Article

Ligamentum flavum hematoma due to stretching exercise

Abstract

Ligamentum flavum hematoma is a rare cause of spinal cord or root compression and usually occurs in the lower thoracic and lumbar spine. The differential diagnosis includes lumbar disk herniation and lumbar spinal canal stenosis caused by neoplasm, infection, epidural hematoma, and facet cyst.

Herein we present a case of ligamentum flavum hematoma of the lumbar spine. To date, a review of the English literature revealed only 30 cases, including our case, of hematoma in the lumbar ligamentum flavum. In case of ligamentum flavum hematoma, surgical excision of the lesion was recommended. The patient underwent laminectomy with excision of the epidural lesion from L4 to L5 region and led an uneventful postoperative hospital course. Histopathology revealed hematoma with fibrotic change of the ligamentum flavum.

A 76-year-old man with history of hypertension and type 2 diabetes mellitus with regular medication presented to the emergency department with a 1-day duration of dizziness and nausea, and general malaise. He also complaint about progressive low back pain and bilateral lower limb numbness and weakness for more than 1 month. The weakness deteriorated to bedridden status. He had practiced stretching exercise as hyperflexion (bending upper trunk forward) and hyperextension (bending upper trunk backward) of waist every day for many years. He denied trauma history in the past few months. He consulted local clinics and medical center for help, but after series examination, the results were unremarkable.

He called at our emergency department where physical examination showed tenderness in the paraspinal region at the level of the lower lumbar spine and limited range of motion of back region without petechiae or ecchymosis. Neurologic examination showed grade 5 as for muscle power in the upper limbs and grade 3 in the lower limbs (Medical Research Council grading), and sensory deficits were found bilateral lower extremities anesthesia below the L4 dermatome. Laboratory data were normal except thrombocytopenia with platelet count of 121 000/uL. The patient was impressed with dengue fever with positive NS-1 antigen results. Hence, he was admitted to infection division for further evaluation. There was no history of significant recent trauma, lumbar surgery, lumbar puncture, or antiplatelet/coagulation therapy. Routine laboratory and coagulatory parameters were within normal limits.

After hospitalization, he was in apparent distress due to dizziness,

nausea, and weakness, although vital signs were stable. Screening tests of influenza and dengue fever, and blood and urine culture all showed negative results. Radiography of L spine showed grade I retrolisthesis over L2 on L3 and L3 on L4, which is not compatible with

the presentation as paraparesis. Despite rehabilitation and muscle relaxant, his symptoms persisted and muscle power of the lower limbs dropped from grade 3 to grade 2.

L-spine magnetic resonance imaging (MRI) was done after the suggestion of neurosurgical specialist, which showed an intrathecal cyst-like lesion with adjacent retrospinal soft tissue enhancement at L4-L5 level with severe thecal sac compression and severe encroach- ment of the bilateral neural foramina (Fig. 1).

Surgical decompression with L4 to L5 laminectomies was performed. During operation, an old hematoma in fibrous change was found at retrospinal space of L4-5 originated from ligamentum flavum with spinal cord compression (Fig. 2). The hematoma was completely removed during operation without any complication.

Samples of tissue and hematoma were sent for bacteriologic culture and pathologic analysis which showed an old hematoma in fibrotic tissue containing elastic fibers with unremarkable microbiology and neoplastic findings.

The patient had led an uneventful postoperative hospital course with partial sensory recover with 4 limbs and muscle power regained to grade 4. He had regained full recovery without any neurologic deficits 1 month later. The follow-up L-spine MRI showed total remission of cord compression 3 months later (Fig. 3).

The ligamenum flavum is a discontinuous structure from the axis to sacrum; it helps to maintain an upright posture and resume an upright posture after bending [1].

Ligamentum flavum hematoma (LFH) is a rare cause of spinal root or cord compression and usually occur in the lower thoracic and lumbar spine [2,3]. The differential diagnoses include lumbar disk herniation and lumbar spinal canal stenosis caused by neoplasm, infection, epidural hematoma, and facet cyst. The clinical characteristics of LFH are quite different from those of spontaneous spinal epidural hematoma: spontaneous spinal epidural hematoma becomes symp- tomatic in acute stage while in subacute or chronic stage as for most LFH [4]. From 1992 to date, less than 50 cases have been reported in the English literature, mostly in the form of single case reports [2,4-28]. Ligamentum flavum hematoma usually presents with sudden onset of symptoms after minor trauma or exercise. Forty cases identified [26] suggested that LFH is 1.9 times more common in men and occurs most frequently at L4/5. Low back pain is the most frequently observed symptom.

Among all the reported cases, LFH has the following characteristics [22]:

  1. The most affected level is the lumbar spine.
  2. The disease is caused by minor trauma during normal activity.
  3. Most patient are middle-aged or elderly men with a history of hypertension.

0735-6757/(C) 2016

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Fig. 1. Sagittal and axial T1-weighted (A and B), T2-weighted (C and D) and gadolinium-enhanced T2-weighted (E) magnetic resonance images showing an intrathecal cyst-like lesion with adjacent retrospinal soft tissue enhancement at L4-L5 level with severe thecal sac compression and severe encroachment of the bilateral neural foramina.

A

B

C

D

Fig. 2. Intraoperative findings showed ligamentum flavum (A, arrow). After dissecting ligamentum flavum, dark liquefied material (B, arrow) was found, and a capsule lesion was iden- tified (C, arrow). Decompression and exposure of dura (D, arrow) was performed.

H.-P. Liu et al. / American Journal of Emergency Medicine 34 (2016) 2058.e32058.e6 2058.e5

Fig. 3. Sagittal (A) and axial (B) T2-weighted images of the same area 5 months later showing complete resolution after the patient underwent L4 through L5 laminectomies and removal of intraspinal lesion, which was proven to be a old hemorrhage in fibrous tissue.

  1. MRI study serves the most important diagnostic tool because it reveals the connection between the ligamentum flavum and a mass [24].
  2. All cases were cured by surgical treatment.

Magnetic resonance imaging is the most useful tool to diagnose LFH. Nevertheless, previous reports showed variable signal intensities both on T1-weighted and on T2-weighted images. Administration of contrast medium also yielded divergent findings. These differences in MRI clearly depend on the age of the hematoma, and they reflect the chang- ing deoxyhemoglobin or methemoglobin content in the hematoma [10]. Methemoglobin is an altered state of hemoglobin in which the ferrous (Fe2+) iron of heme is oxidized to ferric (Fe3+) state. The ferric hermes of methemoglobin are unable to bind oxygen. The acute clot is hypointense on T2-weighted images due to the presence of deoxyhemoglobin. Over subsequent weeks, deoxyhemoglobin degrades to methemoglobin, which appears bright on both T1-weighted and T2– weighted images. At several months, only hemosiderin remains, and the clot again becomes hypointense on the T1-weighted images. Here a scheme is provided for differentiating LFH (see Table 1).

Ligamentum flavum is poorly vascularized, and only few small vessels pass through it. The pathogenesis of LFH, compatible with most cases of middle-aged or elderly, is thought to be degenerative change of ligamentum flavum which result in rupture of the its small, thin-walled and irregularly dispersed blood vessels in a minor forces causing trivial trauma or elevated intra-abdominal pressure such as spinal flexion, extension, rotation, or shearing movements [6,8,13,18,29].

In the present case, he has general malaise, lumbago, and muscle weakness of lower limbs for 1 month with an episode of acute paralysis after admission. In MRI of the lumbar spine, the hematoma over L4-5 in T1-weighted image, T2-weighted image, and gadolinium all showed a hyperintensity which indicated early stage of chronic LFH. In the middle

Table 1

The differential diagnosis according to MRI of the epidural space-occupying lesion

T1W1 T2W1 Gd

Metastatic tumor Hypo Hyper + ~ –

Schwannoma Hypo-iso Hyper + +

Lipoma Hyper Hyper –

Lymphoma Hypo-iso Hyper or hypo +

Epidural abscess Hypo-iso Hyper + (rim enhancement) Acute LFH Iso-hyper Hyper + ~ –

Subacute LFH Hyper Iso-slightly hypo – Chronic LFH (early phase) Hyper Hyper – Chronic LFH (late phase) Iso Very hyper –

Abbreviations: T1WI, T1-weighted image; T2WI, T2-weighted image; Gd, gadolinium. Revised from Matsui et al. Chronic spontaneous lumbar epidural hematoma simulating extradural spinal tumor: a case report. Nagoya J Med Sci 2014;76:195-201.) [29]

or lower lumbar spine, the main content in the dural sac is the cauda equina. At the levels of middle or lower spine, tolerance to compressing mass is much higher because the main content in the dural sac is the cauda equina, whereas at other levels, the spinal cord is located in the theca. Therefore, gradual formation of hematoma or granuloma in the lumbar spine takes weeks to months to become symptomatic [2]. The main treatment for LFH is surgical resection of the hematoma and the adjacent ligamentum flavum to achieve complete decompression of the dural sac. Nearly all patients had fair prognosis after surgical intervention.

Ligamentum flavum hematoma is a rare entity, but should be taken into consideration as a delayed complication of minor back injury or acute low back pain, especially in middle-aged male patients.

Hui-Pu Liu, MD Chun-Lin Chen, MD? Nan-Fu Chen, MD Chen-Yi Liao, MD Chien-Yu Ou, MD

Department of Neurosurgery, Kaohsiung Armed Forces General Hospital

Kaohsiung, Taiwan

?Corresponding author at: No. 2, Zhongzheng 1st Rd Lingya District, Kaohsiung City 802, Taiwan, ROC. Tel.: +886

774 906 33; fax: +886 774 052 31

E-mail addresses: [email protected],

[email protected]

http://dx.doi.org/10.1016/j.ajem.2016.03.042

References

  1. Kim HS, et al. Ligamentum flavum hematoma in the adjacent segment after a long level fusion. J Korean Neurosurg Soc 2011;49(1):58-60.
  2. Yamaguchi S, et al. Ligamentum flavum hematoma in the lumbar spine. Neurol Med

Chir (Tokyo) 2005;45(5):272-6.

  1. Miyakoshi N, et al. Ligamentum flavum hematoma in the cervical spine–case report. Neurol Med Chir (Tokyo) 2006;46(11):556-8.
  2. Sweasey TA, et al. Ligamentum flavum hematoma. Report of two cases. J Neurosurg 1992;76(3):534-7.
  3. Cruz-Conde R, Berjano P, Buitron Z. Ligamentum flavum hematoma presenting as progres- sive root compression in the lumbar spine. Spine (Phila Pa 1976) 1995;20(13):1506-9.
  4. Mahallati H, et al. MR imaging of a hemorrhagic and granulomatous cyst of the ligamentum

flavum with pathologic correlation. AJNR Am J Neuroradiol 1999;20(6):1166-8.

  1. Minamide A, et al. Ligamentum flavum hematoma in the lumbar spine. J Orthop Sci 1999;4(5):376-9.
  2. Hirakawa K, et al. A post-traumatic ligamentum flavum progressive hematoma: a case report. Spine (Phila Pa 1976) 2000;25(9):1182-4.
  3. Maezawa Y, et al. Ligamentum flavum hematoma in the thoracic spine. Clin Imaging

2001;25(4):265-7.

2058.e6 H.-P. Liu et al. / American Journal of Emergency Medicine 34 (2016) 2058.e32058.e6

  1. Chi TW, Li KT, Chieng PU. Post-traumatic ligamentum flavum hematoma: a case re- port. Kaohsiung J Med Sci 2004;20(1):41-4.
  2. Chen HC, et al. Symptomatic hematoma of cervical ligamentum flavum: case report. Spine (Phila Pa 1976) 2005;30(16):E489-91.
  3. Miyakoshi N, et al. Ligamentum flavum hematoma in the rigid thoracic spinal seg- ments: case report. J Neurosurg Spine 2005;2(4):495-7.
  4. Mizuno J, et al. Ligamentum flavum hematoma in the lumbar spine. Neurol Med Chir

(Tokyo) 2005;45(4):212-5.

  1. Albanese A, et al. Spontaneous haematoma of ligamentum flavum. Case report and literature review. J Neurosurg Sci 2006;50(2):59-61.
  2. Keynan O, et al. Spontaneous ligamentum flavum hematoma in the lumbar spine. Skeletal Radiol 2006;35(9):687-9.
  3. Shimada Y, et al. Chronic subdural hematoma coexisting with ligamentum flavum hematoma in the lumbar spine: a case report. Tohoku J Exp Med 2006;210(1):83-9.
  4. Spuck S, et al. Case reports: a rare cause of radicular complaints: ligamentum flavum hematoma. Clin Orthop Relat Res 2006;443:337-41.
  5. Gazzeri R, et al. Acute hemorrhagic cyst of the ligamentum flavum. J Spinal Disord Tech 2007;20(7):536-8.
  6. Kotil K, Bilge T. A ligamentum flavum hematoma presenting as an L5 radiculopathy. J Clin Neurosci 2007;14(10):994-7.
  7. Kono H, et al. Foot drop of sudden onset caused by acute hematoma in the lumbar ligamentum flavum: a case report and review of the literature. Spine (Phila Pa 1976) 2008;33(16):E573-5.
  8. Lee HW, et al. Spontaneous ligamentum flavum hematoma in the rigid thoracic spine: a case report and review of the literature. J Korean Neurosurg Soc 2008; 44(1):47-51.
  9. Miyakoshi N, et al. Two-level ligamentum flavum hematoma in the lumbar spine. Case report. Neurol Med Chir (Tokyo) 2008;48(4):179-82.
  10. Sudo H, et al. Spinal cord compression by ligamentum flavum hematoma in the thoracic spine. Spine (Phila Pa 1976) 2009;34(25):E942-4.
  11. Takahashi H, et al. Ligamentum flavum haematoma: a report of two cases. J Orthop Surg (Hong Kong) 2009;17(2):212-5.
  12. Tamura T, et al. Hematoma in the cervical ligamentum flavum. Report of a case and review of the literature. Skeletal Radiol 2010;39(3):289-93.
  13. Ohba T, et al. Lumbar ligamentum flavum hematoma treated with endoscopy. Or- thopedics 2011;34(7):e324-7.
  14. Takahashi M, et al. Ligamentum flavum hematoma in the lumbar spine. J Orthop Sci 2012;17(3):308-12.
  15. Wild F, et al. Ligamentum flavum hematomas of the cervical and thoracic spine. Clin Neurol Neurosurg 2014;116:24-7.
  16. Matsui H, Imagama S, Ito Z, Ando K, Hirano K, Tauchi R, et al. Chronic spontaneous lumbar epidural hematoma simulating extradural spinal tumor: a case report. Na- goya J Med Sci 2014;76(1-2):195-201.
  17. Chen CL, Lu CH, Chen NF. Spontaneous spinal epidural hematoma presenting with quadriplegia after sit-ups exercise. Am J Emerg Med 2009;27(9):1170 [e3-7].

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