS

AUTHORS LIST

1st author- Kamal Kant Sahu, M.D, PGIMER, Chandigarh 2nd author-Pruthvi Sanamandra, M.D, PGIMER, Chandigarh

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3rd author-Preethi Jeyaraman M.D, PGIMER, Chandigarh 4rth author-Ganesh Kumar, M.D, PGIMER, Chandigarh

5th author-Gaurav Prakash, M.D, DM Medical Oncology, PGIMER, Chandigarh

CORRESPONDENCE Dr Pankaj Malhotra Department of Internal Medicine Ph. no- +91-7087009680

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Email ID- [email protected]

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Fax no - 0091-172-2746018

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7th author- Pankaj Malhotra, M.D, PGIMER, Chandigarh

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6th author- Narender Kumar, M.D, PGIMER, Chandigarh

ACCEPTED MANUSCRIPT Dear Editor, We read with great pleasure the recent article by Joseph JR et al regarding an unusual presentation of acute myeloid leukaemia in your esteemed journal (1). We hereby share a

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similar case who presented to us with acute onset paraparesis and orbital swelling. A 28-year-old male presented with 1 week history of right eye swelling and visual obscuration (Figure 1A). Two weeks later he started feeling band like sensation along the

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waist below umbilicus. It was soon followed by bilateral lower limb numbness, weakness and symptoms of urinary retention. CEMRI orbit and spine showed soft tissue mass at supero-

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lateral aspect of right orbit and D12-L2 vertebral level respectively (Figure 1 B, 2A). He underwent orbital tissue tru-cut biopsy which showed tissue infiltration by leukemic cells. These atypical cells were positive for myeloperoxidase (MPO) and CD117 on immunostaining. In view of clinical features of neurological compromise secondary to

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compressive myelopathy, he underwent urgent involved field radiotherapy of dorsolumbar spine (12 Gray in 4 fractions) and recieved high dose dexamethasone (96 mg followed by tapering doses). Bone marrow examination subsequently confirmed the diagnosis of acute

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myeloid leukaemia – M 2 type (Figure 3). He was started on AML induction regimen (3 + 7 regimen) with 60mg /m2 OD of daunorubicin for 3 days and 100mg/m 2 BD of cytrabine for 7

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days. With timely intervention by chemo radiotherapy, he showed excellent response with complete resolution of orbital swelling (Figure 1C) and lower limb weakness. Follow up imaging confirmed the improved clinical outcomes at day 28 of chemotherapy (Figure 1D, 2B). Considering high chances of relapse and non-availability of sibling match, he is being planned for matched unrelated donor stem cell transplantation. Joseph JR et al have pointed out very well the need of considering radiation as soon as the diagnosis of spinal compression is made (1). Unlike their case, our case did not have

ACCEPTED MANUSCRIPT recurrent cord compression and responded dramatically to chemo radiotherapy without need of surgical intervention. Spinal involvement by cholorma to the extent of producing symptoms of compressive myelopathy are uncommon and limited to case reports (2, 3). We have also reported isolated cases of chronic myeloid leukaemia who presented with extra

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medullary (EM) blast crisis at paravertebral site (4, 5). Apart from spine, there are rare reports of AML presenting with EM involvement at atypical sites like pancreas, pleura, and mediastinum (6, 7).

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We conclude with the remark that reporting of such unusual cases will not only sensitize

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neurosurgeons to consider AML and related haematological disorders in the differentials of compressive myelopathy but will also hasten treatment initiation. This will certainly minimize the need of surgical interventions and residual neurological deficits in most cases. References

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1.Joseph JR, Wilkinson DA, Bailey NG, Lieberman AP, Tsien CI, Orringer DA. Aggressive Myeloid Sarcoma Causing Recurrent Spinal Cord Compression. World neurosurgery. 2015. Epub 2015/04/19. 2.Eom KS, Kim TY. Intraparenchymal myeloid sarcoma and subsequent spinal myeloid sarcoma for acute myeloblastic leukemia. Journal of Korean Neurosurgical Society. 2011;49(3):171-4. 3.Amalraj P, Syamlal S. Unusual case of paraplegia. Annals of Indian Academy of Neurology. 2009;12(3):188-90. 4.Sahu KK, Malhotra P, Uthamalingam P, Prakash G, Bal A, Varma N, et al. Chronic Myeloid Leukemia with Extramedullary Blast Crisis: Two Unusual Sites with Review of Literature. Indian Journal of Hematology and Blood Transfusion. 2014:1-7. 5.Chauhan S, Suri V, Varma S, Malhotra P, Varma N, Kaur A, et al. Granulocytic sarcoma: an unusual cause of compressive myelopathy. American journal of hematology. 2007;82(7):687-8. 6.Sahu KK, Tyagi R, Law A, Khadwal A, Prakash G, Rajwanshi A, et al. Myeloid Sarcoma: An Unusual Case of Mediastinal Mass and Malignant Pleural Effusion with Review of Literature. Indian Journal of Hematology and Blood Transfusion. 2015:1-6. 7.Tabriz N, Tannapfel A, Griesinger F, Weyhe D. Chloroma of pancreas-initial manifestation of a secondary leukemia after stem cell transplantation case report and review of the literature. Journal of gastrointestinal surgery : official journal of the Society for Surgery of the Alimentary Tract. 2013;17(7):1331-5.

ACCEPTED MANUSCRIPT Legends of Figures Figure 1A & B. Clinical photograph of face of patient from front which shows (A) right eye proptosis and conjunctival chemosis at the time of diagnosis (B) resolution of orbital swelling after chemotherapy

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Figure 1 C & D. CEMRI Orbit showing (C) dense homogenously enhancing extraconal soft tissue density lesion in the supero-lateral aspect of right orbit causing mass effect over the extra ocular muscles {lateral and superior rectus muscles} (D) Follow up images after completion of chemotherapy showing resolution of soft tissue density.

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Figure 2 A, B. CEMRI spine (A) Baseline imaging showing diffuse sheet like enhancing epidural soft tissue, extending from D12-L2 vertebral level with near total ( > 80%) effacement of spinal canal and compression of spinal cord (B) Follow up imaging showing no

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evidence of extradural/intradural mass lesions.

Figure 3. (A) Myeloid blasts in peripheral blood with a few showing Auer rods (MGG x100). (B) And (C) Dysplasia in neutrophils in blood (hypogranulation) and in myelocytes in bone marrow (hypogranulation and differential cytoplasmic staning) (MGG x100). (D) Blasts are

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positive for cytochemical myeloperoxidase (x100).

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Unusual Cause of Cord Compression-A Pressing Issue for Neurosurgeons.

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