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VOLUME 1 , ISSUE 2 ( April-June, 2014 ) > List of Articles

CASE REPORT

Langerhan's Cell Histiocytosis of the Lumbar Spine during Pregnancy: A Rare Case with Literature Review

Rully H Dahlan, Akhmad Y Pramatirta, Agung B Sutiono, Ahmad Faried, Muhammad Z Arifin

Citation Information : Dahlan RH, Pramatirta AY, Sutiono AB, Faried A, Arifin MZ. Langerhan's Cell Histiocytosis of the Lumbar Spine during Pregnancy: A Rare Case with Literature Review. J Spinal Surg 2014; 1 (2):94-97.

DOI: 10.5005/jp-journals-10039-1019

Published Online: 01-09-2016

Copyright Statement:  Copyright © 2014; The Author(s).


Abstract

Background

Langerhan's cell histiocytosis (LCH), previously known as histiocytosis X, is a reactive proliferative dendritic cells of unknown pathogenesis characterized by the proliferation of Langerhan's cells and is extremely rare in the lumbar spines of adults. This condition is most common among young males under the age of 15 years old (with a peak incidence at 2-4 years old), and the most frequent site of these osteolytic bony lesions of the spine is the thoracic region.

Purpose

To highlight an interesting and rare presentation for Langerhans cell histiocytosis of the spinal cord in pregnant woman.

Study design

This is a case report of a single patient in whom a Langerhans cell histiocytosis was resected from the lumbal spine in pregnant woman with return to normal functioning.

Patient sample

A 26-year-old pregnant woman at 20 to 22 weeks presenting with acute cauda equina syndrome, a 1-month history of pain and numbness and paraparese of right limb had gradually progressed to involve all the lower limbs.

Outcome measures

Frankel grading of neural function and Visual Analogue Score are included to evaluate the therapeutic efficiency.

Methods

Magnetic resonance imaging revealed the widespread involvement of an extradural contrast-enhancing mass in the lumbar spine of L2-L5.

Results

The patient underwent decompression and surgical resection of the tumor in a three-quarters prone position, fetal heart monitoring was performed by our obstetrician; there was no fetal distress during the surgery. The diagnosis was confirmed by histological analysis. She entered spontaneous labor at 36 to 37 weeks and birthed a baby weighing 3000 gm. The child began crying immediately and had Apgar scores of 8 and 10 in the 1st and 5th minutes respectively. No residual disease or recurrence was noted at follow-up longer than 1 year.

Conclusion

Our case's unique presentation involves LCH in an adult pregnant patient at the lumbar spine with no osteolytic activity. The management of pregnant women and maintenance of fetal well-being should be coordinated among the spinal neurosurgeon, obstetrician and anesthesiologist.

How to cite this article

Dahlan RH, Yudoyono F, Ompusunggu SE, Pramatirta AY, Sutiono AB, Faried A, Arifin MZ. Langerhan's Cell Histiocytosis of the Lumbar Spine during Pregnancy: A Rare Case with Literature Review. J Spinal Surg 2014;1(2):94-97.


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  1. Langerhans cell histiocytosis in adults: more questions than answers? Eur J Cancer 2004;40(10):1467-1473.
  2. Surgery for lumbar disc herniation during pregnancy. Spine (Phila Pa 1976) 2001;26(4):440-443.
  3. Optimal therapy for adults with langerhans cell histiocytosis bone lesions. PLoS One 2012;7(8):e43257.
  4. Histiocytes and histiocytosis. Blood 1994;84(9):2840-2850.
  5. Fundamentals of operative techniques in neurosurgery. Thieme 2010.
  6. Langerhans cell histiocytosis: malignancy or inflammatory disorder doing a great job of imitating one? Dis Model Mech 2009;2(9-10):436-439.
  7. Percutaneous vertebroplasty for langerhans cell histiocytosis of the lumbar spine in an adult: case report and review of the literature. Exp Ther Med 2013 Jan;5(1):128-132.
  8. Langerhans cell histiocytosis causing cervical myelopathy in a child. J Korean Neurosurg Soc 2010;47(6):458-460.
  9. Expert's comment concerning grand rounds case entitled ‘Langerhan's cell histiocytosis of the atlas in an adult. Eur Spine J 2010 Jan;19(1):23-24.
  10. Histiocytosis X characteristics, behaviour, and treatments as illustrated in a case series. Surg Neurol Int 2011;2:57.
  11. Radiologic manifestations of langerhans cell histiocytosis in pediatrics. Pakistan J Radiol 2010;20(3):114-120.
  12. Multiple involvement of seventh thoracic and fifth lumbar vertebral body with langerhans cell histiocytosis and development of incomplete paraparesis. J Med Sci 2012;32(3):143-146.
  13. Langerhans cell histiocytosis with extensive spinal and thyroid gland involvement presenting with quadriparesis: an unusual case in an adult patient. Malaysian Orthopaedic J 2011;5(3):28-31.
  14. Langerhans cell histiocytosis followed by Hodgkin's lymphoma. Korean J Intern Med 2012;27(4):459-462.
  15. Adult case of Langerhan's cell histiocytosis with single site vertebral involvement. J Musculoskelet Neuronal Interact 2011 Jun;11(2):212-214.
  16. Congenital letterer-siwe disease. Archives of Perinatal Med 2013;19(1):55-57.
  17. Risks and benefits of patient positioning during neurosurgical care. Anesthesiol Clin 2007;25(3):631-653.
  18. Physiological changes in pregnancy. In heart disease in pregnancy, 2nd ed. Celia Oakley C, Warnes CA, editors. Blackwell Publishing 2007. p 6-17.
  19. Adult pulmonary Langerhan's’ cell histiocytosis. Eur Respir J 2006;27(6):1272-1285.
  20. Langerhans’ cell histiocytosis involving posterior elements of the dorsal spine: an unusual cause of extradural spinal mass in an adult. J Craniovertebr Junction Spine 2011;2(2):93-95.
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