Case report 549

How does rectus femoris fibrosis affect gait? Subash Balakrishnan, Leanne N. Purcell, Anita J. Mudge, Elizabeth A. Wojciechowski, Joshua Burns and Matthias W. Axt We report on a female patient with stiff-knee gait resulting from rectus femoris fibrosis, following multiple injections into the quadriceps musculature. Treatment planning and outcome were aided by instrumented three-dimensional gait analysis. One year after surgery, improvements were found in walking speed, step length, anterior pelvic tilt, knee flexion in swing, and ankle dorsiflexion in swing. Instrumented three-dimensional gait analysis was a useful tool for characterizing gait impairments and detecting changes after surgical intervention. Intramuscular injections into the rectus femoris muscle should be avoided to prevent stiff-knee gait and associated disability. Level of evidence:

Introduction Limping, walking with stiff knees, and restricted knee flexion after multiple injections of antibiotics into the quadriceps muscle in infants and young children have been reported over the past 50 years, predominantly in Asian and African countries [1–4]. Common sites for intramuscular injections are the gluteus medius, deltoid, vastus lateralis, and rectus femoris muscles. Although reports of impaired walking exist as a result of thigh muscle injections, these sites are still recommended in current practice guidelines [5–7]. Assessment of stiff-knee gait, a condition in which swingphase knee flexion is significantly reduced [8], and subsequent treatment planning can be difficult. This can be overcome by instrumented three-dimensional gait analysis (3DGA), which helps establish a treatment plan and monitor outcomes [9–11]. We report on a female patient with injection-induced fibrosis of the rectus femoris muscle in both legs. She was treated successfully and her progress was monitored by 3DGA.

Case report The mother of a 5-year-old girl was concerned by her daughter’s stiff knees and increased lumbar lordosis while walking. The family emigrated from Africa to Australia when the child was 3½ years old. The girl’s developmental milestones were normal, with no family history of muscular or neurological disorders. At 12 months of age, the girl was treated for pneumonia with benzyl-penicillin injections into her thigh muscles, with no adverse reactions noted at the time. Her parents observed abnormalities in her gait at 2 years of age. The 1060-152X © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins

Level IV, case report. J Pediatr Orthop B 23:549–553 © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins. Journal of Pediatric Orthopaedics B 2014, 23:549–553 Keywords: fibrosis, gait analysis, intramuscular injection, rectus femoris, stiff-knee gait Orthopaedic Department and Paediatric Gait Analysis Service of New South Wales, The Children’s Hospital at Westmead, Westmead, New South Wales, Australia Correspondence to Matthias W. Axt, MD, Orthopaedic Department, The Children’s Hospital at Westmead, Locked Bag 4001, Westmead, NSW 2145, Australia Tel: +61 2 9845 3020; fax: +61 2 9845 3180; e-mail: [email protected]

abnormalities became so pronounced with growth that a neurological disorder was considered. Preoperative assessment

Initial physical examination in clinic showed a stiff-knee gait in a 5-year-old girl of normal weight and stature, with compensatory circumduction of the swinging leg, and short rectus femoris muscles. In sitting, knee flexion was only achievable by tilting the pelvis forward. Skin dimples at the front and the lateral aspect of the thigh indicated a history of intramuscular injections of antibiotics in early childhood, confirmed by the parents. The patient underwent instrumented 3DGA using a Vicon MX system (Vicon Motion Systems Ltd., Oxford, UK) with eight infrared video cameras and three AMTI force plates (Advanced Mechanical Technology Inc., Watertown, Massachusetts, USA) in our gait laboratory. Multiple walks were collected and analyzed before and after surgery (six and seven walks, respectively). Temporo-spatial parameters (walking speed, step length, and cadence), kinematic key event data and kinematic average traces over the gait cycle were used to aid clinical decision making and outcome measurement. Gait analysis assessment also included a comprehensive lower limb standardized physical examination [12] and 2D video analysis. Informed consent for case report publication was obtained from the child’s family. Preoperative report

Gait by observation showed a stiff-knee gait, with the left side worse than the right, increased anterior pelvic tilt, increased lumbar lordosis and flexion of the hips and knees throughout the stance phase. The abnormalities DOI: 10.1097/BPB.0000000000000078

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550 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6

Fig. 1

MRI of both femora in the transverse plane, proximal level, T1 weighted image: the large white circled area on the left side shows fibrosis, respectively, scarring, of the rectus femoris muscle, adjacent to and involving vastus lateralis. The small white circled area on the right side shows fibrosis within the rectus femoris. Note the atrophy of rectus femoris muscle on the left side compared with the right as well as the reduced muscle bulk of the left thigh. MRI, magnetic resonance imaging.

were compensated for by mild circumduction of both legs and increased ankle dorsiflexion during swing. Lying prone with the hip extended, knee flexion was 20° on the left and 55° on the right before the pelvis started to rise. Maximum passive knee flexion (with the hips flexed), however, was only mildly limited, 135° on the left and 132° on the right. These findings coincided with more marked fibrosis of rectus femoris and vastus lateralis musculature on the left side, and atrophy of the left rectus femoris muscle in the magnetic resonance imaging (Fig. 1). The difference in maximum knee flexion could be attributed to measurement variability. The mildly limited knee flexion on either side may result from some fibrosis of the vastus lateralis muscles, the targeted injection sites. In healthy children, passive knee flexion of 149 ± 4° has been reported [13]. Kinematic data in the sagittal plane (Table 1, Fig. 2) showed a marked increase in anterior pelvic tilt of 35° Table 1

(compared with normal 9°), related to shortening of the rectus femoris muscle. Hip kinematics followed a normal pattern with a shift into increased hip flexion of ∼ 30° over the gait cycle, compared with our normative data (n = 50, age 4–16 years). Knee kinematics showed incomplete knee extension in mid to terminal stance and reduced peak knee flexion during swing, worse on the left side (39 ± 17° left, 54 ± 6° right, compared with normal values of 65 ± 8°). The knee range of motion during the gait cycle was approaching half the normal range, confirming stiff-knee gait. Ankle kinematics showed increased dorsiflexion during swing as a compensation to help clear the foot. In the coronal plane, there were signs of circumduction on the left side and in the transverse plane, pelvic rotation was excessive. Treatment

Surgery consisted of bilateral release of the rectus femoris origin and insertion, followed by immediate mobilization and physiotherapy, including continuous passive motion,

Key kinematic data in degrees of the left and right lower limbs before and after surgery Left

Key event Mean pelvic tilt Peak knee flexion in swing Minimum hip flexion Maximum hip flexion in swing Peak knee extension in stance Peak plantar flexion in swing Peak dorsiflexion in swing

Right

Before surgery

After surgery

Before surgery

After surgery

Norms (n = 50)

35.3 ± 3 39.0 ± 17 19.3 ± 7 63.3 ± 6 15.1 ± 4 − 0.1 ± 4 13.4 ± 4

26.7 ± 2 54.4 ± 6 19.3 ± 3 57.4 ± 3 15.9 ± 2 − 8.2 ± 4 6.2 ± 3

35.3 ± 3 50.3 ± 6 21.9 ± 2 62.7 ± 6 15.0 ± 4 − 3.9 ± 3 11.6 ± 2

26.7 ± 2 56.5 ± 4 14.7 ± 3 55.6 ± 3 16.8 ± 4 − 5.7 ± 4 6.7 ± 1

8.8 ± 4 65.4 ± 8 − 8.9 ± 5 35.9 ± 7 8.1 ± 6 − 15.5 ± 7 5.4 ± 3

Values are represented as mean ± SD. The SD values represent the within-patient variation over multiple trials.

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Rectus femoris fibrosis affects gait Balakrishnan et al. 551

Fig. 2 Pelvic tilt (deg.)

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Sagittal plane pelvis, hips, knees and ankles, transverse plane pelvis, and coronal plane hip kinematics of one gait cycle before and after surgery. (a) shows the left side and (b) shows the right side. The solid lines show the presurgical average and ± 1 SD. The dark gray shaded band shows the postoperative average and ± 1 SD. The light gray shaded band represents the gait lab pediatric normative reference data (n =50). Interpretation: The exaggerated anterior pelvic tilt and thus the increased lumbar lordosis have improved after surgery. Hip extension has benefited from the altered pelvic tilt. Peak knee flexion in swing has increased postoperatively. Ankle dorsiflexion during swing has normalized.

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552 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6

to maintain intraoperatively achieved range of motion at the hips and knees. There was no need to lengthen vastus lateralis muscle on either side because maximum knee flexion was sufficient for daily activities, including sport. On the contrary, lengthening of vastus lateralis may have weakened quadriceps function. Postoperative assessment

One year after surgery, physical examination indicated that the patient was able to flex the left knee to 40° (20° improvement) and the right to 90° (35° improvement) in prone with the pelvis flat on the table. The knee flexion remained unchanged with hips flexed. 2D gait video showed less lumbar lordosis than was present preoperatively. Kinematic data showed an improved anterior pelvic tilt by 9° in the sagittal plane, with subsequently improved hip extension in stance on the right side. The average peak knee flexion improved 15° on the left and 6° on the right in mid swing. Left ankle plantar flexion at push-off improved towards normative values. Increased compensatory ankle dorsiflexion in swing also normalized (13–6° on the left, 11–6° on the right). Presurgical and postsurgical temporal spatial data (Table 2) were normalized to leg length [14], to account for the patient’s leg length growth over time. The results are expressed as a percentage of our gait laboratory normative dataset. Cadence, walking speed, and step length increased after surgery.

Discussion We carried out instrumented gait analysis to quantify the problem of rectus femoris fibrosis after an intramuscular injection of antibiotics and used it as a clinical outcome measure following surgery. The previous authors solely relied on clinical observation. Mukherjee and Das [4] used knee range of motion to verify the results. They measured the arc of active and passive flexion/extension of the knee as well as the active extension lag and used this to grade their results as ‘good’, ‘fair’, or ‘poor’. Gbenou et al. [15] used knee flexion gain and quality of walking as a measure of treatment outcome. Results were rated ‘good’ if flexion gained was above 90°, allowing normal gait and squatting, which is often essential for

Table 2

Temporal spatial data before and after surgery % of norms 4–16 years

Cadence Walking speed Step length (left) Step length (right)

Before surgery

After surgery

Percentage change

95 96 96 101

107 107 100 111

12 11 4 10

toileting in African and Asian countries. Results were considered ‘acceptable’ if flexion gained was between 45° and 90°, normal gait was observed and a comfortable sitting position was achieved. Results were considered ‘poor’ if flexion gained was less than 45°, associated with limping and uncomfortable sitting. Akyüz et al. [16] used sonography to diagnose and follow a case of rectus femoris fibrosis treated with physiotherapy. In general, authors recognized limping related to stiff-knee gait, which is difficult to quantify without instrumented gait analysis. They also acknowledged squatting problems and used knee range of motion as their primary outcome measure. Gait disturbances are arguably more disabling than limited knee range of motion and can best be quantified by instrumented gait analysis [10,11]. It provides evidence of rectus femoris malfunctioning, which has a more adverse effect on gait than on limited range of motion of the knee. Rectus femoris is a two-jointed muscle that acts both as a flexor at the hip and as an extensor at the knee. It acts during rapid gait in preswing and initial swing [17]. During the gait cycle, the muscle transfers energy from the shank to the hip by flexing the hip while allowing controlled knee flexion through eccentric and isometric action. In our patient, the rectus femoris muscle was adherent to the surrounding quadriceps muscles, acting as a tether, preventing simultaneous hip extension and knee flexion required in the early swing phase. Vastus lateralis involvement was minimal as maximum knee flexion (with the hip flexed) was only marginally reduced. Various treatment modalities have been recommended for rectus femoris fibrosis to counteract stiff-knee gait. In advanced fibrosis, surgery has been the treatment of choice. Sengupta [18] proposed proximal release only in early stages of fibrosis. Mukherjee and Das [4] performed a z-plasty or a v-y-plasty of the quadriceps initially and, depending on intraoperative findings, divided rectus femoris distally and reattached it more proximally if adequate knee flexion could not be achieved. Surgical releases of rectus femoris were performed to maximize the function of the remaining quadriceps muscles. Instrumented gait analysis improved our understanding of the underlying pathology and the effect of treatment on our patient. We addressed the pathology surgically by a proximal and distal release of the rectus femoris muscle. Although separation of this muscle from adjacent muscles and full quadriceps function restoration was impossible to achieve, peak knee flexion and stiff-knee gait, as documented by instrumented gait analysis, improved markedly. To avoid rectus fibrosis and subsequent gait problems, we recommend that the rectus femoris be spared as an injection site, especially in young children.

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Rectus femoris fibrosis affects gait Balakrishnan et al. 553

Acknowledgements

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Conflicts of interest

There are no conflicts of interest.

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References

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How does rectus femoris fibrosis affect gait?

We report on a female patient with stiff-knee gait resulting from rectus femoris fibrosis, following multiple injections into the quadriceps musculatu...
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