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International Journal of Surgery Case Reports 114 (2024) 109040

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International Journal of Surgery Case Reports


journal homepage: www.elsevier.com/locate/ijscr

Case report

Spinal epidural cavernous hemangiomas in the lumbar spine: A case report


Dingyan Zhao 1, Yukun Ma 1, Xing Yu *, Lianyong Bi, Xinliang Yue
Dongzhimen Hospital Afliated to Beijing University of Chinese Medicine, Beijing 100700, China

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: Spinal epidural cavernous hemangiomas (SECHs) are relatively rare intradural epidural lesions of
Cavernous malformation the spinal canal, and those occurring in the lumbar spine are even rarer.
Spinal epidural cavernous hemangiomas Case presentation: A 60-year-old man presented for low back pain with right leg pain. His pee and feces were both
SECHs
normal but symptoms were very similar to a typical lumbar disc herniation. The MRI findings suggest an epidural
Case report
space of unknown nature in the spinal cord at the L2–3 level and a definite diagnosis of SECHs was made by
postoperative pathological examination.
Clinical discussion: Patients who are suspected of having SECHs should undergo initial classification and differ­
ential diagnosis based on MRI imaging features. It is crucial to identify the responsible segment in correlation
with the presenting symptoms. During surgery, the primary objective should be the complete removal of the
mass, while taking utmost care to protect the nerves. Dynamic stabilization systems, utilizing pedicle rods, can be
considered as one of the treatment options for such patients.
Conclusion: Patients presenting with low back pain and neurological symptoms should undergo MRI, and diag­
nosed with SECHs should undergo early surgical intervention. For patients with an intradural mass in the spinal
canal, complete resection should be performed while prioritizing nerve protection.

1. Introduction pressure and percussion (+). Muscle strength of each group of the lower
limbs was grade 4 bilaterally, muscle tone was normal. There were no
Cavernous malformation (CM) is a type of intradural lesion charac­ superficial and deep sensory abnormalities in the hip and pre-femoral
terized by developmental deformities caused by vascular abnormalities area of both lower limbs, the knee and the following areas. The right
in the central nervous system. Among these lesions, spinal epidural leg raising and strengthening test was 60◦ (+) and the right femoral
cavernous hemangiomas (SECHs) are relatively uncommon, accounting nerve pull test was +. The preoperative Japanese Orthopedic Associa­
for only 4 % of all epidural lesions [1]. SECHs can occur anywhere along tion (JOA) lumbar score was 8, Oswestry Disability Index (ODI) score
the spine, with a higher frequency observed in the dorsal thoracic region was 27, and Visual Analogue Scale (VAS) scores were 6 and 7 for lumbar
and a lower incidence in the lumbar region [2]. We outline the treatment and lower limb pain, Preoperative imaging results are shown in Fig. 1a-
provided to a patient admitted to our hospital with SECHs at the L2-L3 h. The preoperative diagnosis was an intradural mass, SECHs?
level of the lumbar spine. The patient underwent lumbar posterior laminectomy, mass removal
The work has been reported in line with the SCARE 2020 criteria [3]. and transpedicular internal fixation 3d after admission. Under general
anesthesia, a posterior median incision was performed to expose the
2. Presentation of a case vertebral plate and the L2-L3 spinous processes. The surface of the mass
was smooth, fish-egg shaped, soft, with clear margins, rich in blood
A 60-year-old man experiencing “low back pain with right leg pain supply and intact covering. After leaving the pathology for evaluation,
for 5 years, aggravated for 1 month” was taken to the hospital. He was the tumor was complete and thorough removal (Fig. 1i). The patient's
diagnosed with “lumbar disc herniation” and his pee and feces were both vital signs were steady both throughout and following the procedure.
normal. Examination: Lumbar spine activity was limited, muscle tension Postoperative pathology confirmed a cavernous hemangioma. The
on both sides of the L2-L4 spinous processes, tenderness and pain to tumor was mainly composed of a large number of thin-walled blood

* Corresponding author.
E-mail address: yuxingbucm@sina.com (X. Yu).
1
Dingyan Zhao and Yukun Ma are co first authors in this article.

https://doi.org/10.1016/j.ijscr.2023.109040
Received 7 October 2023; Received in revised form 3 November 2023; Accepted 9 November 2023
Available online 22 November 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
D. Zhao et al. International Journal of Surgery Case Reports 114 (2024) 109040

vessels, with a single layer of endothelial cells and collagen fibres in the hormonal expressions and haemodynamic factors [10] as potential in­
tube wall, lacking the elastic and muscular layers, and the immunohis­ fluences on its formation. The clinical symptoms of SECHs depend on
tochemical results were Vim (+), EMA (+), CD34 (vasculature +), CD31 their specific genesis site and how they interact with anatomical struc­
(vasculature +), Ki-67 (individually +) and SMA (+). At 12 days post- tures such as the spinal cord and nerve roots. Reviews suggest that up to
operatively, the patient was discharged from hospital with a lumbar 86 % of SECHs have been chiefly situated at the dorsal or dorsolateral
JOA score of 21, an ODI score of 39, and lumbar and lower extremity aspect of the spinal cord, with 9 % ventral and a mere 5 % occurring at
VAS scores of 3 and 2. At the 4-month postoperative follow-up, the the intervertebral foramen [1]. Hence, most patients will still exhibit
lumbar JOA score of 19, an ODI score of 41, lumbar and lower extremity signs of spinal cord and nerve root compression. SECHs usually follow a
VAS scores of 2 and 2. Postoperative imaging results are shown in Fig. 1j- course characterized by gradual onset. SECHs with sudden onset being a
o. Patient was satisfied with the results of his treatment. rarity, potentially resulting from an increased pressure due to intra-
tumoral hemorrhage caused by trauma [4]. In this particular case, the
3. Discussion patient had a history of 5 years, with symptoms exacerbating gradually
and pain concentrated locally and extending to lower extremities – an
Cavernous hemangiomas (CM), first characterized by Globus et al. in observation that aligns with previously documented cases.
1929, are uncommon yet insidiously developing, tumor-like vascular At present, the clinical diagnoses for SECHs are reliant on MRI and
malformations. Though they typically manifest in the supratentorial pathological examinations. MRI permits sagittal and coronal scanning,
region of the brain parenchyma, these formations can indeed occur thereby elucidating the location, size, and relationship of the lesion to
anywhere within the body [4]. Depending on their proximities to the surrounding tissues. SECHs typically exhibit an isosignal on T1-
structures such as the dura mater, these vascular malformations can be weighted images and a slightly lower high signal than that of cerebro­
catagorized into four types [5]: intramedullary, extramedullary intra­ spinal fluid (CSF) on T2-weighted images [1,2,11]. When there's an
dural, epidural, and vertebral. The instances of spinal epidural acute rupture and hemorrhage in SECHs due to trauma or various other
cavernous hemangiomas (SECHs) are quite rare, constituting approxi­ factors, both T1- and T2-weighted images display high signals on the
mately just 4 % of epidural tumors [1]. Whether there exists a significant MRI [11]. Some patients may warrant an enhanced MRI to distinguish it
gender disparity in the prevalence of SECHs remains contentious [1,2]. from other intraspinal masses, because SECHs may reflecting inhomo­
It is agreed that they are most often found in the thoracic vertebrae - geneous enhancement. In our case, where the posterior aspect of the L2-
followed by lumbar, then cervical and sacral vertebrae - and most likely L3 spinal cord displayed a low signal on T1WI and a high signal on T2WI
to be located dorsal or dorsal-lateral to the spinal canal [2,6]. during the preoperative MRI.
SECHs were considered as a vascular malformation initiated by The conclusive diagnosis of spinal epidural cavernous hemangiomas
dysplasia [7]. Investigations of CM imply genetics [8], trauma [9], (SECHs) is reliant on pathological results. The initial differential

Fig. 1. Imaging data of the case


a,b: The lumbar spine was seen to be degenerated, with the presence of overall physiological curvature and rostral osteophyte formation at the anterior margin of the
L1–2 vertebral body c,d:Anterior flexion and posterior extension demonstrate fair mobility of the L2–3 segments. e,f,g: MRI showed obvious compression of the spinal
cord at the level of L2–3, and an oval-shaped mass was seen in the dorsal aspect of the spinal canal with clear borders, smooth edges and intact periosteum. The mass
showed isointense signal on T1WI and high signal on T2WI and compression fat images. h:MRI showed that 75 % of the spinal canal was encroached, and the tumor
was on the right side. i: The tumor was removed intact. j,k:The nail rod system was in good position. l,m,n:MRI showed that the spinal cord space at L2–3 was
significantly enlarged. o:The L2–3 level showed that the morphology of the cauda equina in the spinal cord was restored and dispersed, surrounded by cerebrospinal
fluid, and the intervertebral foraminal space was good bilaterally.

2
D. Zhao et al. International Journal of Surgery Case Reports 114 (2024) 109040

diagnosis can also be made preoperatively on the basis of MRI. It is Ethical approval
crucial to differentiate SECHs from other types of epidural lesions, such
as metastatic tumors, lymphomas, abscesses, among others [12]. Met­ This study was approved by the Medical Ethics Committee of
astatic tumors usually manifest as sizable soft tissue masses, commonly Dongzhimen Hospital, Beijing University of Traditional Chinese Medi­
accompanied by significant vertebral and accessory bone destruction cine (approval number: 2022DZMEC-085-05).
and a history of the principal tumor, and exhibit slightly higher signals
on T2-weighted images in MRI scans. Differentiating lymphoma from Research registration
the epidural type of SECHs can be challenging, especially when they
occur as a mass in the epidural space, indicated by a moderate signal on None.
both T1WI and T2WI. Epidural abscesses may display a moderate signal
on T1WI and a high signal on T2WI; these patients typically have a Guarantor
history of trauma, surgery, or other infections. SECHs also need to be
distinguished from other vascular-rich tumors like spinal tumors, as Dingyan Zhao.
their signal and enhancement patterns are very similar. It's noteworthy
to identify the differences in morphology and growth patterns between Provenance and peer review
these two types of SECHs. Variance in the location is also distinctive of
the different types of SECHs. Epidural SECHs, positioned in the epidural Not commissioned, externally peer-reviewed.
space, tend to exhibit a creeping growth pattern, resembling a staggered
or elongated oval shape. Conversely, a chordoma is mostly broad-based, CRediT authorship contribution statement
slow-growing mass adhering to the dura mater and is primarily situated
in the extramedullary subdural space. Dingyan Zhao: Writing- Original draft preparation and Methodology;
The cornerstone of treatment for SECHs is surgical resection. Early Yukun Ma: Writing- Reviewing and Editing; Xing Yu: Conceptualization,
diagnosis and timely surgical intervention are imperative. For most Supervision; Xinliang Yue, Lianyong Bi: Data curation.
SECHs, complete resection is a possibility, particularly for the epidural
type. Protective measures for the spinal cord and nerve roots are indis­ Declaration of competing interest
pensable during surgery, especially for intramedullary and extra-
medullary intradural CM. Additionally, selecting the right fixation The authors report no conflict of interest.
method must take into account the lesion's location and the involved
segments. In this case, the preoperative X-rays showed minor activity in References
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Sources of funding

No funding was obtained for this study.

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