Clinical Neurology and Neurosurgery 116 (2014) 24–27
Contents lists available at ScienceDirect
Clinical Neurology and Neurosurgery journal homepage: www.elsevier.com/locate/clineuro
Ligamentum flavum hematomas of the cervical and thoracic spine Florian Wild a,∗ , Jochen Tuettenberg b , Armin Grau c , Joachim Weis d , Joachim K. Krauss a a
Department of Neurosurgery, Medical School Hannover, Hannover, Germany Department of Neurosurgery, Clinical Center Idar-Oberstein, Idar-Oberstein, Germany c Department of Neurology, Municipal Hospital Ludwigshafen, Ludwigshafen, Germany d Institute of Neuropathology, RWTH Aachen, Aachen, Germany b
a r t i c l e
i n f o
Article history: Received 5 August 2013 Received in revised form 17 October 2013 Accepted 9 November 2013 Available online 16 November 2013 Keywords: Ligamentum flavum Hematoma Cervical spine Thoracic spine
a b s t r a c t Objective: To report extremely rare cases of ligamentum flavum hematomas in the cervical and thoracic spine. Only six cases of thoracic ligamentum flavum hematomas and three cases of cervical ligamentum flavum hematomas have been reported so far. Methods: Two patients presented with tetraparesis and one patient presented with radicular pain and paresthesias in the T3 dermatome. MRI was performed in two patients, which showed a posterior intraspinal mass, continuous with the ligamentum flavum. The mass was moderately hypointense on T2-weighted images and hyperintense on T1-weighted images with no contrast enhancement. The third patient underwent cervical myelography because of a cardiac pacemaker. The myelography showed an intraspinal posterior mass with compression of the dural sac at C3/C4. Results: All patients underwent a hemilaminectomy to resect the ligamentum flavum hematoma and recovered completely afterwords, and did not experience a recurrence during follow-up of at least 2 years. Conclusion: This case series shows rare cases of ligamentum flavum hematomas in the cervical and thoracic spine. Surgery achieved complete recovery of the preoperative symptoms in all patients within days. © 2013 Elsevier B.V. All rights reserved.
1. Introduction Delayed onset of neurological symptoms in the lower extremities is associated most frequently with degenerative disk disorders [2,6,7,11]. Bleeding in the epidural and subdural spaces or within a spinal tumor are other well-known causes [10]. Progressive myelopathy due to hemorrhage into the ligamentum flavum of the thoracic or cervical spine, however, has received only very little attention [3,16,17,21,22]. Available data on diagnosis and treatment is scarce and long-term prognosis after surgery has been unclear. Here, we report our experiences in a series of 3 patients seen within a period of 10 years. 2. Patients Three patients presented with delayed onset of neurological symptoms due to acute hemorrhage into the ligamentum flavum. One patient was female (age, 56 years), and the other two were
∗ Corresponding author at: Department of Neurosurgery, Medical School Hannover, MHH, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany. Tel.: +49 511 532 6652; fax: +49 511 532 5864. E-mail address: wild.fl[email protected] (F. Wild). 0303-8467/$ – see front matter © 2013 Elsevier B.V. All rights reserved. http://dx.doi.org/10.1016/j.clineuro.2013.11.008
male (age, 54 and 59 years). Two hematomas were localized in the cervical spine at level C3/C4; the hematoma in the third patient was localized in the thoracic spine at level T3/T4. All patients were referred for emergency treatment when symptoms deteriorated. 3. Results Both patients with a cervical ligamentum flavum hematoma presented with tetraparesis; the patient with the thoracic ligamentum flavum hematoma had radicular pain and paresthesias in the T3 dermatome. One patient with cervical ligamentum flavum hematoma was on anticoagulant therapy. MRI was performed in two patients, which showed a posterior intraspinal mass, continuous with the ligamentum flavum. The mass was moderately hypointense on T2-weighted images and hyperintense on T1-weighted images (see Fig. 1a–c) with no contrast enhancement. The third patient underwent cervical myelography because of a cardiac pacemaker. The myelography showed an intraspinal posterior mass with compression of the dural sac at C3/C4. All patients underwent a hemilaminectomy at the levels of the lesion with complete removal of the hematomas within the ligamentum flavum. After opening the flavum at the site of the cyst, the hematoma was evacuated which was partially organized. In
F. Wild et al. / Clinical Neurology and Neurosurgery 116 (2014) 24–27
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Fig. 1. (a) Ligamentum flavum hematoma at C3/C4 in a 54-year-old man with degenerative spinal cord disease. Sagittal T2-weighted MRI shows a hypointense posterior intraspinal mass at C3/C4 continuous with the ligamentum flavum. (b) On T1-weighted MRI the lesion has a mixed density. (c) Axial T1-weighted scans demonstrate the compression of the spinal cord due to the lesion.
all patients there were dense adhesions to the dura which made the surgery more difficult as compared to removal of ligamentum flavum cysts without hematoma. To prevent recurrence of the cyst the flavum was removed at a safely margin of 3 mm. The postoperative course of all patients was uneventful. Preoperative symptoms subsided within days. Histological examination of the operative specimen of the thoracic lesion showed the typical appearance of the ligamentum flavum with densely packed elastic fibers (Fig. 2a). There was focal proliferation of granulation tissue and accumulation of hemosiderin-laden macrophages (Fig. 2b and c) indicative of previous hemorrhage. Widespread dystrophic calcification was found in the elastic tissue (Fig. 2d and e). No evidence of infectious or neoplastic changes was found. Follow-up examinations of at least 2 years postoperatively were unremarkable without any residual symptoms in all instances. There was no recurrence of hematoma or of a ligamentum flavum cyst. 4. Discussion Ligamentum flavum hematomas are rare lesions. From 1992 to date less than 50 cases have been reported in the English literature, mostly in the form of single case reports [1,3–5,9,12–18,20–32]. While the hematoma was located in the lumbar spine in most
instances, only nine cases were described with locations in the thoracic or cervical spine. Remarkably, the first case of a thoracic ligamentum flavum hematoma was reported only in 2001, and the first case of a cervical ligamentum flavum hematoma in 2005. All cervical ligamentum flavum hematomas reported so far were localized at level C3/C4, and all thoracic lesions were localized at level T7/T8 or below (see Table 1). Most of the patients reported so far had a history of minor trauma with a progressive clinical course over several weeks which had initially been attributed to disk herniation or spinal canal stenosis. All patients reported so far were at least 30 years of age (mean 62.7 years) which clearly indicates that ligamentum flavum hematoma is a disease of advanced age. Even though cervical ligamentum flavum hematomas are rare, they pose a special problem since they usually present with much more severe deficits than lumbar lesions as demonstrated by our cases. It is puzzling to note that most ligamentum flavum hematomas reported earlier occurred in the lumbar spine. We suggest that the reason for this is that the lumbar spine is stressed more often by load-bearing forces than the thoracic and the cervical spine. As opposed to the thoracic spine, the lumbar spine is more mobile which makes it more prone to shearing forces after minor trauma. Considering these factors, it still remains unclear why the cervical spine is affected only exceptionally. The major reason for this might be a different composition of the connective tissue on the cervical spine compared to the thoracic and lumbar spine.
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F. Wild et al. / Clinical Neurology and Neurosurgery 116 (2014) 24–27
Table 1 Reported cases of ligamentum flavum hematoma in the cervical and thoracic spine. Author (year)
Age (sex)
Level
Surgery
Outcome (follow-up time)
Maezawa et al. (2001) Chen et al. (2005) Miyakoshi et al. (2005) Miyakoshi et al. (2006) Lee et al. (2008) Sudo et al. (2009) Tamura et al. (2010) Matsumoto et al. (2011) Matsumoto et al. (2011) Wild et al. (2013) Wild et al. (2013) Present series (2013)
66 (m) 72 (m) 66 (f) 67 (m) 72 (m) 61 (m) 69 (m) 54 (m) 57 (m) 59 (m) 54 (m) 56 (f)
T11/T12 C3/C4 T9/T10 C3/C4 T7/T8 T10/T11 C3/C4 T11/T12 T11/T12 C3/C4 C3/C4 T3/T4
Laminectomy of T11 and T12 Laminectomy of C3 and C4 Laminectomy of T9 and T10 Laminectomy of C3 and C4 Laminectomy T7 Laminectomy T10 Laminectomy of C3 and C4 Laminectomy T11 Partial laminectomy T11 and T12 Hemilaminectomy C3 Hemilaminectomy C3 Interlaminotomy T3/T4
Numbness of both legs (2 years) Symptom-free (1 year) Walked with cane (4 weeks) Numbness of both hands (1 year) Symptom-free (4 weeks) Symptom-free (after surgery) Walked without aid (4 weeks) Symptom-free (7 years) Symptom-free (1 year) Symptom-free (2 years) Symptom-free (2 years) Symptom-free (5 years)
Fig. 2. (A) Largely intact elastic fibers of the ligamentum flavum paraffin section, Elastica van Gieson; scale bar = 140 m. (B) Turnbull blue-positive hemosiderin deposits and granulation tissue within the operative specimen. Paraffin section; scale bar = 125 m. (C) Higher magnification of hemosiderin-laden macrophages (blue). Paraffin section; scale bar = 60 m. (D) Dystrophic calcification of ligamentous structures. Paraffin section, HE; scale bar = 125 m. (E) Elastica van Gieson stain of a parallel section of the same region as in (D). Fragments of degenerated elastic fibers are stained dark brown (arrows). Paraffin section; scale bar = 60 m. (For interpretation of the references to color in this legend, the reader is referred to the web version of the article.)
The normal ligamentum flavum consists of elastic fibers and collagen. It is poorly vascularized, and only few small vessels pass through it [12]. Furusawa et al. reported the presence of proliferating small blood vessels in the degenerative ligamentum flavum with aging [8]. Chen et al. suggested that degenerative or hypertrophic changes of the ligamentum flavum could result in a more fibrotic structure rich in collagen fibers, which is weaker in strength than the elastic fibers [3]. As shown in the histopathological examination in one of our patients focal calcification secondary to degeneration might be another risk factor.
Minor forces such as spinal flexion, rotation, or sharing movements could cause the rupture of the very small, thin-walled and irregularly dispersed blood vessels within the degenerated ligament [9,12,18,24]. It was also suggested that increased intraabdominal pressure, transmitted via the epidural venous plexus and intraligamentous vessels may cause rupture of small vessels within the degenerative ligamentum flavum [20]. Finally, hemolytic and fibrinolytic changes within the small bleeding cavity might result in an increasing volume [5,20]. MR imaging is the most useful tool to diagnose ligamentum flavum hematomas. Nevertheless, previous reports showed variable signal intensities both on T1-weighted and on T2-weighted images [1,3–5,9,12–18,20–32]. Administration of contrast medium also yielded divergent findings [4,12,14,16–18,20,24,25,29,31]. These differences in MR imaging clearly depend on the age of the hematoma, and they reflect the changing deoxyhemoglobin or methemoglobin content in the hematoma [4]. Thus far, all reported cases were treated with surgical resection of the hematoma and the adjacent ligamentum flavum to achieve complete decompression of the dural sac. Procedures included laminectomy [1,3,5,16–23,27–29,31,33], hemilaminectomy [9,24,28–30,32], facetectomy [12,13] and interlaminotomy [26,30]. All neurosurgical operations produced an excellent clinical outcome in almost all patients [1,3–5,9,12–33]. Thus far, there is no consensus which approval is suited best for removal of these unusual lesions. We choose to perform a hemilaminectomy both to provide adequate decompression but also insure removing all cyst remnants at the adjacent ligamentum flavum to prevent recurrence. While hemilaminectomy has a low risk to compromise stability in the lumbar spine, this risk is only minimal and may be neglected in the cervical and thoracic spine. Persistent numbness of the hands or the legs was noted in single instances of cervical or thoracic ligamentum flavum hematomas, respectively. 5. Conclusion Ligamentum flavum hematoma is a rare spinal mass lesion which occurs mostly in the lumbar spine. It has to be considered in the differential diagnosis in patients presenting first with mild symptoms, which slowly progress to paresis or other symptoms of spinal cord compression. Surgical resection of ligamentum flavum hematoma usually results in excellent recovery. As demonstrated in our report, even when the cervical spine is affected and patients present with tetraparesis early decompression can lead to a positive outcome. References [1] Albanese A, Braconi A, Anile C, Mannino S, Sabatino G, Mangiola A. Spontaneous haematoma of ligamentum flavum: case report and literature review. J Neurosurg Sci 2006;50:59–61.
F. Wild et al. / Clinical Neurology and Neurosurgery 116 (2014) 24–27 [2] Brant-Zawadzki MN, Dennis SC, Gade GF, Weinstein MP. Low back pain. Radiology 2000;217:321–30. [3] Chen HC, Hsu PW, Lin CY, Tzaan WC. Symptomatic hematoma of cervical ligamentum flavum: case report. Spine 2005;30:489–91. [4] Chi TW-C, Li K-T, Chieng P-U. Post-traumatic ligamentum flavum hematoma: a case report. Kaohsiung J Med Sci 2004;20:41–4. [5] Cruz-Conde R, Berjano P, Buitron Z. Ligamentum flavum hematoma presenting as progressive root compression in the lumbar spine. Spine 1995;20:1506–9. [6] Epstein JA, Epstein BS, Lavine L. Nerve root compression associated with narrowing of the lumbar spinal canal. J Neurol Neurosurg Psychiatry 1962;25:165–76. [7] Epstein JA, Lavine LS, Epstein BS, Carras R. Herniated discs an related disorders of the lumbar spine: surgical treatment in the geriatric patient. J Am Med Assoc 1967;202(3):187–90. [8] Furusawa N, Baba H, Maezawa Y, Uchida K, Wada M, Imura S, et al. Calcium crystal deposition in the ligamentum flavum. Clin Exp Rheumatol 1997;15:641–7. [9] Gazzeri R, Canova A, Fiore C, Galarza M, Neroni M, Giordano M. Acute hemorrhagic cyst of the ligamentum flavum. J Spinal Disord Tech 2007;20:536–8. [10] Groen RJM, van Alphen HAM. Operative treatment of spontaneous spinal epidural hematomas: a study of the factors determining postoperative outcome. Neurosurgery 1996;39(3):494–509. [11] Gundry CR, Heithoff KB. Epidural hematoma of the lumbar spine: 18 surgically confirmed cases. Radiology 1993;187:427–31. [12] Hirakawa K, Hanakita J, Suwa H, Matsuoka N, Oda M, Muro H, et al. A posttraumatic ligamentum flavum progressive hematoma: a case report. Spine 2000;25:1182–4. [13] Keynan O, Smorgick Y, Schwartz AJ, Ashkenazi E, Floman Y. Spontaneous ligamentum flavum hematoma in the lumbar spine. Skeletal Radiol 2006;35:687–9. [14] Kono H, Nakamura H, Seki M, Motoda T. Foot drop of sudden onset caused by acute hematoma in the lumbar ligamentum flavum. Spine 2008;23:573–5. [15] Kotil K, Bilge T. A ligamentum flavum hematoma presenting as an L5 radiculopathy. J Clin Neurosci 2007;14:994–7. [16] Lee H-W, Song J-H, Chang I-B, Choi H-C. Spontaneous ligamentum flavum hematoma in the rigid thoracic spine: a case report and review of the literature. J Korean Neurosurg Soc 2008;44:47–51. [17] Maezawa Y, Baba H, Uchida K, Kokubo Y, Kubota C, Noriki S. Ligamentum flavum hematoma in the thoracic spine. J Clin Imaging 2001;25:265–7. [18] Mahallati H, Wallace CJ, Hunter KM, Bilbao JM, Clark AW. MR imaging of a hemorrhagic and granulomatous cyst of the ligamentum flavum with pathologic correlation. Am J Neuroradiol 1999;20:1166–8.
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[19] Matsumoto Y, Fujiwara T, Imamura R, Okada Y, Harimaya K, Doi T, et al. Hematoma of the ligamentum flavum in the thoracic spine: report of two cases and possible role of the transforming growth factor beta-vascular endothelial growth factor signalling axis in its pathogenesis. J Orthop Sci 2011 [Published online 31 August]. [20] Minamide A, Yoshida M, Tamaki T, Natsumi K. Ligamentum flavum hematoma in the lumbar spine. J Orthop Sci 1999;4:376–9. [21] Miyakoshi N, Shimada Y, Okada K, Hongo M, Kasukawa Y, Itoi E. Ligamentum flavum hematoma in the rigid thoracic spinal segments. J Neurosurg Spine 2005;2:495–7. [22] Miyakoshi N, Shimada Y, Kasukawa Y, Ando S. Ligamentum flavum hematoma in the cervical spine – case report. Neurol Med Chir (Tokyo) 2006;46: 556–8. [23] Miyakoshi N, Kasukawa Y, Ando S, Shimada Y. Two-level ligamentum flavum hematoma in the lumbar spine: case report. Neurol Med Chir (Tokyo) 2008;48:179–82. [24] Mizuno J, Nakagawa H, Inoue T, Hashizume Y. Ligamentum flavum hematoma in the lumbar spine: case report. Neurol Med Chir (Tokyo) 2005;45:212–5. [25] Ohba T, Ebata S, Ando T, Ichikawa J, Clinton D, Haro H. Lumbar ligamentum flavum hematoma treated with endoscopy. Orthopedics 2011;34:e324–7. [26] Spuck S, Stellmacher F, Wiesmann M, Kranz R. A rare cause of radicular complaints: ligamentum flavum hematoma. Clin Orthop Relat Res 2006;443:337–41. [27] Sudo H, Abumi K, Ito M, Kotani Y, Takahata M, Hojo Y, et al. Spinal cord compression by ligamentum flavum hematoma in the thoracic spine. Spine 2009;34:942–4. [28] Sweasey TA, Coester HC, Rawal H, Blaivas M, McGillicuddy JE. Ligamentum flavum hematoma: report of two cases. J Neurosurg 1992;76:534–7. [29] Takahashi H, Wada A, Yokoyama Y, Fukushi S, Sakurai T, Shibuya K. Ligamentum flavum haematoma: a report of two cases. J Orthop Surg (Hong Kong) 2009;17:212–5. [30] Takahashi M, Satomi K, Hasegawa M, Taki N, Ichimura S. Ligamentum flavum hematoma in the lumbar spine. J Orthop Sci 2011 [Published online 21 May]. [31] Tamura T, Sakai T, Sairyo K, Takao S, Kagawa S, Katoh S, et al. Hematoma in the cervical ligamentum flavum: report of a case and review of the literature. Skeletal Radiol 2010;39:289–93. [32] Yamaguchi S, Hida K, Akino M, Seki T, Yano S, Iwasaki Y. Ligamentum flavum hematoma in the lumbar spine: case report. Neurol Med Chir (Tokyo) 2005;45:272–6. [33] Yüceer N, Bas¸kaya MK, Smith P, Willis BK. Hematoma of the ligamentum flavum in the lumbar spine: case report. Surg Neurol 2000;53:598–600.
Contents lists available at ScienceDirect
Clinical Neurology and Neurosurgery journal homepage: www.elsevier.com/locate/clineuro
Ligamentum flavum hematomas of the cervical and thoracic spine Florian Wild a,∗ , Jochen Tuettenberg b , Armin Grau c , Joachim Weis d , Joachim K. Krauss a a
Department of Neurosurgery, Medical School Hannover, Hannover, Germany Department of Neurosurgery, Clinical Center Idar-Oberstein, Idar-Oberstein, Germany c Department of Neurology, Municipal Hospital Ludwigshafen, Ludwigshafen, Germany d Institute of Neuropathology, RWTH Aachen, Aachen, Germany b
a r t i c l e
i n f o
Article history: Received 5 August 2013 Received in revised form 17 October 2013 Accepted 9 November 2013 Available online 16 November 2013 Keywords: Ligamentum flavum Hematoma Cervical spine Thoracic spine
a b s t r a c t Objective: To report extremely rare cases of ligamentum flavum hematomas in the cervical and thoracic spine. Only six cases of thoracic ligamentum flavum hematomas and three cases of cervical ligamentum flavum hematomas have been reported so far. Methods: Two patients presented with tetraparesis and one patient presented with radicular pain and paresthesias in the T3 dermatome. MRI was performed in two patients, which showed a posterior intraspinal mass, continuous with the ligamentum flavum. The mass was moderately hypointense on T2-weighted images and hyperintense on T1-weighted images with no contrast enhancement. The third patient underwent cervical myelography because of a cardiac pacemaker. The myelography showed an intraspinal posterior mass with compression of the dural sac at C3/C4. Results: All patients underwent a hemilaminectomy to resect the ligamentum flavum hematoma and recovered completely afterwords, and did not experience a recurrence during follow-up of at least 2 years. Conclusion: This case series shows rare cases of ligamentum flavum hematomas in the cervical and thoracic spine. Surgery achieved complete recovery of the preoperative symptoms in all patients within days. © 2013 Elsevier B.V. All rights reserved.
1. Introduction Delayed onset of neurological symptoms in the lower extremities is associated most frequently with degenerative disk disorders [2,6,7,11]. Bleeding in the epidural and subdural spaces or within a spinal tumor are other well-known causes [10]. Progressive myelopathy due to hemorrhage into the ligamentum flavum of the thoracic or cervical spine, however, has received only very little attention [3,16,17,21,22]. Available data on diagnosis and treatment is scarce and long-term prognosis after surgery has been unclear. Here, we report our experiences in a series of 3 patients seen within a period of 10 years. 2. Patients Three patients presented with delayed onset of neurological symptoms due to acute hemorrhage into the ligamentum flavum. One patient was female (age, 56 years), and the other two were
∗ Corresponding author at: Department of Neurosurgery, Medical School Hannover, MHH, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany. Tel.: +49 511 532 6652; fax: +49 511 532 5864. E-mail address: wild.fl[email protected] (F. Wild). 0303-8467/$ – see front matter © 2013 Elsevier B.V. All rights reserved. http://dx.doi.org/10.1016/j.clineuro.2013.11.008
male (age, 54 and 59 years). Two hematomas were localized in the cervical spine at level C3/C4; the hematoma in the third patient was localized in the thoracic spine at level T3/T4. All patients were referred for emergency treatment when symptoms deteriorated. 3. Results Both patients with a cervical ligamentum flavum hematoma presented with tetraparesis; the patient with the thoracic ligamentum flavum hematoma had radicular pain and paresthesias in the T3 dermatome. One patient with cervical ligamentum flavum hematoma was on anticoagulant therapy. MRI was performed in two patients, which showed a posterior intraspinal mass, continuous with the ligamentum flavum. The mass was moderately hypointense on T2-weighted images and hyperintense on T1-weighted images (see Fig. 1a–c) with no contrast enhancement. The third patient underwent cervical myelography because of a cardiac pacemaker. The myelography showed an intraspinal posterior mass with compression of the dural sac at C3/C4. All patients underwent a hemilaminectomy at the levels of the lesion with complete removal of the hematomas within the ligamentum flavum. After opening the flavum at the site of the cyst, the hematoma was evacuated which was partially organized. In
F. Wild et al. / Clinical Neurology and Neurosurgery 116 (2014) 24–27
25
Fig. 1. (a) Ligamentum flavum hematoma at C3/C4 in a 54-year-old man with degenerative spinal cord disease. Sagittal T2-weighted MRI shows a hypointense posterior intraspinal mass at C3/C4 continuous with the ligamentum flavum. (b) On T1-weighted MRI the lesion has a mixed density. (c) Axial T1-weighted scans demonstrate the compression of the spinal cord due to the lesion.
all patients there were dense adhesions to the dura which made the surgery more difficult as compared to removal of ligamentum flavum cysts without hematoma. To prevent recurrence of the cyst the flavum was removed at a safely margin of 3 mm. The postoperative course of all patients was uneventful. Preoperative symptoms subsided within days. Histological examination of the operative specimen of the thoracic lesion showed the typical appearance of the ligamentum flavum with densely packed elastic fibers (Fig. 2a). There was focal proliferation of granulation tissue and accumulation of hemosiderin-laden macrophages (Fig. 2b and c) indicative of previous hemorrhage. Widespread dystrophic calcification was found in the elastic tissue (Fig. 2d and e). No evidence of infectious or neoplastic changes was found. Follow-up examinations of at least 2 years postoperatively were unremarkable without any residual symptoms in all instances. There was no recurrence of hematoma or of a ligamentum flavum cyst. 4. Discussion Ligamentum flavum hematomas are rare lesions. From 1992 to date less than 50 cases have been reported in the English literature, mostly in the form of single case reports [1,3–5,9,12–18,20–32]. While the hematoma was located in the lumbar spine in most
instances, only nine cases were described with locations in the thoracic or cervical spine. Remarkably, the first case of a thoracic ligamentum flavum hematoma was reported only in 2001, and the first case of a cervical ligamentum flavum hematoma in 2005. All cervical ligamentum flavum hematomas reported so far were localized at level C3/C4, and all thoracic lesions were localized at level T7/T8 or below (see Table 1). Most of the patients reported so far had a history of minor trauma with a progressive clinical course over several weeks which had initially been attributed to disk herniation or spinal canal stenosis. All patients reported so far were at least 30 years of age (mean 62.7 years) which clearly indicates that ligamentum flavum hematoma is a disease of advanced age. Even though cervical ligamentum flavum hematomas are rare, they pose a special problem since they usually present with much more severe deficits than lumbar lesions as demonstrated by our cases. It is puzzling to note that most ligamentum flavum hematomas reported earlier occurred in the lumbar spine. We suggest that the reason for this is that the lumbar spine is stressed more often by load-bearing forces than the thoracic and the cervical spine. As opposed to the thoracic spine, the lumbar spine is more mobile which makes it more prone to shearing forces after minor trauma. Considering these factors, it still remains unclear why the cervical spine is affected only exceptionally. The major reason for this might be a different composition of the connective tissue on the cervical spine compared to the thoracic and lumbar spine.
26
F. Wild et al. / Clinical Neurology and Neurosurgery 116 (2014) 24–27
Table 1 Reported cases of ligamentum flavum hematoma in the cervical and thoracic spine. Author (year)
Age (sex)
Level
Surgery
Outcome (follow-up time)
Maezawa et al. (2001) Chen et al. (2005) Miyakoshi et al. (2005) Miyakoshi et al. (2006) Lee et al. (2008) Sudo et al. (2009) Tamura et al. (2010) Matsumoto et al. (2011) Matsumoto et al. (2011) Wild et al. (2013) Wild et al. (2013) Present series (2013)
66 (m) 72 (m) 66 (f) 67 (m) 72 (m) 61 (m) 69 (m) 54 (m) 57 (m) 59 (m) 54 (m) 56 (f)
T11/T12 C3/C4 T9/T10 C3/C4 T7/T8 T10/T11 C3/C4 T11/T12 T11/T12 C3/C4 C3/C4 T3/T4
Laminectomy of T11 and T12 Laminectomy of C3 and C4 Laminectomy of T9 and T10 Laminectomy of C3 and C4 Laminectomy T7 Laminectomy T10 Laminectomy of C3 and C4 Laminectomy T11 Partial laminectomy T11 and T12 Hemilaminectomy C3 Hemilaminectomy C3 Interlaminotomy T3/T4
Numbness of both legs (2 years) Symptom-free (1 year) Walked with cane (4 weeks) Numbness of both hands (1 year) Symptom-free (4 weeks) Symptom-free (after surgery) Walked without aid (4 weeks) Symptom-free (7 years) Symptom-free (1 year) Symptom-free (2 years) Symptom-free (2 years) Symptom-free (5 years)
Fig. 2. (A) Largely intact elastic fibers of the ligamentum flavum paraffin section, Elastica van Gieson; scale bar = 140 m. (B) Turnbull blue-positive hemosiderin deposits and granulation tissue within the operative specimen. Paraffin section; scale bar = 125 m. (C) Higher magnification of hemosiderin-laden macrophages (blue). Paraffin section; scale bar = 60 m. (D) Dystrophic calcification of ligamentous structures. Paraffin section, HE; scale bar = 125 m. (E) Elastica van Gieson stain of a parallel section of the same region as in (D). Fragments of degenerated elastic fibers are stained dark brown (arrows). Paraffin section; scale bar = 60 m. (For interpretation of the references to color in this legend, the reader is referred to the web version of the article.)
The normal ligamentum flavum consists of elastic fibers and collagen. It is poorly vascularized, and only few small vessels pass through it [12]. Furusawa et al. reported the presence of proliferating small blood vessels in the degenerative ligamentum flavum with aging [8]. Chen et al. suggested that degenerative or hypertrophic changes of the ligamentum flavum could result in a more fibrotic structure rich in collagen fibers, which is weaker in strength than the elastic fibers [3]. As shown in the histopathological examination in one of our patients focal calcification secondary to degeneration might be another risk factor.
Minor forces such as spinal flexion, rotation, or sharing movements could cause the rupture of the very small, thin-walled and irregularly dispersed blood vessels within the degenerated ligament [9,12,18,24]. It was also suggested that increased intraabdominal pressure, transmitted via the epidural venous plexus and intraligamentous vessels may cause rupture of small vessels within the degenerative ligamentum flavum [20]. Finally, hemolytic and fibrinolytic changes within the small bleeding cavity might result in an increasing volume [5,20]. MR imaging is the most useful tool to diagnose ligamentum flavum hematomas. Nevertheless, previous reports showed variable signal intensities both on T1-weighted and on T2-weighted images [1,3–5,9,12–18,20–32]. Administration of contrast medium also yielded divergent findings [4,12,14,16–18,20,24,25,29,31]. These differences in MR imaging clearly depend on the age of the hematoma, and they reflect the changing deoxyhemoglobin or methemoglobin content in the hematoma [4]. Thus far, all reported cases were treated with surgical resection of the hematoma and the adjacent ligamentum flavum to achieve complete decompression of the dural sac. Procedures included laminectomy [1,3,5,16–23,27–29,31,33], hemilaminectomy [9,24,28–30,32], facetectomy [12,13] and interlaminotomy [26,30]. All neurosurgical operations produced an excellent clinical outcome in almost all patients [1,3–5,9,12–33]. Thus far, there is no consensus which approval is suited best for removal of these unusual lesions. We choose to perform a hemilaminectomy both to provide adequate decompression but also insure removing all cyst remnants at the adjacent ligamentum flavum to prevent recurrence. While hemilaminectomy has a low risk to compromise stability in the lumbar spine, this risk is only minimal and may be neglected in the cervical and thoracic spine. Persistent numbness of the hands or the legs was noted in single instances of cervical or thoracic ligamentum flavum hematomas, respectively. 5. Conclusion Ligamentum flavum hematoma is a rare spinal mass lesion which occurs mostly in the lumbar spine. It has to be considered in the differential diagnosis in patients presenting first with mild symptoms, which slowly progress to paresis or other symptoms of spinal cord compression. Surgical resection of ligamentum flavum hematoma usually results in excellent recovery. As demonstrated in our report, even when the cervical spine is affected and patients present with tetraparesis early decompression can lead to a positive outcome. References [1] Albanese A, Braconi A, Anile C, Mannino S, Sabatino G, Mangiola A. Spontaneous haematoma of ligamentum flavum: case report and literature review. J Neurosurg Sci 2006;50:59–61.
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