SUBMITTED 25 OCT 21 1 REVISION REQ. 26 DEC 21; REVISION RECD. 24 JAN 22 2 ACCEPTED 22 FEB 22 3 ONLINE-FIRST: MARCH 2022 4 DOI: https://doi.org/10.18295/squmj.3.2022.024 5 6 Large Intraosseous Haemangioma of the Sacral Vertebra 7 The radiological imaging findings 8 Nerbadyswari Deep,1 *Sudipta Mohakud,1 Mantu Jain,2 Suprava Naik,1 9 Manas Baisakh3 10 11 Departments of 1Radiodiagnosis and 2Orthopedics, All India Institute of Medical Sciences, 12 Bhubaneswar, India; 3Department of Pathology, Prolife Diagnostic, Bhubaneswar, India 13 *Corresponding Author’s e-mail: drsudipta.m@gmail.com 14 15 A 28-year-old male technologist presented to the Orthopedics department of the All India 16 Institute of Medical Sciences, Bhubaneswar, in 2020 with a complaint of dull aching low 17 back pain on prolonged sitting for six months. There was local tenderness in the sacral region 18 on deep palpation without local swelling or pain radiation to the limbs. The straight leg 19 raising test was negative. He was intact neurologically (ASIA E), and the pain score was low 20 (VAS-2/10). An x-ray showed some suspicious lytic lesion in the sacral vertebra. He 21 underwent computed tomography (CT) scan and a contrast-enhanced magnetic resonance 22 imaging to characterize the lesion further. The CT highlighted a large expansile lucent lesion 23 associated with a soft tissue component involving the S2 - S5 vertebrae producing a presacral 24 bulge and extension into bilateral sacral foramina (Figure- 1A). The lesion had internal bony 25 septations with preserved vertebral height and bony outline. The MRI showed an expansile 26 well-marginated T1 hypointense and T2 hyperintense lesion, which was hyperintense in the 27 short tau inversion recovery sequence (STIR) (Figure- 1B). Post-gadolinium injection T1 fat-28 suppressed images showed avid homogeneous lesion enhancement (Figure- 2A). The 29 imaging findings were suggestive of a benign lesion, most likely vertebral body 30 haemangioma (VBH). 31 32 mailto:drsudipta.m@gmail.com A biopsy was planned to exclude malignancy as there was a presacral soft-tissue bulge. The 33 histopathological study revealed readily recognizable vascular structures with red blood cells 34 or transudate, lined by a monolayer of endothelial cells characteristic of haemangioma 35 (Figure- 2B). The patient was managed conservatively with yearly follow-up; there was no 36 interval change in the lesion's size on follow-up MRI. 37 38 Informed consent was obtained from the patient for using his medical data for publication 39 purposes. 40 41 Comment 42 VBH occurs in more than 11% of the population, yet sacral involvement is uncommon. They 43 are seen in adults with a male to female ratio of 1:1.5.1 They are indolent except in < 1% 44 when they become symptomatic either by bone expansion with or without an associated 45 pathological fracture, extension into the neural foramen, or the spinal canal causing 46 radiculopathy or myelopathy and known as aggressive haemangiomas. 2,3 Aggressive 47 haemangiomas present with pain, and they may have an extraosseous soft tissue component 48 contiguous with the osseous lesion. 49 50 The differential diagnoses are chordoma, giant cell tumors, enchondroma, chondrosarcoma, 51 aneurysmal bone cyst, metastases, and rarely hydatid cysts in endemic areas. 1,4,5 52 A haemangioma is well defined with a hyperintense signal on T1- weighted imaging (T1WI) 53 and T2WI due to the fat content and avid homogeneous enhancement on post-contrast 54 imaging. The vascular elements make the signal high on fluid-sensitive sequences. The 55 thickened vertical trabeculae are more appreciated on the CT scans producing the "polka dot 56 sign." Sometimes atypical presentation occurs due to variable amount of fat and vascular 57 components producing an atypical hypo to isointense signal on T1WI and heterogeneous 58 hyperintensity on T2WI and STIR-sequences.2 59 60 Sacral haemangiomas do not require any treatment until they become painful or encroach the 61 sacral nerves. 62 63 This case highlights the presence of a presacral soft tissue component in a haemangioma 64 mimicking a malignant lesion. Accurate identification of imaging findings can reduce patient 65 anxiety and morbidity due to surgical intervention. 66 67 Authors’ Contribution 68 ND, SN and MB were involved in diagnosis, manuscript editing and reviewing the 69 manuscript. SM and MJ were involved in data collection, drafting, editing and reviewing the 70 manuscript. All authors approved the final version of the manuscript. 71 72 References 73 1. 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J Neurosci Rural Pract 2019;10(3):565-6. doi: 86 10.1055/s-0039-1695700. 87 88 Figure 1A: Sagittal CT bone window image shows an expansile soft tissue density lytic 89 lesion involving the S2 to S5 sacral vertebrae with a presacral bulge and extension into sacral 90 foramina. 91 92 Figure 1B: Sagittal T1 Weighted image shows a well-marginated expansile, predominantly 93 hypointense lesion involving the S2 to S5 vertebrae. 94 95 96 Figure 2A: Sagittal post intravenous gadolinium injection T1 fat-suppressed image showing 97 avid enhancement of the lesion with a presacral bulge, extension into the sacral foramen and 98 spinal canal. 99 100 Figure 2B: The histopathology of the biopsy specimen of the sacral lesion (H& E stained, x 101 4 magnification) showing variable-sized blood-filled vascular spaces between mature bony 102 trabeculae, lined by a monolayer of endothelial cells. 103