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Embolization followed by resection of the heterotopic hip joint ossification with spinal cord injury

Open AccessPublished:November 08, 2022DOI:https://doi.org/10.1016/j.jos.2022.10.006

      Abstract

      Background

      Heterotopic ossification of large joints, such as knees and hips, has been reported after spinal cord injury, possibly leading to decreased activity of daily living due to a limited range of motion of the affected joint. Therefore, heterotopic ossification resection is performed to improve the range of motion, but it might cause massive bleeding as a complication.

      Methods

      In this case, the patient had a history of spinal cord injury and developed heterotopic ossification after the left hip injury. He had left hip ankylosis and could not transfer to a wheelchair by himself; therefore, heterotopic ossification resection was planned. On conducting contrast-enhanced computed tomography, the supplying arteries extending to the heterotopic ossification could be identified. A day before the surgery, embolization of the branches by interventional radiology was performed.

      Results

      Heterotopic ossification resection was performed with an 820-ml blood loss. Postoperative rehabilitation was continued, and range of motion continued to improve without heterotopic ossification recurrence 2 years post-surgery.

      Conclusions

      The combination of preoperative contrast-enhanced computed tomography and embolization was useful in treating heterotopic ossification.

      Keywords

      1. Introduction

      The cause of heterotopic ossification (HO) remains unclear. However, HO often occurs after total hip arthroplasty, brain or spinal cord injury, and acetabulum and elbow fractures [
      • Forsberg J.A.
      • Pepek J.M.
      • Wagner S.
      • Wilson K.
      • Flint J.
      • Andersen R.C.
      • et al.
      Heterotopic ossification in high-energy wartime extremity injuries: prevalence and risk factors.
      ]. Although the pathogenesis of HO is not elucidated completely, it is known that the local inflammation, hypoxic environment, change in pH, and mechanical stress cause cytokine release and cellular interactions, resulting in progenitor cell transformation from mesenchymal to osteogenic cells and leading to HO [
      • Brunnekreef J.J.
      • Hoogervorst P.
      • Ploegmakers M.J.
      • Rijnen W.H.
      • Schreurs B.W.
      Is etoricoxib effective in preventing heterotopic ossification after primary total hip arthroplasty?.
      ]. HO is frequently seen in cervical and thoracic spinal injuries and mainly affects the hip joint [
      • Ranganathan K.
      • Loder S.
      • Agarwal S.
      • Wong V.W.
      • Forsberg J.
      • Davis T.A.
      • et al.
      Heterotopic ossification: basic-science principles and clinical correlates.
      ].
      HO of the hip joint leads to immobility and a significant decrease in the activity of daily living (ADL) as it progresses. Although surgical resection improves ADL, it can cause massive bleeding due to the proximity of the supplying arteries [
      • Ramirez D.M.
      • Ramirez M.R.
      • Reginato A.M.
      • Medici D.
      Molecular and cellular mechanisms of heterotopic ossification.
      ,
      • Dilling C.F.
      • Wada A.M.
      • Lazard Z.W.
      • Salisbury E.A.
      • Gannon F.H.
      • Vadakkan T.J.
      • et al.
      Vessel formation is induced prior to the appearance of cartilage in BMP-2-mediated heterotopic ossification.
      ,
      • Kim J.H.
      • Park C.
      • Son S.M.
      • Shin W.C.
      • Jang J.Y.
      • Jeong H.S.
      • et al.
      Preoperative arterial embolization of heterotopic ossification around the hip joint.
      ]. Recently, with the widespread use of computed tomography (CT), preoperative contrast-enhanced CT has made it easier to evaluate blood vessels [
      • Zielinski E.
      • Chiang B.J.L.
      • Satpathy J.
      The role of preoperative vascular imaging and embolisation for the surgical resection of bilateral hip heterotopic ossification.
      ]. Here we report a case of a patient who underwent contrast-enhanced CT and embolization of supplying arteries followed by HO resection.

      2. Report of the case

      A 56-year-old male was diagnosed with cervical cord injury (C5, Frankel classification A, Zancolli classification C6A) 6 years ago due to a fall after drinking alcohol. After rehabilitation, he could independently transfer to a wheelchair. Past medical history included neuropathic bladder and angina. He fell from the wheelchair, injured his left hip 9 months ago, and visited a nearby clinic. There were no obvious fractures on radiographs (Fig. 1). In addition, based on the magnetic resonance imaging (short inversion-time inversion recovery), he was diagnosed with muscles (left iliacus, abductors and rectus femoris) and bone (left lessor trochanter) bruise and treated conservatively (Fig. 2a and b). After that, left groin pain and left hip flexion contracture appeared, increasing the burden of care. On return to the clinic 7 months later, the left hip HO was identified on radiographs (Fig. 3a and b), and he was referred to our department for further examination and treatment. At his first visit to our hospital, he presented with ankylosis of the left hip (fused at 25° on flexion, 20° on abduction, and 10° on external rotation) and could not transfer to a wheelchair by himself. Due to ankylosis of the left hip and resulting limitation of life activity, surgical resection was planned. Preoperative contrast-enhanced three-dimensional CT showed large HO anteriorly at the hip joint (Fig. 4a), and the supplying arteries extending to the HO were detected as the branches of the deep femoral artery (Fig. 4b). After consultation with a radiologist, embolization of the branches by interventional radiology was performed the day before surgery (Fig. 4c). Surgical resection was performed in a supine position under general anesthesia. The HO of the anterior hip was resected piece by piece with a chisel through the direct anterior approach. Intraoperatively, a C-arm image intensifier was used to confirm the ossification lesion. The estimated blood loss was 820 mL during the surgery. The range of motion on the left hip improved to 90° on flexion and −10° on extension.
      Fig. 1
      Fig. 1Initial radiograph revealing no obvious fractures of the left hip.
      Fig. 2
      Fig. 2Initial magnetic resonance imaging (short inversion-time inversion recovery) revealed wide extent of muscle bruise around left iliacus, abductors and rectus femoris. And also revealed bone bruise around left lessor trochanter (a, b).
      Fig. 3
      Fig. 3Radiographs after seven months revealing heterotopic ossification of the left hip (a, b).
      Fig. 4
      Fig. 4Preoperative three-dimensional computed tomography (CT) (a) and contrast-enhanced CT (b). The arteries extending to heterotopic ossification are seen as the branches of the deep femoral artery. Preoperative embolization of the branches (c).
      After the surgery, oral administration of bisphosphonate (Etidronate 800 mg/day) and cyclooxygenase (COX)-2 inhibitor (Celecoxib 200 mg/day) was started to prevent an HO recurrence. Prophylactic radiation was not performed after the surgery. Physical therapy was also started. Three weeks later, the patient could transfer to the wheelchair with light caregiving, and he was discharged from the hospital. Fourteen months after the surgery, there was no radiographic evidence of HO (Fig. 5a and b), and the range of motion of the left hip was 90° on flexion, −10° on extension, 10° on abduction, 0° on adduction, 30° on eternal rotation and 10° on internal rotation. His ADL improved, and he could independently transfer to a wheelchair.
      Fig. 5
      Fig. 5Radiograph after 14 months of heterotopic ossification (a, b).

      3. Discussion

      The patient in this case had a history of spinal cord injury and developed HO after the left hip injury. According to Garland et al., HO after spinal cord injury was almost always evident on radiographs within 6 months of injury, and some patients tended to develop HO after 6 months [
      • Garland D.E.
      A clinical perspective on common forms of acquired heterotopic ossification.
      ]. Although the mechanism of HO after spinal cord injury remains unknown, it is possible that HO in this case was related to the history of spinal cord injury.
      Shehab et al. [
      • Shehab D.
      • Elgazzar A.H.
      • Collier B.D.
      Heterotopic ossification.
      ] previously reported the criteria for resection of HO, and one of which was a significantly limited range of motion. Kim et al. [
      • Kim J.H.
      • Park C.
      • Son S.M.
      • Shin W.C.
      • Jang J.Y.
      • Jeong H.S.
      • et al.
      Preoperative arterial embolization of heterotopic ossification around the hip joint.
      ] also reported that a grade Ⅲ or Ⅳ HO, according to the Brooker classification [
      • Warren S.B.
      • Brooker Jr., A.F.
      Intramedullary nailing of tibial nonunions.
      ], leads to limitations of range of motion, and surgical treatment is needed in order to improve the condition. In this case, owing to ankylosis of the left hip and the resulting limitation of life activities, surgical resection was planned.
      One of the major complications of surgical resection of HO is intraoperative bleeding [
      • Yoon B.H.
      • Park I.K.
      • Sung Y.B.
      Ankylosing neurogenic myositis ossificans of the hip: a case series and review of literature.
      ]. As increasing vascularization of the HO was reported [
      • Dilling C.F.
      • Wada A.M.
      • Lazard Z.W.
      • Salisbury E.A.
      • Gannon F.H.
      • Vadakkan T.J.
      • et al.
      Vessel formation is induced prior to the appearance of cartilage in BMP-2-mediated heterotopic ossification.
      ] and HO of the hip joint was adjacent to the femoral arteries and veins, it was possible to lacerate vessels during HO resection and cause massive bleeding. Garland et al. [
      • Garland D.E.
      • Hanscom D.A.
      • Keenan M.A.
      • Smith C.
      • Moore T.
      Resection of heterotopic ossification in the adult with head trauma.
      ] reported a case of a patient in whom surgical resection resulted in a blood loss of 5000 mL due to femoral vein laceration. Contrast-enhanced CT is commonly used to evaluate blood vessel, and can identify supplying vessels as well as the extent of heterotopic ossification. The images taken by contrast-enhanced CT can be discussed with the radiologist to plan for embolization, and can also help in surgical planning by identifying the adjacent arteriovenous vessels. For these reasons, contrast-enhanced CT might be useful for HO resection. In our study, the arteries supplying the HO were identified as the branches of the deep femoral artery. Thus, preoperative embolization of these arteries was performed. Consequently, surgical resection could be performed with the 820-mL blood loss.
      Verifying that preoperative embolization is useful [
      • Vogl T.J.
      • Wolff J.D.
      • Balzer J.
      • Skripitz R.
      Preoperative arterial embolization in heterotopic ossification: a case report.
      ], Kim et al. [
      • Kim J.H.
      • Park C.
      • Son S.M.
      • Shin W.C.
      • Jang J.Y.
      • Jeong H.S.
      • et al.
      Preoperative arterial embolization of heterotopic ossification around the hip joint.
      ] reported a case of bilateral hip HO with the right side bleeding of 1500 mL, whereas the left side underwent preoperative embolization of the superior gluteal and the lateral circumflex femoral arteries, limiting the left side bleeding to 500 mL. Zielinski et al. [
      • Zielinski E.
      • Chiang B.J.L.
      • Satpathy J.
      The role of preoperative vascular imaging and embolisation for the surgical resection of bilateral hip heterotopic ossification.
      ] also reported a case of bilateral hip HO with preoperative embolization of both medial femoral circumflex arteries with a bleeding volume of 750 mL and 800 mL on the right and left sides, respectively. Embolization before HO resection might allow a safer surgery with less blood loss. As other reports included the intravenous administration of tranexamic acid during the hip surgery to prevent intraoperative bleeding [
      • Qi Y.M.
      • Wang H.P.
      • Li Y.J.
      • Ma B.B.
      • Xie T.
      • Wang C.
      • et al.
      The efficacy and safety of intravenous tranexamic acid in hip fracture surgery: a systematic review and meta-analysis.
      ], it might also be effective in hip HO resection.
      Garland et al. [
      • Garland D.E.
      • Hanscom D.A.
      • Keenan M.A.
      • Smith C.
      • Moore T.
      Resection of heterotopic ossification in the adult with head trauma.
      ] classified HO into five types according to the neural residua (cognitive and physical disability), and showed that the more severe type was associated with a worse outcome and a higher the recurrence rate. This case was classified as Ⅴ type with severe physical disability, and was predicted to possibly have a high recurrence rate. For postoperative medications, a COX-2 inhibitor and a bisphosphonate were used. NON-steroidal anti-inflammatory medications are known to prevent HO by inhibiting COX function [
      • Banovac K.
      • Williams J.M.
      • Patrick L.D.
      • Levi A.
      Prevention of heterotopic ossification after spinal cord injury with COX-2 selective inhibitor (rofecoxib).
      ]. As COX-1 inhibitors have gastrointestinal side effects, the use of COX-2 inhibitors should be considered. In Japan, bisphosphonates are commonly indicated for the prevention of HO after spinal cord injury (800–100 mg/day for 3 months). These medications are helpful to prevent HO recurrence, and thus, they were used in this case. Radiation is also effective in preventing HO, but its indications are limited. It was reported that a single dose of 700–800 cGy administered from 24 h preoperatively to 48–72 h postoperatively could prevent HO [
      • Popovic M.
      • Agarwal A.
      • Zhang L.
      • Yip C.
      • Kreder H.J.
      • Nousiainen M.T.
      • et al.
      Radiotherapy for the prophylaxis of heterotopic ossification: a systematic review and meta-analysis of published data.
      ]. However, radiation has serious side effects, including fibrosis, nonunion, and delayed wound healing [
      • Hamid N.
      • Ashraf N.
      • Bosse M.J.
      • Connor P.M.
      • Kellam J.F.
      • Sims S.H.
      • et al.
      Radiation therapy for heterotopic ossification prophylaxis acutely after elbow trauma: a prospective randomized study.
      ]. HO-preventative radiation therapy is uncommon in our country and was not used in this case because of concerns about delayed wound healing. Although it has not yet been reported that embolization significantly reduces the postoperative recurrence of HO, there are some case reports in which concomitant embolization was useful for preventing HO recurrence [
      • Kim J.H.
      • Park C.
      • Son S.M.
      • Shin W.C.
      • Jang J.Y.
      • Jeong H.S.
      • et al.
      Preoperative arterial embolization of heterotopic ossification around the hip joint.
      ,
      • Zielinski E.
      • Chiang B.J.L.
      • Satpathy J.
      The role of preoperative vascular imaging and embolisation for the surgical resection of bilateral hip heterotopic ossification.
      ,
      • Vogl T.J.
      • Wolff J.D.
      • Balzer J.
      • Skripitz R.
      Preoperative arterial embolization in heterotopic ossification: a case report.
      ]. Additionally several reports have implied the existence of arteries supplying HO when HO has formed [
      • Vogl T.J.
      • Wolff J.D.
      • Balzer J.
      • Skripitz R.
      Preoperative arterial embolization in heterotopic ossification: a case report.
      ,
      • Dilling C.F.
      • Wada A.M.
      • Lazard Z.W.
      • Salisbury E.A.
      • Gannon F.H.
      • Vadakkan T.J.
      • et al.
      Vessel formation is induced prior to the appearance of cartilage in BMP-2-mediated heterotopic ossification.
      ]. Therefore, identification and embolization of the supply arteries may help prevent recurrence. Increasing the number of embolization cases before HO resection will reveal the effectiveness of embolization in preventing recurrence.

      Informed consent

      The patients and their families were informed that data from the research would be submitted for publication, and gave their consent.

      Declaration of competing interest

      None declared.

      Acknowledgments

      No acknowledgments.

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