CASE REPORT |
https://doi.org/10.5005/jojs-10079-1159 |
Navigation-assisted Excision of Heterotopic Ossification in Bilateral Hips of a Paraplegic Patient: A Case Report with Technical Note
1,4–6Department of Orthopaedics, Kauvery Hospital, Chennai, Tamil Nadu, India
2Department of Orthopaedics; Department of Spine Surgery, Kauvery Hospital, Chennai, Tamil Nadu, India
3Department of Spine Surgery, Kauvery Hospital, Chennai, Tamil Nadu, India
Corresponding Author: Keerthivasan Panneerselvam, Department of Orthopaedics; Department of Spine Surgery, Kauvery Hospital, Chennai, Tamil Nadu, India, Phone: +91 9500102299, e-mail: medkeerthi@gmail.com
Received: 21 February 2024; Accepted: 17 March 2024; Published on: 14 Jane 2024
ABSTRACT
Aim and background: Heterotopic ossification (HO), which is common among patients with neurological disorders or chronic bedridden individuals, is a major concern for mobility and rehabilitation. Here, we have discussed a novel technique in the surgical management of HO in a 27-year-old posttraumatic paraplegic patient.
Case description: This is the very first case to our knowledge in which excision of HO in bilateral hips was performed with navigation assistance, after which the patient regained full range of joint movement and intensive physiotherapy was possible.
Conclusion: Excision of HO in this paraplegic patient with bilateral hip ankylosis and knee stiffness and challenges imposed due to preexisting conditions were surpassed with navigation assistance. Three-dimensional (3D) navigation guidance in HO resection may aid in overcoming the surgical challenges and optimizing the final outcome.
Clinical significance: Surgical excision in extensive HO lesions can be a prompt decision for patients, especially in terms of better rehabilitation and prevention of complications. In addition to meticulous preoperative planning, technical guidance with navigation assistance during surgery may serve as a viable option.
How to cite this article: Thangavelan M, Panneerselvam K, Venugopal S, et al. Navigation-assisted Excision of Heterotopic Ossification in Bilateral Hips of a Paraplegic Patient: A Case Report with Technical Note. J Orth Joint Surg 2024;6(2):197–201.
Source of support: Nil
Conflict of interest: None
Patient consent statement: The author(s) have obtained written informed consent from the patient for publication of the case report details and related images.
Keywords: Ankylosis, Case report, Computer navigation, Heterotopic ossification, Hip heterotopic ossification, Myositis ossificans, Navigation-assisted excision, Paraplegic patient, Surgical excision
INTRODUCTION
In heterotopic ossification (HO), abnormal ossification of lamellar bone in the extraskeletal soft tissue is observed, which is most often seen as a complication in rehabilitation settings.1 Heterotopic ossification was first coined as paraosteoarthropathy by two French physicians, Dejerne and Ceilier, who described them as a consequence of traumatic paraplegic patients of the First World War.2 There is a broad age distribution in HO from infancy to late adulthood, showing male predilection with a male-to-female ratio of 3:2. Individuals are prone to develop HO if trauma-induced or due to repetitive mechanical stress from unrecognized microtrauma, especially after a head injury or other categories.2,3 Acute rehabilitation and transfer activities of bedbound patients or repeated microtrauma during activities of daily living may predispose them to HO. Tissue expression of bone morphogenic proteins stimulates mesenchymal spindle stem cells, aka satellite cells, to migrate to the injured areas and metamorphose into fibroblasts, and eventually transform into osteoblasts. Heterotopic ossification consists of two main etiology—traumatic, which includes fracture, muscular trauma, joint dislocation, burns, arthroplasty, and neurogenic, which includes stroke, spinal cord injury, traumatic brain injury, and brain tumors.1 Nongenetic forms of HO are the most common, but rare genetic forms such as fibrodysplasia ossificans progressiva and progressive osseous heteroplasia are also reported. The severity and clinical presentation of the genetic form will differ from that of the nongenetic form of HO.1,3 The clinical presentation depends on the temporal stage of nongenetic HO, which has two phases, namely the early (inflammatory) phase, which presents as localized pain, tenderness, and swelling, and the late phase in which there is gradual maturation into bony tissue and deformed joints. The characteristic clinical features include limitation of range of motion adjacent to the joint involved, complete bony ankylosis (in severe form), deformity in the affected areas like hip, elbow, shoulder, spine, and fingers, jaw exostosis or temporomandibular joint ankylosis.2,3
Heterotopic ossification in paraplegic patients is a common entity that hinders adequate rehabilitation. Owing to various sociopsychological factors, these patients usually present late with complications like pressure sores, recurrent urinary tract infections, and nutritional deficiency. Supple joints are the prerequisite for proper rehabilitation care. Paraplegic and chronic bedridden patients suffer from loss of muscle tone, atrophied skeletal muscles, osteopenia, and stiff joints. Restoring joint anatomy by excision of heterotopic tissues as much as possible and rebuilding the muscle by physical rehabilitation form the main goals of management in such patients. However, the literature reports higher complication rates in these patients due to bacteremia, pressure sores, malnutrition, and the resultant immunocompromised state.
Further, the osteopenic bones secondary to immobility and neurogenic cause an increase in the chance of iatrogenic fractures during the surgical intervention. The decision to proceed with surgical excision in this patient was made to offer improved rehabilitation and prevention of recurrent complications. Several clinical work and evaluations are necessary while planning to manage such cases to overcome the complications and achieve a better outcome. This case report discusses the nuances and benefits of three-dimensional (3D) navigation-guided excision in a paraplegic patient who had developed extensive HO in bilateral hips, complicated with pressure sore, poor soft tissue covering at the hip region, and associated knee joint stiffness. Navigation assistance was used as a valuable tool to surmount the surgical challenges.
CASE DESCRIPTION
Clinical History
A 27-year-old male with a 4-year duration of posttraumatic paraplegia following a D8 vertebral fracture presented with stiffness of bilateral hips and knees. After the initial examination, he was found to have complete loss of both active and passive movements in bilateral hips, 30–70° of passive flexion in both knees, ASIA-A neurology below T10 level. The patient was on an indwelling urinary catheter with recurrent episodes of urinary tract infection. Multiple episodes of pressure sores over the trochanteric region were treated with surgical debridement and skin grafting in the past. The pressure sore on the sacral region was treated conservatively. Blood investigations showed mildly elevated erythrocyte sedimentation rate and C-reactive protein levels. Urine culture showed no growth of organisms. X-ray and computed tomography (CT) imaging (Figs 123) showed bilateral extensive HO involving the gluteal muscles vastus group of muscles extending from the iliac bones to the proximal femur in anterolateral aspect and confirmed the maturity and corticalization of HO bones. 3D reconstructed CT imaging and CT angiogram revealed the proximity of the femoral neurovascular bundles and profound vessels engulfed by the mass.
Fig. 1: Computed tomography pelvis—anteroposterior view
Fig. 2: Computed tomography pelvis—posteroanterior view
Fig. 3: Computed tomography pelvis—oblique view
Surgical Proceedings
The procedure was done under general anesthesia. Through the anterior iliofemoral approach, exposure was done with the assistance of a vascular surgeon. Meticulous dissection was performed, and step-by-step hemostasis was achieved to control blood loss. Major vessels and femoral nerves were identified and isolated. Intraoperative (IO) navigation played a pivotal role in delineating the excessive heterotopic ossified tissues from normal native bone. As there is no navigation software for the excision of myositis ossificans to the best of our knowledge, we used the spine navigation system with some modifications to aid the procedure. The patient tracker of the navigation system was anchored to the iliac crest, and reference array points were placed over the native bone and HO mass and registered, which served as a precise guide showing the 3D anatomical orientation using the preloaded CT images of the patient (Fig. 4).
Fig. 4: Intraoperative navigation assistance for marking reference points.
Another major concern was previous scar tissue from skin grafting, which was found adherent to the bone mass. Navigation played a significant role in identifying the extent of bone attached to the skin. The adherent bone was left unremoved to prevent wound-healing complications.
The ossified extraskeletal mass was intended to be excised so that there is good functional restoration instead of removing the entire mass. Care was taken to neither succeed the excision, keeping in mind the chances of recurrence, nor to remove in excess than the requisite in order to cut down the associated morbidity. The edges of the lesion remnants were smoothened and burred down to prevent impingement of neurovascular structures and skin over the bony prominences, which could be a potential risk for further pressure sores.
Intraoperative Findings
Intraoperative range of movements with flexion of 110°, full range of abduction and rotation was achieved in both hips after removing about a total of 710 gm of bony tissues (Fig. 5). The wound was closed with two surgical drains in situ, one on each side to avoid hematoma complicating the dead space. The total surgical duration for both hips was about 90 minutes. Blood loss of approximately 1500 mL was recorded, where majority of blood loss was encountered during the removal of the cancellous part of the bony mass, for which a liberal amount of bone wax was used. IO blood loss and hypotensive episodes were managed with blood and blood product transfusion.
Fig. 5: Amount of bony tissue removed during surgery from both hips—weighing about 710 gm
Postoperative Recovery and Follow-up
Transfusion was done again in the postoperative period to correct anemia. Indomethacin was started in the immediate postoperative period to reduce the chances of recurrence. Other measures included deep vein thrombosis prophylaxis with low molecular weight heparin and prophylactic antibiotics. A high-protein diet was encouraged. Limited passive lower limb movements and chest physiotherapy were started on postoperative day 1. The patient was mobilized to a wheelchair on postoperative day 2 (Fig. 6). Regular monitoring of blood loss in the surgical drain was done. The wound healed well, and sutures were removed 2 weeks postsurgery. The patient was shifted to our rehabilitation center, where a full range of movement exercises and intensive physiotherapy were initiated after suture removal. The patient is on regular follow-up and is able to sit on a chair, stand with support (Fig. 7), travel in a car to work, and take care of his personal hygiene at 9 months follow-up.
Fig. 6: Postoperative day 2—sitting in a wheelchair
Figs 7A and B: Standing with support—3 months follow-up
Fig. 8: Informed patient consent form
DISCUSSION
Surgical excision in HO is the most effective treatment option, and timely intervention can improve functional outcomes and prevent complications. Excision is usually planned when there is no active inflammatory response and after maturity or mineralization of the HO mass to reduce the recurrence rate.4,5 Shehab et al. have outlined a similar criterion to recommend surgical removal of HO in their publication.4 In neurogenic HO, there is evidence that excision is beneficial even in the early phase before neurological recovery, provided the ectopic tissue is mature enough. It even hastens the neurological recovery in some individuals. Inadvertent delay in surgery might increase the chance of intra-articular involvement, worsening the ankylosis,4 which further increases the risk of iatrogenic fractures.5,6 The main indication of early surgery in paraplegic patients with HO is usually based on the affected mobility due to ankylosis or extreme derangement in joint movements.4-7
In a paraplegic patient, pressure sores are the result of a vicious cycle of prolonged immobilization and improper positioning due to contractures or ankylosis.8 Similarly, in this case, the young patient had trochanteric and sacral pressure sores, complicating the mobilization and vice versa, necessitating surgical measures. To the best of our knowledge, IO navigation was not used to excision the HO. The excision, in this case, posed various surgical challenges, a few of which included the extensive ectopic mass engulfing the profunda artery, poor soft tissue covers secondary to wound scarring, and skin adhered to the bony mass. Hence, 3D IO navigation with a few modifications served as a technical guide to combat the difficulties in accessing the bony tissues and to demarcate the anatomical bone from the ectopic bone mass.
The bone density of heterotopic mass similar to that of native bone was a challenging entity, which was demarcated with IO navigation. Apart from this, other difficulties like lost muscle anatomy due to disuse atrophy and distorted bony outline of the pelvis, as well as the 3D spatial orientation of pelvis bone compared to other long bones, require adequate IO guidance. IO navigation, therefore, was used to surpass the challenges and to transmit the complexity of the surgical site. In addition, navigation played other roles, such as precisely identifying the amount of bone to be left in situ along with the healed adherent skin graft over it. Navigation enhanced the 3D orientation and hence reduced the total duration of surgery, indirectly reducing the amount of blood loss. The contribution of 3D navigation intraoperatively has, therefore, eased the procedure and improved the functional outcome.
It is reported that IO blood loss should be anticipated during surgical removal due to increased vascularity, typically in HO lesions.4 Proximity or encroachment of the ectopic ossification over the blood vessels is another important cause of hemorrhage during resection. In this case, meticulous dissection and isolation of blood vessels were carried out during the procedure to avoid injury. Preoperative arterial embolization can be performed to control IO bleeding,9 but in view of the anticipated morbidity from multiple procedures, it was not adopted as a part of management in this patient. It has been suggested that prostaglandin e2 acts as a mediator in the differentiation of progenitor cells from HO.4,10 Literature offers various clinical trials that support the prophylactic role of nonsteroidal anti-inflammatory drugs like indomethacin in the perioperative period to reduce the chances of recurrence of lesions.4,5,11–14 In the case of this patient, indomethacin was prescribed in the postoperative period for three weeks as prophylaxis.
CONCLUSION
Surgical excision of HO may serve as a prompt choice for patients similar to this case report. Careful patient selection, surgical planning, and technical assistance with 3D navigation add value to the precision of surgery and thereby help meet the anticipated functional outcomes for the patient.
Clinical Significance
Heterotopic ossification can be distressing in paraplegic patients and is a roadway to several complications. Resection at the optimal stage with meticulous planning promotes earlier rehabilitation, which may reduce the burden and prevent further complications. Navigation enhances the 3D orientation to define the surgical landmarks and thereby improves the functional outcome.
ORCID
Mirunaalini Thangavelan https://orcid.org/0009-0007-9941-9213
Keerthivasan Panneerselvam https://orcid.org/0000-0002-5762-2631
Sathish Venugopal https://orcid.org/0000-0002-6764-1908
Aravindan Selvaraj https://orcid.org/0009-0007-3362-4476
Mukunth Krishnamoorthy https://orcid.org/0009-0006-6215-6168
Vikraman Arunachalam https://orcid.org/0009-0005-1054-7645
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