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Rotational Osteotomy of the Humeral Shaft for Malunion in Osteogenesis Imperfecta

A Case Report

Nicholson, Thema A. MSc1; Elder, Alexandra BS2; Gallen, Kate1; Namdari, Surena MD, MSc1

doi: 10.2106/JBJS.CC.18.00161
Case Reports
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Disclosures

Case: We report a case of an adult with osteogenesis imperfecta, who underwent humeral shaft osteotomy to correct an internal rotation deformity results from a humeral shaft malunion. The patient incurred a temporary radial nerve palsy.

Conclusions: Humeral shaft malunion can be successfully treated with rotational osteotomy of the humerus through an anterior approach; however, the risk of radial nerve palsy exists and may be minimized by a posterior approach.

1Department of Orthopaedic Surgery, Rothman Institute—Thomas Jefferson University, Philadelphia, Pennsylvania

2Sidney Kimmel Medical College—Thomas Jefferson University, Philadelphia, Pennsylvania

E-mail address for S. Namdari: surena.namdari@rothmaninstitute.com

Investigation performed at the Rothman Institute, Thomas Jefferson University Hospitals, Philadelphia, Pennsylvania

Disclosure: The Disclosure of Potential Conflicts of Interest forms are provided with the online version of the article (http://links.lww.com/JBJSCC/A836).

Fractures of the shaft of the humerus account for 1% to 3% of all fractures and approximately 20% of all humeral fractures1. Humeral shaft fractures are generally treated nonoperatively by immobilization using a functional brace. Given the wide range of motion of the shoulder, malalignment of the humeral shaft is often well tolerated. Because of this, parameters for acceptable fracture alignment have traditionally included 20° of anterior bowing, 30° of varus angulation, 15° of malrotation, and 3 cm of shortening or bayonet apposition2. Severe functional deficits as a result of humeral shaft fractures are rare with proper reduction and functional bracing, with 2% of patients treated nonoperatively developing a varus deformity of more than 25° in a large series of patients treated by Sarmiento et al3. Despite the generally excellent results with nonoperative treatment, malunion can result in some range of motion deficits4.

Rotational osteotomy is a surgical technique used for the purpose of correcting functional rotational deficits of extremities. Rotational osteotomies are most commonly performed at the level of the proximal third of the humerus (proximal to the deltoid insertion) for sequelae of brachial plexus birth injury5. Though rare, rotational osteotomy can also be indicated in selected cases of fracture malunion that result in functional deficits.

Type 1 osteogenesis imperfecta (OI) is a rare genetic disorder of the connective tissue resulting in osteopenia and bone fragility6. Most cases are caused by a mutation in the type 1 collagen genes COL1A1 or COL1A2, with 90% of mutations occurring in one of these 2 genes7. There is no cure for OI and treatment is directed toward preventing or controlling symptoms, maximizing mobility, as well as developing optimal bone mass and muscle strength.

This case illustrates a patient with OI and a long history of left humeral shaft fracture malunion in which a humeral osteotomy was performed to correct the internal rotation deformity.

The patient was informed that data concerning the case would be submitted for publication, and he provided consent.

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Case Report

A 28-year-old, righthand-dominant Asian male with a history of type 1 OI presented for evaluation of a malunited humeral shaft fracture that interfered with his daily activities. As a child, he sustained a humeral fracture that was treated with functional bracing. In adulthood, he experienced difficulty with external rotation tasks. Primarily, the patient worked as a software engineer and had difficulty externally rotating the left arm to gain access to the entirety of the keyboard.

On physical examination, the patient had full range of motion of the elbow. On the affected side, active and passive shoulder motion was 175° of flexion, 170° of abduction, 45° of external rotation with the arm at the side, and internal rotation to the T2 vertebral level. Active and passive shoulder motion on the contralateral side was 175° of flexion, 170° of abduction, 90° of external rotation with the arm at the side, and internal rotation to the T6 vertebral level. Plain radiographs (Fig. 1) and a computed tomography (CT) scan demonstrated a healed humeral shaft fracture. The CT scan was utilized to confirm complete fracture union but not to determine the exact rotational deformity. To obtain the most reliable assessment of rotational deformity, bilateral CT scans would be needed to compare version to the normal side8. Given that the patient had presented with a CT scan, in discussion with the patient, it was decided not to incur further radiation by performing 2 additional scans.

Fig. 1

Fig. 1

Given the patient's history of OI, lack of pain, and fairly well-preserved function, the patient was counseled at length regarding the risks of surgical correction. The patient ultimately presented to 4 preoperative visits in the clinic for further discussion and counseling. In particular, given the location of the malunion, the risk of radial nerve injury was emphasized. Having undergone previous successful and uncomplicated osteotomies in his foot, he elected to undergo a humerus rotational osteotomy to correct the malunion. The goal of this surgery was to correct his structural and functional deficit created by the 45° internal rotation malunion.

On the day of the procedure, the patient underwent a preoperative interscalene nerve block. In the operating room, the extremity was examined and compared to the contralateral side (Fig. 2) before draping on the operative table. The same 45° rotational deformity was present under anesthesia. The patient was placed in the supine position and a hand table was used. An anterior incision was made along the length of the humerus. Dissection was carried to the level of the biceps fascia, which was opened. The short head of the biceps was retracted medially and the musculocutaneous nerve was identified and protected. The radial nerve was the identified distally between the brachialis and brachioradialis and traced proximally to the point at which it crossed the humerus posteriorly. By retracting the biceps medially and the pectoralis major proximally, the radial nerve was also identified proximally approximately 2 to 3 cm medial to the latissimus dorsi insertion and traced laterally to the point at which it crossed the humerus posteriorly. The radial nerve was then able to be palpated as it crossed the posterior humerus and protected. The brachialis muscle was then split in its internervous plane to expose the humeral shaft. Two 3-mm Steinmann pins were placed above and below the site of the planned osteotomy to judge rotation. The nerve was palpated along its course behind the humerus and care was taken to ensure that it was not in the osteotomy site. Two Homan retractors were placed around the humerus to protect the radial nerve. The osteotomy was initiated in a transverse fashion distal to the deltoid insertion. The distal aspect of the humeral shaft was rotated 45° externally with the assistance of the Steinman pins. The ends of the osteotomy were held reduced and compressed with modified pointed reduction clamps. An 8-hole limited contact dynamic compression plate (DePuy Synthes) was placed anteriorly and additional compression was obtained sequentially with placement of eccentrically drilled screws. For added rotational stability, a 5-hole 3.5 mm reconstruction plate (Depuy-Synthes) was placed on the lateral aspect of the shaft at the osteotomy site (Fig. 3). The osteotomy position, reduction, and all hardware positions were confirmed by fluoroscopy. Restoration of shoulder passive external rotation of the shoulder was determined on the operative table. A layered wound closure was undertaken. The patient's arm was placed in a sling.

Fig. 2

Fig. 2

Fig. 3

Fig. 3

The patient was discharged on the same day and seen again 11 days after surgery. During this visit, the patient had a well-healing surgical incision and x-rays revealed a well-positioned plate and screw construct. However, the patient noted difficulty with wrist and thumb extension, indicative of a radial nerve palsy. The patient had a normal sensory examination. He was prescribed a “cock-up” wrist splint and prescribed occupational therapy for the hand and wrist. At 6 weeks after surgery, there were no complaints of pain and x-ray confirmed that the plate and screws were still well positioned. A visible 1-mm fracture line still existed. Symptoms of his radial nerve palsy persisted. At 3 months after surgery, an electromyography obtained, which confirmed the presence of a subacute, severe axonal radial nerve lesion. At his 4-month postoperative visit, wrist extension had returned, but the patient still lacked finger and thumb extension. At 6 months after surgery, the patient had regained full finger and thumb extension and was only lacking strength with extension of his index finger. X-rays were obtained during this visit, which showed a healed humeral osteotomy (Fig. 4). At 1-year after surgery, nerve function had fully recovered and he was performing all of daily activities without problems or complaints. On examination, the left humerus demonstrated a well-healed incision, and rotation was equal on both sides at approximately 90° (Fig. 5). At 29 months after surgery, Quick Disability of Arm Shoulder and Hand Score was 0.

Fig. 4

Fig. 4

Fig. 5

Fig. 5

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Discussion

We describe a case of humeral rotational osteotomy used to correct an internal rotation malunion after a humeral shaft fracture in a patient with OI. An anterior approach with dual plating was used. Although the radial nerve was identified and protected, transient nerve injury occurred. This may suggest a role for a posterior approach with radial nerve neurolysis before corrective osteotomy in these cases.

Rotational osteotomy of the humerus is most commonly used to improve upper extremity function and correct deformity in patients with internal rotation deformity after brachial plexus birth palsy9. Proximal humeral osteotomies have been described for sequelae of locked shoulder dislocations10 and supracondylar osteotomies have been described for cubitus varus deformity11. Humeral shaft osteotomies for sequelae of nonoperatively treated humeral shaft fractures have not been described.

The patient discussed sustained a radial nerve palsy. The nerve is particularly vulnerable at the midshaft of the humerus where it is in direct contact with the periosteum at the spiral groove. Because of its proximity to the humeral shaft, the radial nerve is the most frequently injured major nerve in the upper limb12. In this case, the nerve could have been damaged by surgical instruments (saw blade, Kirschner wires, or retractors), hardware implanted (screws), during manipulation of the osteotomy site, or during nerve dissection. Although we performed the surgery through an anterior approach, the radial nerve was identified both proximal and distal to the osteotomy site and was protected. For this reason, the nerve was not felt to have been lacerated and was not explored in the early postoperative setting. In the future, we would consider performing humeral shaft osteotomies through a posterior approach to allow for a formal radial nerve neurolysis13. Additionally, an interscalene nerve block was performed in this case and delayed the diagnosis of the nerve palsy until the first postoperative visit. We do not believe that this influenced the final result because the nerve would not have been explored even if diagnosis was made earlier.

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References

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