Orthopedics

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Case Report 

Symptomatic Spinal Epidural Lipomatosis with Severe Obesity at a Young Age

Tetsuro Ohba, MD, PhD; Toshiki Saito, MD, PhD; Nobuchika Kawasaki, MD, PhD; Shingo Maekawa, MD, PhD; Hirotaka Haro, MD, PhD

Abstract

Symptomatic spinal epidural lipomatosis is a rare disorder characterized by overgrowth of fat in the extradural space. Most patients have an underlying endocrine disorder, such as Cushing’s syndrome, or have taken exogenous steroids chronically. Although less common, obesity alone is thought to be a cause of spinal epidural lipomatosis, representing <25% of reported cases. Patients rarely become symptomatic before middle age without chronic exogenous steroid use. The usual clinical manifestations are similar to degenerative lumbar stenosis with neurogenic claudication, resulting in decreased walking and standing endurance with variable neurological deficits.

This article describes 2 unique cases of spinal epidural lipomatosis, both in young patients with underlying morbid obesity who presented with acute progressive leg weakness and urinary retention. The patients had no underlying endocrinopathy, nor any history of exogenous steroid use. They underwent emergency laminectomy and removal of epidural fat, and histopathological examination confirmed the diagnosis of epidural lipomatosis. Postoperatively, the patients demonstrated significant improvement.

We conducted a review of the available English literature and compared the age distribution in each group. Based on our review, our 2 patients are considerably younger than those in past reports, especially in the patient group to which the steroid was not administered. In addition, few cases exist of spinal epidural lipomatosis with acute sphincter dysfunction and paraparesis. Our cases suggest that morbid obesity can lead to juvenile spinal epidural lipomatosis with acute neurological changes.

Drs Ohba, Saito, Kawasaki, Maekawa, and Haro are from the Department of Orthopedic Surgery, University of Yamanashi, Japan.

Drs Ohba, Saito, Kawasaki, Maekawa, and Haro have no relevant financial relationships to disclose.

Spinal epidural lipomatosis is a rare condition characterized by an excessive deposition of mature a dipose tissue that proliferates in an infiltrative manner. Most patients have an underlying endocrine disorder, such as Cushing’s syndrome, or have taken exogenous steroids chronically. 1,2 Although less common, obesity alone is thought to be a cause of spinal epidural lipomatosis, representing <25% of reported cases. 1 The majority of these patients are middle-aged or elderly. 2–5 Juvenile spinal epidural lipomatosis is especially rare in the absence of exogenous steroid use.

This article describes 2 unique cases of spinal epidural lipomatosis, both in young patients with underlying morbid obesity who presented with acute progressive weakness and urinary retention. The patients had no underlying endocrinopathy, nor any history of exogenous steroid use. They underwent emergency laminectomy and removal of epidural fat. Postoperatively, both patients demonstrated significant improvement.

A 19-year-old man presented with a 1-year history of low back pain and bilateral thigh numbness. Prior to his visit to our hospital, he had developed progressive bilateral lower extremity weakness over 1 week, with 48 hours of urinary retention.

The patient was obese (body mass index [BMI]=45.4) and had no evidence of clinical spinal deformity. Neurological examination revealed bilateral lower extremity weakness throughout (power 2/5), numbness on the anterior thighs, and symmetric hyporeflexia. Magnetic resonance imaging (MRI) of the lumbosacral spine revealed multilevel degenerative changes and spinal stenosis. An axial view revealed epidural fat extending from L1 to S1 (Figure ).

Figure 1:. Sagittal (A) and Axial (B) MRIs Showing Epidural Lipomatosis Compressing the Spinal Cord.

An urgent laminectomy was performed from L1 to S1. Intraoperatively, the thecal sac and bilateral L5 nerve roots were seen to have excessive focal compression because of adipose tissue. Epidural fat was completely removed, and histopathological examination confirmed our diagnosis of epidural lipomatosis (Figure ).

Figure 2:. Photomicrograph Showing Mature Adipocytes. Hematoxylin-Eosin Staining.

Postoperatively, the patient demonstrated gradual improvement and recovered bladder function in 2 weeks. At 12-month follow-up, the patient demonstrated normal muscular strength and bladder function with…

Symptomatic Spinal Epidural Lipomatosis with Severe Obesity at a Young Age

Abstract

Symptomatic spinal epidural lipomatosis is a rare disorder characterized by overgrowth of fat in the extradural space. Most patients have an underlying endocrine disorder, such as Cushing’s syndrome, or have taken exogenous steroids chronically. Although less common, obesity alone is thought to be a cause of spinal epidural lipomatosis, representing <25% of reported cases. Patients rarely become symptomatic before middle age without chronic exogenous steroid use. The usual clinical manifestations are similar to degenerative lumbar stenosis with neurogenic claudication, resulting in decreased walking and standing endurance with variable neurological deficits.

This article describes 2 unique cases of spinal epidural lipomatosis, both in young patients with underlying morbid obesity who presented with acute progressive leg weakness and urinary retention. The patients had no underlying endocrinopathy, nor any history of exogenous steroid use. They underwent emergency laminectomy and removal of epidural fat, and histopathological examination confirmed the diagnosis of epidural lipomatosis. Postoperatively, the patients demonstrated significant improvement.

We conducted a review of the available English literature and compared the age distribution in each group. Based on our review, our 2 patients are considerably younger than those in past reports, especially in the patient group to which the steroid was not administered. In addition, few cases exist of spinal epidural lipomatosis with acute sphincter dysfunction and paraparesis. Our cases suggest that morbid obesity can lead to juvenile spinal epidural lipomatosis with acute neurological changes.

Drs Ohba, Saito, Kawasaki, Maekawa, and Haro are from the Department of Orthopedic Surgery, University of Yamanashi, Japan.

Drs Ohba, Saito, Kawasaki, Maekawa, and Haro have no relevant financial relationships to disclose.

Correspondence should be addressed to: Hirotaka Haro, MD, PhD, Department of Orthopedic Surgery, University of Yamanashi, 1110 Shimokato, Chuo, Yamanashi 409-3898, Japan (haro@yamanashi.ac.jp).
Posted Online: June 14, 2011

Spinal epidural lipomatosis is a rare condition characterized by an excessive deposition of mature a dipose tissue that proliferates in an infiltrative manner. Most patients have an underlying endocrine disorder, such as Cushing’s syndrome, or have taken exogenous steroids chronically. 1,2 Although less common, obesity alone is thought to be a cause of spinal epidural lipomatosis, representing <25% of reported cases. 1 The majority of these patients are middle-aged or elderly. 2–5 Juvenile spinal epidural lipomatosis is especially rare in the absence of exogenous steroid use.

This article describes 2 unique cases of spinal epidural lipomatosis, both in young patients with underlying morbid obesity who presented with acute progressive weakness and urinary retention. The patients had no underlying endocrinopathy, nor any history of exogenous steroid use. They underwent emergency laminectomy and removal of epidural fat. Postoperatively, both patients demonstrated significant improvement.

Case Reports

Patient 1

A 19-year-old man presented with a 1-year history of low back pain and bilateral thigh numbness. Prior to his visit to our hospital, he had developed progressive bilateral lower extremity weakness over 1 week, with 48 hours of urinary retention.

The patient was obese (body mass index [BMI]=45.4) and had no evidence of clinical spinal deformity. Neurological examination revealed bilateral lower extremity weakness throughout (power 2/5), numbness on the anterior thighs, and symmetric hyporeflexia. Magnetic resonance imaging (MRI) of the lumbosacral spine revealed multilevel degenerative changes and spinal stenosis. An axial view revealed epidural fat extending from L1 to S1 (Figure ).

Sagittal (A) and Axial (B) MRIs Showing Epidural Lipomatosis Compressing the Spinal Cord.

Figure 1:. Sagittal (A) and Axial (B) MRIs Showing Epidural Lipomatosis Compressing the Spinal Cord.

An urgent laminectomy was performed from L1 to S1. Intraoperatively, the thecal sac and bilateral L5 nerve roots were seen to have excessive focal compression because of adipose tissue. Epidural fat was completely removed, and histopathological examination confirmed our diagnosis of epidural lipomatosis (Figure ).

Photomicrograph Showing Mature Adipocytes. Hematoxylin-Eosin Staining.

Figure 2:. Photomicrograph Showing Mature Adipocytes. Hematoxylin-Eosin Staining.

Postoperatively, the patient demonstrated gradual improvement and recovered bladder function in 2 weeks. At 12-month follow-up, the patient demonstrated normal muscular strength and bladder function with some mild residual thigh numbness.

Patient 2

A 22-year-old man presented with a 2-year history of neurogenic back and bilateral lower extremity pain. He underwent a series of 4 epidural steroid injections with improvement of pain. Prior to presentation, he had a 1-month history of progressive weakness in the right lower extremity and 1 week of difficulty initiating a stream during urination.

On physical examination, the patient was 179 cm tall and weighed 167 kg (BMI=52.1). He had no evidence of clinical spinal deformity and no history of steroid use or an endocrinopathy. Neurological examination revealed right lower extremity weakness (power 2/5), thigh numbness, and hyporeflexia throughout. Magnetic resonance imaging of the lumbosacral spine revealed multilevel degenerative changes and spinal stenosis secondary to extensive epidural fat extending from L2 to S1.

Urgent laminectomy was performed from L1 to S1. Epidural fat was completely removed, and histopathological examination confirmed a diagnosis of epidural lipomatosis.

Postoperatively, the patient was able to void at 2 weeks and had a full return of muscle strength with only mild residual numbness at most recent follow-up.

Discussion

Spinal epidural lipomatosis is a rare disease. It was first reported by Lee et al 6 in 1975 as occurring in a patient following a renal transplant. Thereafter, approximately 100 reports of spinal epidural lipomatosis have been published, but the underlying pathological mechanism of spinal epidural lipomatosis remains unknown. The underlying cause was classified in 4 groups as exogenous steroid use (55.3%), endogenous elevation of steroids (3.2%), obesity (24.5%), and idiopathic (17%). 1 Moreover, spinal epidural lipomatosis is a disease that affects people later in adulthood, with an average age of 46 years at presentation, influenced by denaturation of the spine. 2 In 1 of the few reported cases in young people, George et al 7 described a child with steroid-induced spinal epidural lipomatosis following a renal transplant.

We conducted a review of the available English literature and compared the age distribution in each group. We found 134 cases of spinal epidural lipomatosis reported in the literature, including our 2 cases (Figure ). Based on our review, the average age of patients with spinal epidural lipomatosis attributed to exogenous steroid use is 40.1 years, vs 51.4 years for patients with spinal epidural lipomatosis attributed to obesity.

Age Distribution of 134 Cases of Spinal Epidural Lipomatosis in 4 Groups. The Black Line and Number Shows the Average Age of Each Group.

Figure 3:. Age Distribution of 134 Cases of Spinal Epidural Lipomatosis in 4 Groups. The Black Line and Number Shows the Average Age of Each Group.

Juvenile spinal epidural lipomatosis is described in patients taking exogenous steroids for rheumatoid arthritis or as a means of immunosuppression in organ transplants. In the exogenous steroid use group, juvenile spinal epidural lipomatosis is a result of juvenile rheumatism or organ transplant. 6–9 Both of our patients were young, with no history of exogenous steroid use or an underlying endocrinopathy (Figure ).

Epidural lipomatosis is typically a slowly progressive disorder similar to degenerative spinal stenosis. 10 In contrast, our 2 patients had an acute, progressive neurological deterioration with bilateral lower extremity paraplegia and bladder dysfunction. To our knowledge, few cases of spinal epidural lipomatosis present with acute sphincter dysfunction and paraparesis. The treatment of spinal epidural lipomatosis depends on the severity of the symptoms. It ranges from conservative therapy to surgical decompression. Conservative care includes weight loss, 5 steroid tapering, 11 analgesics, bed rest, and observation, which is approximately 80% effective. 1 Conservative care should be attempted unless a progressive neurological deficit is present, as was the case in our 2 patients. Neurological improvement has been reported providing urgent decompression is performed. 10

Acknowledgments

The authors thank Takaski Ando, MD, PhD; and Jiro Ichikawa, MD, PhD, Department of Orthopedic Surgery, University of Yamanashi, Japan; and Clinton Devin, MD; Jonathan Schoenecker, MD, PhD; and Lynda Orear, Department of Orthopedics and Rehabilitation, Vanderbilt University, Nashville, Tennessee.

References

  1. 1. Fogel GR, Cunningham PY, IIIEsses SI. Spinal epidural lipomatosis: case reports, literature review and meta-analysis. Spine J. 2005; 5(2):202–211. doi: 10.1016/j.spinee.2004.05.252 [CrossRef]
  2. 2. Al-Khawaja D, Seex K, Eslick GD. Spinal epidural lipomatosis—a brief review [published online ahead of print October 26, 2008]. J Clin Neurosci. 2008; 15(12):1323–1326.
  3. 3. Maillot F, Mulleman D, Mammou S, Goupille P, Valat JP. Is epidural lipomatosis associated with abnormality of body fat distribution? A case report [published online ahead of print July 9, 2005]. Eur Spine J. 2006; 15(1):105–108. doi: 10.1007/s00586-005-0955-x [CrossRef]
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  5. 5. Beges C, Rousselin B, Chevrot A, et al. Epidural lipomatosis. Interest of magnetic resonance imaging in a weight-reduction treated case. Spine (Phila Pa 1976). 1994; 19(2):251–254. doi: 10.1097/00007632-199401001-00022 [CrossRef]
  6. 6. Lee M, Lekias J, Gubbay SS, Hurst PE. Spinal cord compression by extradural fat after renal transplantation. Med J Aust. 1975; 1(7):201–203.
  7. 7. George WE, JrWilmot M, Greenhouse A, Hammeke M. Medical management of steroid-induced epidural lipomatosis. N Engl J Med. 1983; 308(6):316–319. doi: 10.1056/NEJM198302103080605 [CrossRef]
  8. 8. Guegan Y, Fardoun R, Launois B, Pecker J. Spinal cord compression by extradural fat after prolonged corticosteroid therapy. J Neurosurg. 1982; 56(2):267–269. doi: 10.3171/jns.1982.56.2.0267 [CrossRef]
  9. 9. Arroyo IL, Barron KS, Brewer EJJr. Spinal cord compression by epidural lipomatosis in juvenile rheumatoid arthritis. Arthritis Rheum. 1988; 31(3):447–451. doi: 10.1002/art.1780310320 [CrossRef]
  10. 10. López-González A, Resurrección Giner M. Idiopathic spinal epidural lipomatosis: urgent decompression in an atypical case [published online ahead of print September 18, 2007]. Eur Spine J. 2008; 17(Suppl 2):S225–227. doi: 10.1007/s00586-007-0465-0 [CrossRef]
  11. 11. Crayton HE, Partington CR, Bell CL. Spinal cord compression by epidural lipomatosis in a patient with systemic lupus erythematosus. Arthritis Rheum. 1992; 35(4):482–484. doi: 10.1002/art.1780350422 [CrossRef]

10.3928/01477447-20110427-25

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