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ISSN 2689-8268 Volume 2
American Journal of Surgery and Clinical Case Reports
Case Report Open Access
Vertebroplasty of Primary Vertebral Ewing’s Sarcoma in Child (Case Report)
Hilmani S1*
, Fathi In and Haouass Y
1
Department of Neurosurgery, Hassan II university, Casablanca, Morocco
*Corresponding author:
Said Hilmani,
Department of Neurosurgery, Hassan II University,
UHC Casablanca, CP 20330, Casablanca,
Morocco, E-mail: hilmani.said@yahoo.fr
Received: 07 Jan 2021
Accepted: 27 Jan 2021
Published: 01 Feb 2021
Copyright:
©2021 Hilmani S. This is an open access article distribut-
ed under the terms of the Creative Commons Attribution
License, which permits unrestricted use, distribution, and
build upon your work non-commercially.
Citation:
Hilmani S, Vertebroplasty of Primary Vertebral Ewing’s
Sarcoma in Child (Case Report). Ame J Surg Clin Case
Rep.2021; 2(5): 1-3.
Keywords:
Ewing’s sarcoma, Primary spinal tumor,
Vertebroplasty
1. Abstract
1.1. Purpose: Ewing's sarcoma is a malignant tumor that affects
young people. Primary vertebral involvement is extremely rare
and is redoubtable due to its high metastatic potential. The aim is
to present the advantage of vertebroplasty of this lesion which has
never been reported in child.
1.2. Case Report: We report here the case of 16-year-old child
suffering from isolated low back pain and whose radiological ex-
plorations showed a single involvement of L4 with radicular com-
pression and epiduritis. He underwent a laminectomy with biopsy
followed by vertebroplasty of the affected vertebra and lumbar
fusion. This conducts to a considerable reduction in pain and an
improvement in mobility. The histopathology study confirmed the
diagnosis of Ewing's sarcoma. The patient was referred for the ad-
juvant therapy. In the last reevaluation, there was no sign of tumor
recurrence.
1.3. Conclusion: Vertebroplasty of primary spinal Ewing’s sarco-
ma in child is an effective alternative to en bloc resection and can
represent a better prognosis factor for survival than decompression
and fusion alone.
2. Introduction
Primary malignant tumors of the spine account for less than 5% of
primary bone tumors [1]. The primary vertebral location of Ew-
ing's Sarcoma (ES) is extremely rare, affecting adolescents and
young adults with a male predominance and a peak incidence be-
tween 10 and 20 years [2]. This malignant tumor is redoubtable by
its high metastatic potential and poor prognosis [3].
Conventional treatment is based on surgery, chemotherapy and ra-
diotherapy. Vertebroplasty is classically used in adult ES [4], but
referring to existing literature, it was never reported in child.
In this article, we describe our first successful experience and re-
viewing the advantages of vertebroplasty of lumbar ES in child.
3. Case Presentation
A 16 years-old child with 3months history of intense back pain, re-
sisting to analgesic therapy without spinal trauma. Clinical exam-
ination revealed isolated severe back pain (graded EVA= 8) asso-
ciated to back leaning forward when walking (analgesic attitude)
without neurological abnormalities (Frankel E). MRI showed a
vertebral collapse of L4 with an extending epiduritis massfromL3
to L4. The mass was isointense on T1, T2 without enhanced con-
trast and cauda equina compression (Figure 1A and 1B).
Additional assessment based on the thoraco-abdominopelvic CT
and biological examination did not reveal evidence of metastatic
disease. The patient underwent L3-L4 laminectomy and excision
of the epidural mass. We proceeded with a biopsy of L4 vertebra
and we finished the procedure with an open L4 vertebroplasty us-
ing methacrylate cement and L3-L5 fusion (Figure 2). The pathol-
ogy and immunehistochemical studies confirmed the diagnosis of
Ewing's sarcoma. After surgery, we noticed a good improvement
of the back pain (EVA 2) and he can walk normally. The patient
was referred for the adjuvant therapy. In the last reevaluation, he
experienced completere solution of back pain, and there was no
radiological sign of tumor recurrence.
Volume 2 | Issue 5
Figure 1: MRI T1-WI (a) and T1-WI after gadolinium (b) revealed lyti-
clesion of L4 with iso-signal intensity without enhancing after gadolinium
injection and epiduritis extending of L3-L4
Figure 2: Post-operative X-ray, lateral and AP, after spinal fusion and L4
vertebroplasty
4. Discussion
We describe a first case never previously reported of vertebroplas-
ty for primary spinal ES in child. Ewing's sarcoma is a primary
malignant bone tumor, which predominantly affects children and
adolescents. The primary vertebral location of Ewing's sarcoma is
extremely rare, accounting for only 3.5 to 5% (1.5). The Involve-
ment of the non sacrals pinere presents only less than 1% of all
primary spinal ES [6]. Primary vertebral Ewing's sarcoma is often
revealed by back pain. Spinal cord compression is often delayed
presentation. Deterioration of general condition, as well as the
presence of fever, is not uncommon and testifies to metastatic dis-
semination. Our patient only suffered from isolated low back pain,
without neurological deficit. No radiological sign is pathognomon-
ic in Ewing's sarcoma [7], but images of osteolysis with vertebral
compression associated to epiduritis are often found, which is the
case in our study with the presence of an osteolytic compression of
L4. This is important to choose the best surgical strategy.
The current standard treatment involves chemotherapy and local
disease control with surgery and radiotherapy [8.9]. In comparison
to ES in adult, the age is a controversial prognosis factor and could
not influence the survival rate [10]. The most significant factors to
determine the prognosis are the presence of metastatic disease and
local control of the primary site [3, 7]. Local disease control can
be achieved by en-bloc resection and fusion, but in spinal location
this technic is challenging. To increase this local control, vertebro-
plasty could be an alternative procedure to en-bloc resection. Ver-
tebroplasty is a widely used technique in adults4butrarely reported
in children. The indications in pediatric population are mainly ver-
tebral fractures and benign tumors such as vertebral hemangiomas
[11, 12]. To our knowledge, vertebroplasty has never been per-
formed for Ewing's sarcoma in children. We opted for this choice
given the symptomatology dominated by pain and significant com-
pression of the vertebra. Vertebroplasty is an easy and save tech-
nic that has a sclerosing effect that can kill tumors via local high
temperature and cytotoxicity [13]. It allows also to increase ver-
tebral height, participate to stability and alleviate the spinal pain.
However, special attention should be taken during vertebroplasty
for cement leakage at the posterior margin of the vertebral body
caused by excessive injection and increased local pressure in de-
stroyed and fragile vertebra. We believe that it should be restricted
only to carefully selected patients.
5. Conclusion
To the best of our knowledge our case represents the youngest age
of vertebroplasty reported for primary spinal Ewing’s sarcoma in
child and suggests that can have a better prognosis for survival
than decompression and fusion alone. However, further prospec-
tive studies are required to tailor better evidence-based treatment
guidelines for indication and to achieve a better prognosis.
References:
1.	 Sundaresan N, Rosen G, Boriani S. Primary malignant tumors of the
spine. Orthop Clin North Am. 2009; 40: 21-36.
2.	 Hense HW, Ahrens S, Paulussen M, Lehnert M, Jurgens H. Descrip-
tive epidemiology of Ewing’stumor--analysis of German patients
from (EI) CESS 1980-1997. KlinischePadiatrie.1999; 211: 271-5.
3.	 Cash T, McIlvaine E, Krailo MD, Lessnick SL, Lawlor ER, Laack
N, et al. Comparison of clinicalfeatures and outcomes in patients
withextraskeletal versus skeletallocalized Ewing sarcoma: A report
from the Children’s Oncology Group. Pediatr Blood Cancer. 2016;
63: 1771-9.
4.	 Zhao Dy, Zhang WL, Cui Y, CMa CB, Ni SH, Zhang ZY. Primary
Ewing’s sarcoma of the vertebral body (A case report). Medicine
(Baltimore). 2019; 98: e15905.
5.	 Ganapathy S, Subramaniam V, Baliga V. Spinal Ewing’sSarcoma-
Presenting as an Epidural Collection: A Rare Presentation of a Rare
Entity. Asian J Neurosurg. 2020; 15: 445–8.
6.	 Bemporad JA, Sze G, Chaloupka JC, Duncan C. Pseudo hemangi-
Volume 2 | Issue 5
ajsccr.org 2
oma of the vertebra: An unusualradiographic manifestation of pri-
maryEwing’ssarcoma. AJNR Am J Neuroradiol. 1999; 20: 1809-13.
7.	 Gopalakrishnan CV, Shrivastava A, Easwer HV, Nair S. PrimaryE-
wing’ssarcoma of the spinepresenting as acute paraplegia. J Pediatr-
Neurosci. 2012; 7: 64-6.
8.	 Electricwala AJ, Electricwala JT. Primary Ewing’s Sarcoma of the
Spine in a Two-Year-Old Boy. Case Rep in Orthop. 2016; 2016:
8027137.
9.	 Zhang J, Huang Y, Lu J, He A, Zhou Y, Hu H, et al. Impact of first-
line treatment on outcomes of Ewing sarcoma of the spine. Am J
Cancer Res. 2018; 8: 1262-72.
10.	 Shoubash L, Nowak S, Vogelgesang S, Schroeder HW, Muller JU.
Surgical management of an adult manifestation of Ewing sarcoma of
the spine-a case report. AME Case Rep 2018; 2: 34.
11.	 Issa M, Lucas G, Violas P, Griffet J, Courvoisier A. Vertebroplas-
ty for vertebralhemangioma in children:a report of two cases with
2-year follow-up. Child s Nervous System. 2015; 31: 2179-83.
12.	 Polis B, Krawczyk J, Polis L, Nowosławska E. Percutaneous extra-
pedicular vertebroplasty with expandable intravertebral implant in
compression vertebral body fracture in pediatric patient-technical
note. ChildsNervSyst. 2016; 32: 2225-31.
13.	 Belkoff SM, Molloy S. Temperature measurement during polymer-
ization of polymethylmethacrylate cement used for vertebroplasty.
Spine. 2003; 28: 1555-9.
Volume 2 | Issue 5
ajsccr.org 3

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Vertebroplasty of Primary Vertebral Ewing’s Sarcoma in Child (Case Report)

  • 1. ISSN 2689-8268 Volume 2 American Journal of Surgery and Clinical Case Reports Case Report Open Access Vertebroplasty of Primary Vertebral Ewing’s Sarcoma in Child (Case Report) Hilmani S1* , Fathi In and Haouass Y 1 Department of Neurosurgery, Hassan II university, Casablanca, Morocco *Corresponding author: Said Hilmani, Department of Neurosurgery, Hassan II University, UHC Casablanca, CP 20330, Casablanca, Morocco, E-mail: hilmani.said@yahoo.fr Received: 07 Jan 2021 Accepted: 27 Jan 2021 Published: 01 Feb 2021 Copyright: ©2021 Hilmani S. This is an open access article distribut- ed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and build upon your work non-commercially. Citation: Hilmani S, Vertebroplasty of Primary Vertebral Ewing’s Sarcoma in Child (Case Report). Ame J Surg Clin Case Rep.2021; 2(5): 1-3. Keywords: Ewing’s sarcoma, Primary spinal tumor, Vertebroplasty 1. Abstract 1.1. Purpose: Ewing's sarcoma is a malignant tumor that affects young people. Primary vertebral involvement is extremely rare and is redoubtable due to its high metastatic potential. The aim is to present the advantage of vertebroplasty of this lesion which has never been reported in child. 1.2. Case Report: We report here the case of 16-year-old child suffering from isolated low back pain and whose radiological ex- plorations showed a single involvement of L4 with radicular com- pression and epiduritis. He underwent a laminectomy with biopsy followed by vertebroplasty of the affected vertebra and lumbar fusion. This conducts to a considerable reduction in pain and an improvement in mobility. The histopathology study confirmed the diagnosis of Ewing's sarcoma. The patient was referred for the ad- juvant therapy. In the last reevaluation, there was no sign of tumor recurrence. 1.3. Conclusion: Vertebroplasty of primary spinal Ewing’s sarco- ma in child is an effective alternative to en bloc resection and can represent a better prognosis factor for survival than decompression and fusion alone. 2. Introduction Primary malignant tumors of the spine account for less than 5% of primary bone tumors [1]. The primary vertebral location of Ew- ing's Sarcoma (ES) is extremely rare, affecting adolescents and young adults with a male predominance and a peak incidence be- tween 10 and 20 years [2]. This malignant tumor is redoubtable by its high metastatic potential and poor prognosis [3]. Conventional treatment is based on surgery, chemotherapy and ra- diotherapy. Vertebroplasty is classically used in adult ES [4], but referring to existing literature, it was never reported in child. In this article, we describe our first successful experience and re- viewing the advantages of vertebroplasty of lumbar ES in child. 3. Case Presentation A 16 years-old child with 3months history of intense back pain, re- sisting to analgesic therapy without spinal trauma. Clinical exam- ination revealed isolated severe back pain (graded EVA= 8) asso- ciated to back leaning forward when walking (analgesic attitude) without neurological abnormalities (Frankel E). MRI showed a vertebral collapse of L4 with an extending epiduritis massfromL3 to L4. The mass was isointense on T1, T2 without enhanced con- trast and cauda equina compression (Figure 1A and 1B). Additional assessment based on the thoraco-abdominopelvic CT and biological examination did not reveal evidence of metastatic disease. The patient underwent L3-L4 laminectomy and excision of the epidural mass. We proceeded with a biopsy of L4 vertebra and we finished the procedure with an open L4 vertebroplasty us- ing methacrylate cement and L3-L5 fusion (Figure 2). The pathol- ogy and immunehistochemical studies confirmed the diagnosis of Ewing's sarcoma. After surgery, we noticed a good improvement of the back pain (EVA 2) and he can walk normally. The patient was referred for the adjuvant therapy. In the last reevaluation, he experienced completere solution of back pain, and there was no radiological sign of tumor recurrence. Volume 2 | Issue 5
  • 2. Figure 1: MRI T1-WI (a) and T1-WI after gadolinium (b) revealed lyti- clesion of L4 with iso-signal intensity without enhancing after gadolinium injection and epiduritis extending of L3-L4 Figure 2: Post-operative X-ray, lateral and AP, after spinal fusion and L4 vertebroplasty 4. Discussion We describe a first case never previously reported of vertebroplas- ty for primary spinal ES in child. Ewing's sarcoma is a primary malignant bone tumor, which predominantly affects children and adolescents. The primary vertebral location of Ewing's sarcoma is extremely rare, accounting for only 3.5 to 5% (1.5). The Involve- ment of the non sacrals pinere presents only less than 1% of all primary spinal ES [6]. Primary vertebral Ewing's sarcoma is often revealed by back pain. Spinal cord compression is often delayed presentation. Deterioration of general condition, as well as the presence of fever, is not uncommon and testifies to metastatic dis- semination. Our patient only suffered from isolated low back pain, without neurological deficit. No radiological sign is pathognomon- ic in Ewing's sarcoma [7], but images of osteolysis with vertebral compression associated to epiduritis are often found, which is the case in our study with the presence of an osteolytic compression of L4. This is important to choose the best surgical strategy. The current standard treatment involves chemotherapy and local disease control with surgery and radiotherapy [8.9]. In comparison to ES in adult, the age is a controversial prognosis factor and could not influence the survival rate [10]. The most significant factors to determine the prognosis are the presence of metastatic disease and local control of the primary site [3, 7]. Local disease control can be achieved by en-bloc resection and fusion, but in spinal location this technic is challenging. To increase this local control, vertebro- plasty could be an alternative procedure to en-bloc resection. Ver- tebroplasty is a widely used technique in adults4butrarely reported in children. The indications in pediatric population are mainly ver- tebral fractures and benign tumors such as vertebral hemangiomas [11, 12]. To our knowledge, vertebroplasty has never been per- formed for Ewing's sarcoma in children. We opted for this choice given the symptomatology dominated by pain and significant com- pression of the vertebra. Vertebroplasty is an easy and save tech- nic that has a sclerosing effect that can kill tumors via local high temperature and cytotoxicity [13]. It allows also to increase ver- tebral height, participate to stability and alleviate the spinal pain. However, special attention should be taken during vertebroplasty for cement leakage at the posterior margin of the vertebral body caused by excessive injection and increased local pressure in de- stroyed and fragile vertebra. We believe that it should be restricted only to carefully selected patients. 5. Conclusion To the best of our knowledge our case represents the youngest age of vertebroplasty reported for primary spinal Ewing’s sarcoma in child and suggests that can have a better prognosis for survival than decompression and fusion alone. However, further prospec- tive studies are required to tailor better evidence-based treatment guidelines for indication and to achieve a better prognosis. References: 1. Sundaresan N, Rosen G, Boriani S. Primary malignant tumors of the spine. Orthop Clin North Am. 2009; 40: 21-36. 2. Hense HW, Ahrens S, Paulussen M, Lehnert M, Jurgens H. Descrip- tive epidemiology of Ewing’stumor--analysis of German patients from (EI) CESS 1980-1997. KlinischePadiatrie.1999; 211: 271-5. 3. Cash T, McIlvaine E, Krailo MD, Lessnick SL, Lawlor ER, Laack N, et al. Comparison of clinicalfeatures and outcomes in patients withextraskeletal versus skeletallocalized Ewing sarcoma: A report from the Children’s Oncology Group. Pediatr Blood Cancer. 2016; 63: 1771-9. 4. Zhao Dy, Zhang WL, Cui Y, CMa CB, Ni SH, Zhang ZY. Primary Ewing’s sarcoma of the vertebral body (A case report). Medicine (Baltimore). 2019; 98: e15905. 5. Ganapathy S, Subramaniam V, Baliga V. Spinal Ewing’sSarcoma- Presenting as an Epidural Collection: A Rare Presentation of a Rare Entity. Asian J Neurosurg. 2020; 15: 445–8. 6. Bemporad JA, Sze G, Chaloupka JC, Duncan C. Pseudo hemangi- Volume 2 | Issue 5 ajsccr.org 2
  • 3. oma of the vertebra: An unusualradiographic manifestation of pri- maryEwing’ssarcoma. AJNR Am J Neuroradiol. 1999; 20: 1809-13. 7. Gopalakrishnan CV, Shrivastava A, Easwer HV, Nair S. PrimaryE- wing’ssarcoma of the spinepresenting as acute paraplegia. J Pediatr- Neurosci. 2012; 7: 64-6. 8. Electricwala AJ, Electricwala JT. Primary Ewing’s Sarcoma of the Spine in a Two-Year-Old Boy. Case Rep in Orthop. 2016; 2016: 8027137. 9. Zhang J, Huang Y, Lu J, He A, Zhou Y, Hu H, et al. Impact of first- line treatment on outcomes of Ewing sarcoma of the spine. Am J Cancer Res. 2018; 8: 1262-72. 10. Shoubash L, Nowak S, Vogelgesang S, Schroeder HW, Muller JU. Surgical management of an adult manifestation of Ewing sarcoma of the spine-a case report. AME Case Rep 2018; 2: 34. 11. Issa M, Lucas G, Violas P, Griffet J, Courvoisier A. Vertebroplas- ty for vertebralhemangioma in children:a report of two cases with 2-year follow-up. Child s Nervous System. 2015; 31: 2179-83. 12. Polis B, Krawczyk J, Polis L, Nowosławska E. Percutaneous extra- pedicular vertebroplasty with expandable intravertebral implant in compression vertebral body fracture in pediatric patient-technical note. ChildsNervSyst. 2016; 32: 2225-31. 13. Belkoff SM, Molloy S. Temperature measurement during polymer- ization of polymethylmethacrylate cement used for vertebroplasty. Spine. 2003; 28: 1555-9. Volume 2 | Issue 5 ajsccr.org 3