Heterotopic ossification of the shoulder in patients with traumatic brain injury

J Shoulder Elbow Surg (2013) 22, 52-56

www.elsevier.com/locate/ymse

Heterotopic ossification of the shoulder in patients with traumatic brain injury David A. Fuller, MDa, Usha S. Mani, MDb,*, Mary Ann E. Keenan, MDc a

Cooper University, University of Medicine and Dentistry of New Jersey, Camden, NJ, USA Methodist Dallas Medical Center, Dallas, TX, USA c University of Pennsylvania, Philadelphia, PA, USA b

Background: We retrospectively reviewed 10 consecutive patients (11 shoulders) with traumatic brain injury who underwent surgical resection of heterotopic ossification (HO) of the shoulder. Our primary research goal was to determine the change in range of motion (ROM) at the shoulder after resection of heterotopic ossification in patients with traumatic brain injury. Secondary research goals were to determine simple functional outcome gains related to activities of daily living and to determine complications in this patient cohort. Methods and materials: Data were collected retrospectively and included measured ROM of the shoulder and observed ability to perform daily activities. The average age of the patients was 33 years (range, 20 -45). Results: Sagittal plane motion (flexion/extension) increased by 85.0 , coronal plane motion (adduction/ abduction) increased by 59.1 , and axial plane motion (internal/external rotation) increased by 66.8 (P < .001). Nine patients increased independence with improved functional status (7 patients able to perform all 3 activities of feeding, grooming, and toiletry) (P < .001). Conclusion: Surgical resection of heterotopic ossification of the shoulder is an effective procedure to increase joint mobility and improve function. Level of evidence: Level IV, Case Series, Treatment Study. Ó 2013 Journal of Shoulder and Elbow Surgery Board of Trustees. Keywords: Shoulder; heterotopic ossification; traumatic brain injury; surgical ressection; range of motion; activities of daily living

Periarticular heterotopic ossification (HO) can be observed in patients with traumatic brain injury (TBI), often observed clinically as early as 1 month after the initial injury. The formation of periarticular HO is associated with injury to the central nervous system.1–7 Formation of HO requires inductive signaling pathways, inducible A formal IRB review was not required for this study. *Reprint requests: Usha S. Mani, MD, Methodist Dallas Medical Center, 1441 N. Beckley Ave, Dallas, TX 75203, USA. E-mail address: [email protected] (U.S. Mani).

osteoprogenitor cells, and a heterotopic environment conducive to osteogenesis. HO at the shoulder can produce pain, limitations in motion, and difficulty with activities of daily living. Surgical excision of HO at the elbow, hip, and knee have been shown to improve motion and function; but there is little information available regarding surgical resection of HO at the shoulder.2,3,6,8-11 To our knowledge, there are no studies present to date regarding management of shoulder HO in patients with TBI. We questioned whether elective surgical resection of extensive motion-limiting shoulder HO in TBI patients is

1058-2746/$ - see front matter Ó 2013 Journal of Shoulder and Elbow Surgery Board of Trustees. doi:10.1016/j.jse.2012.01.028

None

216 mos

Materials and methods

M, male; F, female; L, left; R, right; XRT, radiation.

3 R, anterior and posterior 28,m 10 (CB)

IV

28,m 10 (CB)

IV

38/f 33/f 39/f 20,f 28,m (RS) (DW) (PF) (RA) (LS) 5 6 7 8 9

I III II 1 I

45/m 21/m 3 (CW) 4 (AV)

III II

effective in increasing active range of motion. Additionally, we wanted to know if surgery improved function and what complications may have occurred with surgery including recurrence of the heterotopic bone. A series of TBI patients with extensive formation of HO of the shoulder, who exhibited severe limitation of shoulder motion and significant functional disability with loss of autonomy in feeding, grooming, and personal hygiene, is reviewed.

XRT and Didronel

Extensive recurrence, osteoarthrosis Extensive recurrence, avascular necrosis XRT and Didronel 3

None

190 mos

No No No No Moderate recurrence XRT XRT and Didronel XRT Didronel and Indocin XRT 2 2 3 2 2

L Humerus fx R Elbow fx L Scapula fx L Humerus fx R Acromion fx

9 months 37 mos 8 mos 8 mos 8 mos

No AC Joint separation XRT XRT 9 mos 10 mos L Floating Elbow L Scapula fx 2 2

No No XRT XRT 9 mos 8 mos R Floating Shoulder L Coronoid fx 2 1

R, axillary, subacromial L, anterior, subacromial, coracoacromial L, posterior, subacroial L, axillary, subacromial, infraclavicular L, anterior R, posterior L, posterior L, posterior R, axillary, infraclavicular, anterior L, anterior and posterior 41/m 40/m 1 (FD) 2 (DD)

I II

Shoulder and HO location Age/ gender

Garland classification

53

Patient number

Table I

Demographic data and complications

Kjaersgard-Anderson classification

Injury

Timing from DOI to DOS

HO prophylaxis

Complications

Surgical and clinical results following resection

We retrospectively reviewed 10 consecutive TBI patients from 1988 to 2004 who had undergone resection of HO for restrictive shoulder heterotopic ossification (Table I). These 10 patients all presented to our clinic requesting mobilization of the shoulder. Preoperative complaints routinely included pain, stiffness, and limitation of activities of daily living. We felt surgery to be indicated if the above complaints existed, the patient had recovered adequately from the initial trauma to tolerate additional surgery, the patient had already undergone a rehabilitation program, and the patient could be compliant with a new post operative rehabilitation program. Range of motion (ROM) and data collection regarding feeding, grooming, and toiletry were carried out by the attending physicians at the follow-up. Average follow-up was 46.5 months (range, 8-216). Patients were graded by the Garland Classification system, which predicts the risk of recurrence of HO in patients with traumatic brain injury.8 A higher Garland score indicated greater neurologic and physical impairment. Shoulder motion measurements were done using a goniometer with the patient awake and standing (Table II). The total arc of active motion in degrees in each anatomic plane was recorded. The 3 planes of motion included sagittal, coronal and axial. These 3 planes of motion correspond to flexion/extension, abduction/adduction, and internal/external rotation respectively. Rotation was recorded with the arm abducted 90 at the side if possible. The pre- and postoperative motions in each plane were compared using a paired t test for statistical analysis. Three basic daily functions important to our patients, self feeding, hygiene, and toiletry were evaluated pre- and postoperatively (Table III). Limitations in shoulder motion impaired these activities as the patients needed to be able to touch their mouths, head, and perineum, respectively, to perform these activities. Patients were given 1 point for each of these activities (minimum 0, maximum 3) and compared from pre- to postoperative using a paired t test again. Radiographs and computer tomography (CT) scans were used to determine the location and maturity of the HO for all patients. The CT scans enabled clarification of the location of the HO. Typically, the HO was located in the motion interfaces, ligaments, or joint capsules but not typically in the muscle itself. No bone scans were used. Resection was carried out only on radiographically mature HO with well defined cortical edges to the bone. The location of the heterotopic ossification is described as being either anterior, posterior, axillary, subacromial, or infraclavicular. The extent of HO formation was judged using the 3 point radiographic scale as described by Kjaersgard-Anderson with 1 representing <50% loss of joint space, 2 representing >50 % loss of joint space, and 3 representing complete bridging of bone across the joint space.12

54

D.A. Fuller et al.

Table II

Range of motion

Patient number

Duration of follow-up

Arc ext-flex pre-op

Arc ext-flex post-op

Arc abd-add pre-op

Arc abd-add post-op

Arc internal-external rotation pre-op

Arc internal-external rotation post-op

1 2 3 4 5 6 7 8 9 10a 10b

7 18 8 6 3 16 6 2 22 28 60

30 90 15 50 70 0 30 90 0 0 0

220 150 110 160 90 70 120 130 100 70 90

30 60 20 30 60 30 30 90 0 0 0

120 90 60 100 90 90 120 120 50 70 90

0 5 15 0 0 0 0 20 0 0 0

115 120 50 95 15 60 90 45 85 45 55

flex, flexion; ext, extension; abd, abduction; add, adduction; pre-op, preoperative; post-op, postoperative.

Table III

Function

Patient number

Pre-op ability to reach head, mouth, perineum

Pre-op functional score

Post-op ability to reach head, mouth, perineum

Post-op functional score

1 2 3 4 5 6 7 8 9 10

N,N,N N,Y,Y N,N,N N,N,N N,N,N N,N,N N,N,N Y,Y,Y Y,Y,N N,N,N

0 2 0 0 0 0 0 3 2 0

Y,Y,Y Y,Y,Y Y,Y,Y Y,Y,Y Y,Y,Y Y,Y,N Y,Y,Y Y,Y,Y Y,Y,Y Y,Y,N

3 3 3 3 3 2 3 3 2 2

Preop, preoperative; Post-op, postoperative. Functional score (0-3). Ability to touch head (1 point). Ability to touch mouth (1 point). Ability to touch perineum (1 point).

Operative technique The location of the HO dictated the surgical approach and patient positioning. For patients with anterior, infraclavicular or axillary HO, the patient was typically positioned supine and an anterior approach was utilized. If the HO was in the axillary recess, then the brachial plexus was identified and mobilized to protect it from iatrogenic injury during the resection of the HO. The shoulder itself was approached through the deltopectoral interval, which allowed access to the subacromial space and the anterior capsule. For HO that was more proximal in the infraclavicular region, the skin incision was extended medially along the inferior border of the clavicle. The medial extension of the surgical approach allows exposure and protection of the brachial plexus proximally when necessary. For HO in the axillary region, the medial extension of the wound also allowed access to the plexus medial and inferior to the coracoid which is necessary to identify the axillary nerve. The HO was often found within the ligaments (coracoclavicular) and joint capsule. For posterior HO, the patient was typically positioned in the lateral decubitus position and a transverse

or angled incision was made over the posterior shoulder. If a combined approach was necessary, then the lateral decubitus was also chosen. Suction drains were used in all patients. Postoperative prophylaxis against recurrence of the HO was individualized with the majority of patients receiving radiation (700 rad) on the first postoperative day. The zone of the irradiation field was based on the location of the HO preoperatively.All patients began immediate ROM therapy after surgery within the limitations of their pain. One patient (patient 3) had concomitant resection of posteromedial elbow HO at the same time as the shoulder surgery.

Results The ROM improved (P < .001) in all 3 planes of motion Figure. Sagittal plane motion (flexion/extension) improved the most by 85.0 . Coronal plane motion (abduction/ adduction) improved by 59.1 and axial plan motion (internal/external rotation) improved by 66.9 . The patients on average gained 2.1 activities (P < .01) of the 3 tested activities. No patient declined functionally as a result of

Surgical and clinical results following resection

Figure

55

The mean changes in arc of shoulder motion measured in patients pre-(preop) and postoperative (PO).

surgery and 8 of the 10 patients could perform all 3 of the tested activities at the end of the study. Prior to the surgery, only 1 of the 10 patients could perform all 3 activities. Three of the 11 shoulders had recurrence of heterotopic ossification. Both shoulders in 1 patient had severe recurrence of the HO, a second patient had moderate recurrence, and 8 shoulders showed no recurrence. The one patient with bilateral severe recurrence also developed severe osteoarthrosis in one shoulder and avascular necrosis in the contralateral shoulder joint after the HO resection. This patient had a greater predisposition to form HO given his multiple joint involvement. The AVN required a hemiarthroplasty for pain relief. The one patient that had moderate recurrence experienced severe postoperative swelling in the operative arm. Evaluation for infection and deep venous thrombosis were negative in this patient. For all 3 of the shoulders with recurrence, more than 50% of the joint was lost. This correlated to a K-A Class 2. One patient had instability of the distal clavicle and had a high grade acromioclavicular separation after resection of infraclavicular HO. This patient was pain free and satisfied with his improved motion and function. No treatment was necessary for the dislocation of the distal clavicle as it was asymptomatic. No infections, fractures, or neurovascular complications occurred.

Discussion There are few or no publications available regarding the management of restrictive shoulder HO in patients with TBI. We found that the surgical resection of shoulder HO can improve shoulder ROM and function of the limb. Complications can be severe such as recurrence. All 3 shoulders that exhibited recurrences received postoperative radiation. Ten of the 11 shoulders in this series received radiation postoperatively as prophylaxis against HO recurrence. Based on this small series, it is difficult to make

recommendations regarding postoperative radiation. Had radiation not been used, it is possible that the recurrence rate may have been even higher. We speculate that recurrence is related to unique genetic factors regulating bone formation. This group of 10 patients demonstrated a predisposition to form ectopic bone. In the 2 patients with recurrence, a stronger gene may be present. Associated local trauma may have contributed to the original formation of HO in the shoulder at the time the head injury occurred. With surgical resection, no additional trauma was incurred other than our surgical manipulation. Indomethicin was not used in this series because of its limited efficacy in preventing recurrence and poor compliance with its use. Technically, the procedure is demanding and despite the lack of neurologic injury in this small series, we believe that the risk of neurologic injury can be substantial, particularly when the HO is in the axillary recess. We did not use pre-, post-, or intraoperative electrodiagnostic studies to assess for occult neurologic compromise. In our patients, the goals were related to achieving improvements in gross joint movement. We felt that physical examination and recording functional activities were adequate for evaluation for nerve function. Subtle nerve dysfunction may have existed either pre- or postoperatively in our patients that we were unable to detect clinically. A weakness to this study is that it is small and retrospective. Based on our review of the literature and our clinical and published experience with HO in TBI, we believe that shoulder HO is much less common than the hip, knee, or the elbow.1,2,6,11 All shoulder HO does not require resection, and this series represents patients that have self-selected themselves for surgical evaluation and treatment. There are certainly many cases of shoulder HO that are asymptomatic or cases in which the surgical risk/ benefit ratio does not favor surgery for the individual patient. Criteria for surgical operation for this problem is not well defined. Reaching one’s mouth for feeding requires shoulder forward flexion. Reaching the top of ’one’s head requires some more. Reaching the perineum requires some shoulder extension and internal rotation. Compensatory

56 cervical and other joint movement make it impossible to place absolute numbers on desired ROM goals. Our patients had profound limitations in motion and function, making even small gains significant. Whether patients with more subtle deficiencies would benefit from this operation is unknown. Perhaps even mild cases of HO can lead to substantial symptoms, particularly in patients without brain injury. We have no knowledge of the incidence of asymptomatic shoulder HO in patients with and without head injury. Without symptoms, we would discourage surgery even if the HO is found incidentally on radiographic studies. That this is a retrospective series does not negate the significant improvement in motion or function for the symptomatic patients.

Conclusion Surgical excision of shoulder HO in patients with TBI can produce significant gains in shoulder ROM. Improvement in function is also observed after resection of shoulder HO with an acceptable complication rate.

Disclaimer The authors, their immediate families, and any research foundations with which they are affiliated have not received any financial payments or other benefits from any commercial entity related to the subject of this article.

D.A. Fuller et al.

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