Ilizarov technique for treating elbow stiffness caused by myositis ossificans: A case report

Abstract

BACKGROUND

Myositis ossificans (MO) is a rare disease involving the formation of bone outside the musculoskeletal system. While surgical intervention is the main treatment approach, preventing recurrence and standardized rehabilitation are also crucial. Here, we present a surgical strategy to prevent the recurrence of MO.

CASE SUMMARY

A 28-year-old female patient was admitted for the first time for a comminuted fracture of the left olecranon. However, incorrect postoperative rehabilitation resulted in the development of elbow joint stiffness with ectopic ossification, causing a loss of normal range of motion. The patient was diagnosed with MO based on physical examination, X-ray findings, and clinical presentation. We devised a surgical strategy to remove MO, followed by fixation with an Ilizarov frame, and implemented a scientifically reasonable rehabilitation plan. The surgery lasted for 3 h with an estimated blood loss of 45 mL. A drainage tube was placed after surgery, and fluid was aspirated through ultrasound-guided puncture. The patient experienced a significant reduction in joint stiffness after surgery. In the final follow-up at 9 mouths, there was evident improvement in the range of motion of the elbow joint, and no other symptoms were reported.

CONCLUSION

The Ilizarov frame is an advantageous surgical technique for facilitating rehabilitation after MO removal. It offers benefits such as passive recovery, individualized treatment, and prompt recovery.

Key Words: Myositis ossificans; Ilizarov frame; Rehabilitation strategy; Stiffness; Case report

Core Tip: In this case report, we report a 28-year-old female with myositis ossificans. The patient had an ulnar humerus fracture early in the course of the disease, and after internal fixation treatment, the wrong rehabilitation method led to the occurrence of myositis ossificans. In this operation, we normalized the rehabilitation plan of the patient by the passive pulling force of Ilizarov technology, which made no recurrence after the removal of myositis ossificans, and achieved a good effect.

INTRODUCTION

Myositis ossificans (MO) is a rare disease characterized by ectopic ossification within soft tissues[1]. It typically occurs following trauma and affects the major muscles of the limbs. However, it can also occur throughout the body, but is more commonly seen in the skeletal muscles of the limbs (such as the quadriceps femoris in the lower limb, the muscles on the inner side of the abdomen, or the muscles in the upper arm) or the soft tissues around joints (such as fibrous tissue, subcutaneous tissue, ligaments, and even blood vessel walls)[2,3]. The etiology and pathogenesis of this disease are still unclear. Trauma and intense exercise can cause muscle rupture, hemorrhage, and hematoma formation[4]. Most patients absorb the hematoma, but some may also develop muscle stiffness and ossification, eventually leading to joint stiffness and disability in the affected muscles. Surgery has been identified as a potential cause of MO, with factors such as surgical trauma, inflammation at the surgical site, postoperative rehabilitation, rough surgical technique, free bone fragments, impaired blood circulation at the surgical site, as well as incorrect rehabilitation exercises and secondary injuries being possible contributing factors[5].

However, MO often presents with atypical clinical symptoms and has limited imaging diagnosis for early lesions, leading to confusion with benign and malignant conditions such as musculoskeletal infections, soft tissue sarcomas, and osteosarcomas, which can result in missed diagnosis and misdiagnosis, thereby delaying timely recovery of patients[6]. Pathological biopsy is the gold standard for diagnosing MO, and computed tomography shows typical features as the lesion matures.

Enhancing clinical awareness and attention to MO is necessary because, if left untreated, it can have a significant impact on physical health and increase the economic burden on the patient's family and society. Timely and appropriate treatment and prevention of recurrence of ectopic ossification are crucial, although surgical strategies commonly used for treating MO are challenging in terms of preventing recurrence and ensuring recovery. Based on this, a strategy using the Ilizarov frame is proposed for treatment and prevention of recurrence[7].

We present a case of a 28-year-old woman with nontraumatic MO of the elbow joint, and review the relevant literature.

CASE PRESENTATION

Chief complaints

The patient was a 28-year-old woman who sought treatment at our hospital because of unsatisfactory results following her first elbow joint surgery.

History of present illness

When the patient came to our hospital for treatment, she experienced stiffness and pain in the elbow joint, along with limited bending and straightening ability.

History of past illness

Six months ago, the patient sustained a cycling injury that resulted in a comminuted fracture of the left olecranon (Figure 1A). At our hospital, the patient underwent open reduction and internal fixation surgery for the fracture (Figure 1B). Upon discharge, the patient experienced pain and had difficulty in extending and flexing the left elbow. Unfortunately, she did not participate in timely rehabilitation exercises after discharge, leading to stiffness in the elbow joint. Realizing this, she attempted rough rehabilitation training and massage, which exacerbated the condition. The patient had no history of high blood pressure, diabetes or tumors.

Figure 1 Olecranon fracture and evolution of myositis ossificans after internal fixation. A: 3D reconstruction shows comminuted fracture of the scaphoid bone; B: 3D after surgery; C: 6 wk after surgery, ossifying myositis appeared; D: 10 wk after surgery, evidence of myositis ossificans.

Personal and family history

The patient had no family history of relevant illnesses.

Physical examination

The patient had a body temperature of 36.4°C, blood pressure of 112/72 mmHg, respiratory rate of 12 breaths/min, heart rate of 68 bpm, and was fully alert. A 5-cm surgical scar in the left elbow region was observed, which exhibited no tenderness or percussion pain. Additionally, a hard mass with mild tenderness was palpable in the distal end of the humerus. The left elbow joint presented with a flexion deformity of 90° and limited extension and flexion movement of 30°. Although the forearm was capable of rotation, there were no abnormalities in skin sensation or peripheral blood flow in the left hand. The patient had muscle strength of grade 3 in the left upper limb and grade 5 in the right upper limb.

Laboratory examinations

No abnormalities were found in the laboratory examinations.

Imaging examinations

X-ray examination conducted 6 wk after surgery revealed progressive MO at the site of the previous fracture of the left ulna and elbow joint (Figure 1C). Further assessment resulted in a diagnosis of postoperative elbow stiffness with MO after internal fixation. The left ulnar fracture remained internally fixed, but heterotopic ossification developed after fracture of the left elbow joint. The H-G classification categorized it as type IIIA (Figure 1D).

FINAL DIAGNOSIS

The clinical diagnosis was MO causing stiffness in the elbow joint.

TREATMENT

After routine preoperative examination and treatment, the decision was made to perform internal fixation removal, MO excision, elbow joint release, and combined orthopedic surgery. The operations were conducted with the patient in the supine position and under nerve block anesthesia. Following routine iodine disinfection of the left upper limb, sterile dressings were applied. The incision was made on the posterior side, extending proximally for 12 cm (Figure 2A). Subsequently, the original tension band on the ulnar side was removed, revealing adhesions between the triceps brachii and the distal humerus. Blunt dissection of the triceps brachii exposed the ossification tissue covering the posterior aspect of the distal humerus, resulting in disappearance of the olecranon fossa. To remove the excess bone, a bone knife was used, which completely exposed the original bone cortex of the distal humerus and the olecranon fossa (Figure 2B). Following this, local release of the elbow joint was performed, followed by passive movement of the elbow joint (Figure 2C). The range of motion achieved for the elbow joint was 150° of flexion (Figure 2D) and 20° of extension (Figure 2E). The preinstalled Ilizarov frame was installed on the upper left limb, with the rotation center of the Ilizarov frame maintained in the inner and outer condyle planes of the humerus. Two 2.0 g pins were crossed and fixed at the proximal end of the ulna, and two 2.0 g Kirschner wires were parallel fixed at the distal end of the radius. Additionally, two 3.5mm partially threaded pins were fixed in the middle section of the humeral bone. To prevent adhesion, a tendon antiadhesive membrane was placed between the posterior side of the distal humerus and the triceps brachii muscle. A specialized drainage tube was also inserted. Finally, the incision was closed layer by layer with sutures, and sterile dressings were applied for dressing purposes.

Figure 2 Clearance of myositis ossificans during surgery and measurement of joint motion. A: The surgical field was adequately exposed before removing the bone spur; B: The bone spur was removed during surgery; C: Visual field of elbow joint after removal of bone spurs; D: Measurement of elbow joint range of motion: Elbow flexion was 150; E: Elbow extension was 20.

OUTCOME AND FOLLOW-UP

In the first 3 d after surgery, a drainage tube was placed and 160 mL bloody fluid was drained. Postoperative X-ray showed that the joint alignment was good and there were no obvious abnormalities (Figure 3A and B). After 3 d, the drainage tube was removed and ultrasound-guided puncture was performed to ensure no local hematoma. Passive flexion adjustment of the Ilizarov frame was conducted once in the morning and once in the evening during this time. 2 wk after surgery, the patient was instructed to perform active flexion and extension exercises with the relaxed Ilizarov frame, as well as muscle exercises for nearby joints such as the hands, wrists and shoulders. At this point, passive adjustment of the Ilizarov frame was increased to three times a day. After 6 wk, when the patient muscle function and elbow joint mobility had fully recovered and there was no excessive stimulation from the Ilizarov frame, the frame was removed (Figure 3C and D). The patient was instructed to engage in routine functional exercises as part of her rehabilitation plan. 8 wk after the operation, X-rays showed that the joint alignment was good and there were no obvious abnormalities (Figure 3E and F). After surgery, the symptoms improved significantly. The patient reported a noticeable recovery of joint mobility and no discomfort symptoms during the 9-month follow-up (Figure 3G and H).

Figure 3 After the Ilizarov frame was installed and removed. A and B: 2 d postoperatively; C and D: 6 wk postoperatively; E and F: 8 wk postoperatively; G and H: 8 months postoperatively.

DISCUSSION

The development of MO in our patient may be attributed to three factors. First, the initial fracture surgery inflicted significant trauma, which laid the foundation for MO development[8]. Second, we suspect that the initial surgery was performed roughly, resulting in joint tension and muscle atrophy in the surrounding area. Moreover, the omission of a drainage tube during the operation led to the formation of a local hematoma that was not promptly treated. Lastly, due to joint pain, the patient did not undergo appropriate rehabilitation exercises and instead engaged in rough rehabilitation training and massage after experiencing joint stiffness, consequently further promoting MO development[9].

Violence-induced joint and surrounding soft tissue injuries have been found to have a significant impact on local blood circulation, resulting in local swelling[10]. During the early stage after trauma, the acute phase reaction leads to an increase in the levels of inflammatory factors at the injury site, thus triggering a local inflammatory response and tissue repair[11,12]. This reaction also increases the levels of signaling pathways associated with signal transduction, as well as bone-inducing factors such as transforming growth factor-β, leading to the proliferation and differentiation of osteoprogenitor cells and angiogenesis, consequently stimulating MO occurrence[13]. Several studies have documented a strong correlation between coronavirus disease 2019 (COVID-19) and the development of ectopic ossification[14,15]. Meanwhile, MO cases have occurred among patients receiving the COVID-19 vaccine[16,17].

Excessive rehabilitation training can lead to tissue damage and inflammation, which activates a series of biochemical reactions[18]. These reactions trigger an inflammatory response, activate immune cells, and produce inflammatory mediators[19]. Prolonged inflammation may cause abnormal changes in local tissues, promoting the occurrence of heterotopic ossification. Moreover, abnormal changes in cell signaling pathways can occur due to excessive rehabilitation training, leading to the gradual ossification of soft tissues[20]. For instance, heightened activity in certain signaling pathways may induce cells to differentiate into bone cells instead of their original cell types, such as muscle cells. Some studies indicate that bone cells might play a role in the process of heterotopic ossification. As a result, the tissue damage and inflammation caused by excessive rehabilitation training can activate bone cells, ultimately leading to the formation of bone within the soft tissues[21,22].

Before surgery, several factors should be considered. First, it is crucial to acknowledge that patients may lack knowledge about conventional rehabilitation. Second, the use of the Ilizarov frame for passive traction can effectively enhance injury repair and facilitate smooth blood supply[23]. Additionally, it aids in performing passive functional exercises and prevents various complications that arise from premature, improper rehabilitation exercises. Third, the Ilizarov frame provides stable skeletal support, reducing the risk of MO caused by accidental trauma. Moreover, it assists in coordinating the patient's rehabilitation exercises, thereby promoting muscle and surrounding soft tissue repair[24]. Lastly, removing the Ilizarov frame allows for better recovery, which is advantageous for providing guidance on subsequent effective rehabilitation exercises.

The Ilizarov frame technique offers several advantages. First, it provides good stability and support, which effectively stabilizes the site of the fracture or bone lesion, prevents displacement, and promotes bone healing. Second, the Ilizarov frame can be adjusted in terms of angle and position according to the specific conditions of different patients, enabling individualized treatment. Third, it allows for the preservation of joint mobility, making it particularly useful in cases of joint injury by facilitating rehabilitation. Lastly, the Ilizarov frame eliminates the need for extensive resection or internal implants, thereby reducing the risks associated with surgery. Compared to open surgery, it involves a smaller incision and potentially lowers the risk of complications.

The installation of the Ilizarov frame may cause pain, discomfort and skin damage, requiring the patient's patience and maintenance. Complications such as skin problems and nerve damage may also occur. Moreover, the Ilizarov frame may increase the risk of infection as it needs to penetrate the skin. However, despite these potential drawbacks, the Ilizarov frame is widely used in orthopedic surgery due to its effectiveness in treating complex fractures and deformities.

CONCLUSION

The Ilizarov frame may be considered as a viable alternative for surgical treatment and prevention of recurrence in MO. It offers the advantages of minimal invasiveness and quick recovery. However, long-term follow-up is necessary to determine the effectiveness of this procedure.