Integrated Physical Therapy Intervention for a Person With Pectus Excavatum and Bilateral Shoulder Pain: A Single-Case Study

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ORIGINAL ARTICLE

Integrated Physical Therapy Intervention for a Person With Pectus Excavatum and Bilateral Shoulder Pain: A Single-Case Study Paul K. Canavan, PhD, PT, Larry Cahalin, MS, PT ABSTRACT. Canavan PK, Cahalin L. Integrated physical therapy intervention for a person with pectus excavatum and bilateral shoulder pain: a single-case study. Arch Phys Med Rehabil 2008;89:2195-204. Objective: To examine the effects of an individualized physical therapy (PT) program for a subject with pectus excavatum and bilateral shoulder pain. Design: Single-case study of a man diagnosed with moderate-to-severe pectus excavatum and constant bilateral shoulder pain. Exercise tolerance was measured through the Bruce protocol and home exercise log, pulmonary function, ventilatory muscle strength, echocardiography, chest wall and abdominal excursion, self-perception of pectus excavatum, and a variety of anthropometric and volumetric tests before and after PT. Setting: University laboratory. Participant: A 22-year-old man. Intervention: A 3-month PT program including breathing exercises and therapeutic exercises. Main Outcome Measures: Exercise tolerance, ventilatory muscle strength, chest wall and abdominal excursion, selfperception of the pectus excavatum, and other anthropometric and volumetric tests. Results: The most striking anthropometric and volumetric test change was the pectus severity index (in H2O), which decreased from 50 to 20mL H2O (60% change). The subject reported no shoulder pain at rest and with recreational activity after 8 weeks of intervention. Conclusion: An individualized PT program provided minimal-to-moderate improvements on many characteristics of pectus excavatum. Bilateral shoulder pain was eliminated. An individualized PT program integrating cardiopulmonary and musculoskeletal interventions that is provided to other patients with pectus excavatum may provide similar results. However, PT provided to younger patients with the pectus excavatum may be of even greater benefit because of a less mature skeleton. Further investigation of the effects of PT intervention provided to younger and older persons with the pectus excavatum is needed. Key Words: Exercise tolerance; Rehabilitation; Shoulder pain. © 2008 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation

From the Department of Physical Therapy, Northeastern University, Boston, MA. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit on the authors or on any organization with which the authors are associated. Reprint requests to Paul K. Canavan, PhD, PT, Dept of Physical Therapy, Northeastern University, 6 Robinson Hall, Boston, MA 02115, e-mail: p.canavan@neu. edu. 0003-9993/08/8911-00279$34.00/0 doi:10.1016/j.apmr.2008.04.014

ECTUS EXCAVATUM IS ONE of the most common anomalies of childhood. It occurs in approximately 1 in P every 400 births. Pectus excavatum has also been described as 1

funnel chest. Pectus excavatum is a depression of the sternum and the center of the rib cage causing an inward deformity. Pectus excavatum has been associated with true physiologic impairment and reduced exercise capacity, predominately because of impaired cardiovascular performance rather than ventilatory limitation.2 The cardiovascular impairments are primarily caused by constrained diastolic filling from the pectus excavatum. The severity of pectus excavatum is often determined with the PSI, which is quotient of the transverse width of the chest by the vertebral-sternal distance. A PSI greater than 4 is indicative of severe pectus excavatum, which may significantly limit filling within the cardiac chambers because of skeletal compression of the pectus excavatum. Idiopathic pectus excavatum may be associated with a variety of lung function abnormalities such as lower airway obstruction and reduced lung capacities.3 Symptoms of pectus excavatum have been described to include easy fatigability, dyspnea with mild exertion, pain in the anterior chest, and tachycardia becoming increasingly severe during the adolescent years and remaining throughout adult life.1 However, there has been no evidence of significant worsening of lung function with development from childhood to early adulthood in persons with pectus excavatum (5–19y).3 Persons with pectus excavatum may also experience deficits with upper-extremity strength and psychologic issues such as self esteem and self-efficacy. It has been previously reported that PT for patients with pectus excavatum without postural impairments is not necessary with the exception of postoperative pulmonary care.4 Many people with pectus excavatum exhibit postural deviations as well as other orthopedic-related complaints such as shoulder pain. Rehabilitation for people with pectus excavatum should incorporate the integration of cardiopulmonary and orthopedic PT because a significant negative correlation between thoracic kyphosis angle and inspiratory capacity, vital capacity, and lateral expansion of the thorax has been shown to exist.5 Furthermore, postural positioning can

List of Abbreviations FEV1 FVC MEP MIP MMT PSI PT QOL ROM RPE VAS

forced expiratory volume in 1 second forced vital capacity maximal expiratory pressure maximal inspiratory pressure manual muscle testing pectus severity index physical therapy quality of life range of motion rating of perceived exertion visual analog scale

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also play a role in airway resistance such that the supine posture may cause upper-airway resistance to increase.6 Surgical repair is common for people with pectus excavatum who have a PSI greater than 4. The 2 most common surgical procedures for pectus excavatum are the Ravitch procedure and the Nuss procedure. Both of these procedures have been shown to have satisfactory results.7 Recently, a new and innovative approach has been used involving the application of a vacuum as an adjunct in surgical and nonsurgical correction of pectus excavatum.8 Although many persons opt for surgical intervention, there are others who elect the nonsurgical approach. Integrated PT intervention of cardiopulmonary and orthopedic PT may help to enhance the QOL of a person with pectus excavatum whether the person elects to have surgery or not. In a search of the literature of the past 20 years using the key words pectus excavatum and physical therapy, we found no case studies related to an integrated approach for people with pectus excavatum. The purpose of this single-case study is to present the integrated approach and intervention for a patient with pectus excavatum and shoulder pain in an outpatient setting. METHODS Participant and History The patient was a 22-year-old white man who was diagnosed with pectus excavatum in his early teens. He was followed serially by several medical doctors through observation, computed tomography scan, and radiography and was recommended to undergo surgical correction of his pectus excavatum. During the previous 2 years, the patient reported that his primary complaints were difficulty playing basketball and running because of dyspnea and fatigue as well as bilateral shoulder pain (right greater than the left). The patient’s secondary complaints were disturbed sleep over the past 6 months caused by bilateral shoulder pain and decreased girth size of his pectoralis major regions. The patient opted to undergo a trial of PT in lieu of surgical correction. His main goals were to (1) avoid surgery for his pectus excavatum, (2) be able to sleep and play basketball without shoulder pain, (3) be able to play basketball without feeling extreme dyspnea and fatigue, and (4) increase his upper-extremity strength and prevent his shoulders from feeling tired. PT Examination Methods Cardiorespiratory tests and measures. The clinician performing the cardiorespiratory tests and measures had over 25 years of clinical research experience and was very familiar and trained in the assessment of persons with cardiovascular and pulmonary disorders. Resting and exertional dyspnea and self-perception of pectus excavatum. Resting and exertional dyspnea were measured with the modified Borg dyspnea scale (0 –10 scale)9 and a VAS (consisting of a 10-cm horizontal line, with no shortness of breath and extreme shortness of breath listed at the left and right ends of the line, respectively).10 The subject’s self-perception of pectus excavatum was obtained through self-report using a scale from 0 (worst self-perception of pectus excavatum) to 10 (best self-perception of pectus excavatum). Pulmonary function tests and echocardiography. Maximally forced expired flow volume loops were measured using a rolling seal spirometer.a FVC and FEV1 were reported from the same loop according to American Thoracic Society guidelines.11 The percentages of maximum age-adjusted and sexadjusted FEV1 and FVC were determined using the tables of Arch Phys Med Rehabil Vol 89, November 2008

Crapo et al12 and Cotes.13 Body plethysmography was performed using standard techniques according to American Thoracic Society guidelines.11 Spirometry was performed before and after PT interventions. Two-dimensional and M-mode echocardiography were performed before PT interventions using standard echocardiographic techniques.14 Ventilatory muscle strength and endurance. Measurement of inspiratory and expiratory pressures were made with the Micro MPM pressure transducerb (accuracy, ⫾.01cm H2O) using the methods of Black and Hyatt.15 The subject was seated with the hip and trunk at a 90° angle and was encouraged to perform the tests with maximal effort. MIP and MEP were obtained after a maximal expiration (near residual volume) and after a maximal inspiration (total lung capacity), respectively. Maintenance of an adequate seal around the mouthpiece was ensured, and tests were repeated until a baseline measurement without further increase was obtained. Inspiratory muscle endurance was measured using the method described by Martyn et al.16 The measurement of inspiratory muscle endurance was performed with a threshold loading device that provided resistance during inspiration. The test began at 20% of MIP, and the patient was asked to breathe with the device for 2 minutes, after which the resistance was progressed by 20% increments (based on the initial MIP) until exhaustion. Oxygen saturation and inspiratory muscle use were monitored throughout the endurance test, and testing was terminated if markedly abnormal oxygen saturation levels (⬍86%) or a paradoxical breathing pattern was observed. Chest wall and abdominal motion. Chest wall and abdominal motion were also measured with the RMMI,c which consisted of 6 lightweight sensors,d a connecting cabinet and cords, and a personal computer with Windows 98 and input devices with specially designed software. The sensors were attached by special clamps to a frame consisting of a larger angled height-adjustable arm and 2 smaller arms mounted on the larger arm. Each small arm bore 3 sensors that were adjusted to measure the motion at 6 different anatomic landmarks. Placement of the ultrasound chest wall and abdominal sensors at the 6 landmarks was performed by drawing a vertical line from the medial one third of the clavicular bone on each side of the thorax to the abdominal wall lateral to the umbilicus. Three landmarks were then marked on each of these vertical lines including 1 at the height of the fourth rib, 1 at the ninth rib, and 1 lateral to the umbilicus. The sensors were then positioned 20cm above these landmarks bilaterally (fig 1), after which the patient rested in the supine position with 1 pillow under his head for 5 minutes. During this time he was instructed to relax and breathe at a comfortable rate with the tidal volume kept constant through respirometer measurements. Ultrasound signals from the 6 sensors were subsequently obtained, digitalized, and relayed to a personal computer into an Excel file for data analysis during both inspiration and exhalation. The precision of the measurements is 0.2mm. The RMMI data were continuously recorded for 60 seconds. Previous measurement of respiratory motion using RMMI as described has demonstrated good reliability with correlation coefficients of .795 and .787 for the abdominal movements, .866 and .798 for the lower thoracic movements, and .879 and .770 for the upper thoracic movements.17 Anthropometric and volumetric measurements. Eight anthropometric measurements and 1 volumetric measurement were performed in the subject. The anthropometric measurements were performed based on previous literature18 and on anticipated thoracic changes using easily identifiable landmarks and a standard tape measure. Anthropometric measure-

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and inclines every 3 minutes.20 The heart rate and rhythm were continuously monitored and recorded every 3 minutes or if abnormalities were observed. The blood pressure was obtained and recorded every minute of exercise. The RPE was obtained at the beginning and end of each stage of exercise. Peak oxygen consumption was estimated based on the Bruce exercise test duration.20

Fig 1. Respiratory motion measurement instrument.

ments were obtained 3 times at each site during tidal volume breathing (normative tidal volume breathing) and during vital capacity breathing (maximal inspiratory and expiratory efforts) to ensure consistency of measurements. An average of the measurements during tidal volume and vital capacity breathing was used as the final anthropometric site measurements. Tape measurements of chest wall and abdominal excursion using these methods have been observed to be reliable with intraclass correlation coefficients between .88 and .98.19 The volumetric measure was performed to measure indirectly the severity of the pectus excavatum because magnetic resonance imaging was not performed, which prevented the measurement of the PSI.2 The volumetric measure was obtained with the subject lying supine for 5 minutes and breathing at a constant tidal volume and respiratory rate (13 breaths/min) while slowly pouring water from a prefilled graduated cylinder until any amount of water began overflowing from the pectus excavatum. The amount of water remaining in the cylinder was subtracted from the initial water level of the cylinder, and the difference determined the PSI (in H2O). The severity of pectus excavatum was also measured with a 30.5-cm (12-in) wood ruler and tape measure in the supine position by placing 1 end of the ruler into the pectus excavatum at the level of the nipples and measuring the depth of the pectus excavatum on the ruler from the tape measure placed circumferentially around the thorax at the nipple level. Treadmill exercise testing. Maximal symptom-limited treadmill exercise testing was performed with a calibrated treadmill using the standard Bruce protocol beginning at 2.7kph (1.7mph), 10% grade, and progressing to greater speeds

Orthopedic Tests and Measures The physical therapist performing the orthopedic tests and measures had over 18 years of clinical experience and was very familiar and trained in the assessment of persons with orthopedic problems. Standing resting posture and forward flexion. The patient stood in a normative self-selected resting posture with hands to the side. The position of the feet was reported for replication, and the patient was instructed to gaze straight forward at a standardized height. The patient was also instructed to bend forward, during which time the back was visualized and a photograph was taken (fig 2). The patient complained of moderate bilateral shoulder pain (VAS score, 5 out of 10) during sleeping and with active left and right external shoulder external rotation with the shoulder held in abduction at 90°. Scapula postural assessment. The patient remained in the resting standing posture, and the scapula measurement instrument was used to document resting scapula position with the left scapula as the reference scapula (fig 3). The scapula measurement instrument has been previously shown to be reliable and valid in the resting posture position for measuring scapula position in the frontal (mediolateral positions) and sagittal (anteroposterior position) planes.21,22 The root of the spine of the scapula was identified and marked with a pen on both the left and right scapulas. The adjacent spinous process to the root of the spine of the left scapula was palpated and marked with a pen. This spinous process was also used for the reference point of the left and the right scapula. The scapula measurement instrument was leveled in the frontal, transverse, and sagittal planes to maintain proper standardization. The device was measured in increments of millimeters and centimeters. The left scapula resting position, medial to lateral position, was measured from the adjacent spinous process to the root of the spine of the scapula. The right scapula was measured from the reference spinous process horizontally only. The anterior to posterior position was also measured using the same spinous process reference point and while maintaining the scapula measurement instrument level.

Fig 2. Forward bending.

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xiphoid process and lower anterior chest upward into the therapist’s hand, the pectus excavatum was judged to be fixed. If the patient moved the xiphoid process and lower anterior chest upward minimally (⬍2cm), the pectus excavatum was judged to be semifixed. If the patient moved the xiphoid process and anterior chest upward freely (⬎2cm), the pectus excavatum was judged to be nonfixed. The other method consisted of visual examination from a posterior view for changes in the rib hump as well as changes in the thoracic spine curve, while the patient performed active forward trunk flexion from a standing position (see fig 2).

Fig 3. Scapula measurement instrument.

The most palpable portion of the inferior angle was identified, and a pen mark was placed 1cm medial to this point. The adjacent palpable spinous process adjacent to the pen mark about the left scapula was identified, and a pen mark was placed in that point. The anterior to posterior position was also measured using the same spinous process reference point and while maintaining the scapula measurement instrument level. The anterior-to-posterior position determined the amount of winging. Measurements were taken preintervention and 8 weeks after the intervention. Leg length. The patient was placed supine, and the patient’s legs were placed in the standardized manner described by Palmer and Epler.23 The most palpable portion of the anterior superior iliac spine and medial malleoli was palpated, and a standard tape measure was used. Manual muscle testing. Standardized MMT was performed as reported by Palmer and Epler.23 Shoulder flexion, abduction, and internal and external rotation were tested in the seated position. Shoulder flexion, horizontal abduction, lower trapezius, and scapula retraction were tested in prone, and scapula protraction was tested in supine. The MMT was tested before and after 8 weeks of intervention. Fixed versus nonfixed pectus deformity. The status of the pectus deformity (fixed vs nonfixed) was examined using 2 different techniques. The patient was placed supine, and the therapist’s hand was lightly placed slightly on the patient’s xiphoid process. The patient was instructed to inspire deeply and attempt to push the xiphoid process and anterior chest into the therapist’s hand. If the patient was unable to move the Arch Phys Med Rehabil Vol 89, November 2008

PT Interventions Exercise prescription and exercise adherence. The exercise prescription is outlined in appendix 1. Based on the examination findings, an integrated PT intervention was developed and implemented by a cardiopulmonary physical therapist and an orthopedic physical therapist. The integrated intervention consisted of breathing exercises, aerobic exercise training, and musculoskeletal stretching and strengthening exercises. The breathing exercises and aerobic exercises were prescribed to be performed 3 to 5 times a week with a progression in exercise duration based on symptoms and the Borg RPE.9 Initially, 5 to 10 minutes of breathing exercises and 5 to 10 minutes of aerobic exercises were prescribed with a goal of 20 and 30 minutes, respectively. The breathing exercises consisted of diaphragmatic breathing exercises and lateral costal breathing exercises with and without manual facilitation using assisted and independent quick stretches to the abdominal and lateral costal areas. Gentle breath-holding was also performed during stretches and exercises that promoted greater chest expansion with less pectus excavatum. Additionally, many of the stretching and strengthening exercises were performed on all fours (fig 4) in anticipation that the effects of gravity would promote greater chest expansion and less pectus excavatum. The patient was instructed to perform 10 to 12 repetitions of the breathing exercises 2 to 3 times a day with independent manual facilitation of the breathing exercises (placing both hands on his lower rib cage at a midpoint between the umbilicus and the xiphoid process along the lateral costal region to promote lower rib cage expansion during inhalation and pursed lip breathing on exhalation) during at least 1 of the sessions. Aerobic exercise training was provided using American College of Sports Medicine guidelines24 at an intensity of 70% to 80% of the peak heart rate obtained during treadmill exercise testing. The prescribed stretching and strengthening program is outlined in appendix 1 and was based on the findings of the physical examination along with the (1) the origin and insertion of skeletal muscles, (2) angle of pennatation of muscular fibers, (3) function of the muscles, and (4) biomechanics of breathing. Several of the key exercises are shown in figure 4 and were prescribed in a progressive manner as described in appendix 1. The patient was initially seen by the cardiopulmonary physical therapist for 8 weeks (4 visits) and was seen for an additional 8 weeks (4 visits) for combined cardiopulmonary and orthopedic PT. The breathing exercises, aerobic exercises, and therapeutic resistance program were progressed during each visit. The patient was educated on proper progression with regard to sets and repetitions as well as exercise order. The adherence to exercise was performed using an exercise log in which the subject was instructed to record the mode, duration, and intensity of his daily exercises as well as the occurrence of any adverse events or problems. The exercise log was also used to document the subject’s exercise program, which was updated every 2 weeks.

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Fig 4. (A–D) Several therapeutic exercises.

RESULTS Orthopedic and Musculoskeletal Standing resting posture and forward flexion. The right iliac crest was slightly superior to the left, and the right scapula was slightly inferior to the left. This was determined by palpation and visual inspection at the level of the ilium. Radiographs were not taken. PT exhibited a minor left thoracic C-curve scoliosis. The right shoulder was visibly anterior relative to the left shoulder, right greater than left. Forward trunk flexion demonstrated possible evidence of a semifixed scoliosis as the thoracic spine straightened a small amount (see fig 2). The patient reported no shoulder pain at rest, while sleeping, or during active left and right shoulder external rotation with the shoulder at 90° shoulder abduction. Scapula postural assessment. The resting scapula position measurements are listed in table 1. The right scapula was initially found to be in a resting position 2.5cm inferior to the left. The right scapula winging decreased by 15% and medialized toward the spine by 9%. The left scapula medialized by 8% with no change in anteroposterior (winging) position. Leg length. The left and right legs were measured to be 93 and 94cm, respectively. Manual muscle testing. The MMT results are shown in table 2. On average, the subject’s manual muscle testing grade

improved by one half grade. The subject reported that his shoulders and arms had no pain and did not feel tired during and/or after participation in activities such as basketball. Exercise adherence. Adherence to exercise was reported to be 100%. Cardiorespiratory Resting and exertional dyspnea and self-perception of pectus excavatum. Resting and exertional dyspnea as well as self-perception of pectus excavatum were observed to improve throughout the treatment period, with a 25% improvement in both resting and exertional dyspnea and a self-perception score of pectus excavatum from 3 to 7.

Table 1: Resting Scapula Position Measurements Spine of the Scapula

Left Pre

Left Post

Right Pre

Right Post

Medial to lateral Anterior to posterior Inferior angle Medial to lateral Anterior to posterior

7.5 7.5

7.0 7.5

8.0 7.5

7.0 6.5

8.5 6.5

8.0 6.0

10.5 5.5

10.0 4.5

NOTE. Values are in centimeters.

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BILATERAL SHOULDER PAIN AND PHYSICAL THERAPY, Canavan Table 2: MMT and Bilateral Upper Extremity Movements Tested

Left Pre

Left Post

Right Pre

Shoulder flexion Shoulder abduction Shoulder internal rotation Shoulder external rotation Prone shoulder flexion Horizontal abduction Lower trapezius Scapula retraction Supine scapula protraction

4⫺ 4 4⫹ 4 4⫺ 4 4 5 4⫺

4⫹ 4⫹ 4⫹ 4⫹ 4 4 4⫺ 5 4⫺

4 4⫺ 4⫹ 4⫺ 3⫹ 4⫹ 4⫹ 4⫺ 4⫹

Right Post

4⫹ 4 5 4 4 4⫹ 4 5 4⫹

insufficiency, and mild pulmonary insufficiency. The estimated ejection fraction was 66%. No other echocardiographic abnormalities were observed. Chest wall and abdominal motion. The chest wall and abdominal motion before and after the training period are shown in figure 5. Chest wall and abdominal motion were observed to increase in all 6 sites, but were most profound in the middle chest wall area (channels 3 and 4) after the training period. The respiratory rate was observed to decrease slightly (13 to 10 breaths/min), and the mean rise time increased after the exercise and training period. Anthropometric and volumetric measurements. Figure 6 provides the anthropometric measurement results. The measurements reflect the chest wall and abdominal motion results presented except for a lack of improvement in the xiphoid and lower chest measurements during vital capacity breathing. The volumetric results demonstrated a 60% improvement, with the PSI H20 decreasing from 50 to 20mL H2O. The severity of pectus excavatum measured with the wooden ruler and tape measure was unchanged at 3.8cm (1.5in). Treadmill exercise testing. The symptom-limited treadmill test results are shown in table 4. Exercise test duration increased by 1 minute with slightly lower measurements of resting and submaximal heart rate, blood pressure, and symptoms. Exercise adherence. Adherence to exercise was calculated to be 76%.

Pulmonary function tests and echocardiography. The pulmonary function, ventilatory muscle strength, and echocardiography results are shown in table 3. The pulmonary function results are suggestive of a mild restrictive lung disease, with the FEV1/ FVC 106% of the predicted value and the residual volume 73% of the predicted value. Otherwise, the pulmonary function test results were relatively normalized and were relatively unchanged after PT interventions. MIP increased 12%, while MEP increased 39%. Ventilatory muscle endurance increased 50%. The echocardiographic results reveal mild valvular disorders consisting of minimal mitral valve prolapse, trace tricuspid

Table 3: Pulmonary Function and Echocardiography Results Pulmonary Function

Observation 1

Spirometry FEV1 (L) FVC (L) FEV1/FVC (%) Peak expiratory flow (L/s) Flow: 50% vital capacity (L/s) Flow: 75% vital capacity (L/s) Forced expiratory flow 25%⫺75% (L/s) Peak inspiratory flow (L/s) Maximal breathing capacity (L/min) Lung volume Total lung capacity (L) Residual volume (L) Residual volume/total lung capacity Functional residual capacity (L) Airway residual (cm H2O·L⫺1· s⫺1) Specific airway conductance (L·s·⫺1cm H2O⫺1·L⫺1) Ventilatory muscle performance MIP (cm H2O) MEP (cm H2O) Ventilatory muscle endurance (min) Echocardiography

4.42 4.94 89 8.26 5.49 2.53 4.79 5.53 155 6.12 1.18 0.19 3.37 1.58 .190

Predicted

% Predicted

4.50 5.34 84 9.59 6.23 3.57 4.88 6.71 158

98 93 106 86 88 71 98 82 98

6.87 1.61 0.23 3.69 0.8–2.4 ⬎.12

89 73 83 91

90 93 4.0

Observation 2

4.45 4.98 8.34 5.55 2.55 4.84 5.73 158 NV NV NV NV NV

NV 101 129 6.0

Anatomic region

Status

Dimensions

Expected Dimension

Mitral valve Left atrium Aortic valve LVOT Left ventricle Tricuspid valve right atrial vena cava Pulmonary valve and pulmonary artery Right ventricle Intra-arterial and intraventricular septum Ejection fraction (%)

Abnormal Normal Normal Normal Normal Normal Normal Normal Normal

— 19 32 42 28 8 8 NV 66

— 25–38mm 24–39mm 37–53mm NV 7–11mm 7–11mm NV ⬎50

Abbreviations: LVOT, left ventricular outflow tract; NV, no values.

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mm

Chest Wall & Abdominal Motion 30 25 20 15 10 5 0

Pre Post

Ch 1

Ch 2

Ch 3

Ch 4

Ch 5

Ch 6

seconds

Mean Rise Time 6 5 4 3 2 1 0

Pre Post

Ch 1

Ch 2

Ch 3

Ch 4

Ch 5

Ch 6

Fig 5. Respiratory motion measurement instrument results.

Outcomes and Progress The patient’s goals were achieved after the 12-week intervention period and consisted of being able to sleep and play basketball without shoulder pain, being able to play basketball without dyspnea, and improved subjective feelings of his pectus excavatum and strength without feelings of upper-extremity fatigue. DISCUSSION The purpose of this case study was to present the effects of an integrated PT intervention for a person with pectus excavatum. The patient tolerated the prescribed interventions very well. The positive results of increased upper-extremity strength, improved cardiopulmonary function, and improvements in the psychologic status helped to improve the overall QOL for this individual. Out results support the efficacy and need for integrated PT intervention for this patient and others like him who opt not to have surgical correction of their pectus excavatum. Conservative treatment for scoliosis has been shown to be an important alternative to those who cannot or will not opt for surgical treatment.25 Conservative treatment for adolescent idiopathic scoliosis can avoid dramatic progression of scoliosis during adulthood.26 Although this individual was a young adult, he demonstrated evidence of a semifixed pectus excavatum that appeared to respond to integrated PT. PT intervention provided to a younger person may have had a more dramatic effect. Previously, a 4-week PT program on patients with neuromuscular scoliosis along with a spinal orthosis was found to have a positive effect on pulmonary function with an 8% improvement in FVC in 15 patients with a mean age of 12 years.27 Resistance exercise along with aerobic exercises can have a positive effect on an individual’s overall physical function. Understanding the role of posture and the function of the skeletal and cardiac muscle is an important factor in developing an effective intervention. Improvements in decreasing the amount of thoracic kyphosis in sitting and standing with patient education and resistance exercise may be very beneficial. Rib mobility and lung volumes have been related to thoracic ky-

phosis.5 Those who present with scoliosis and increased thoracic kyphosis in a static standing posture may have asymmetric rib growth and impaired rib mobility during recreational activity involving increased stress to the cardiopulmonary system. This study describes an individualized rehabilitation intervention for a patient with pectus excavatum and bilateral shoulder pain. The program was based on 2 physical therapists’ examination findings and were based on cardiopulmonary and orthopedic and kinesiologic concepts. Developing an integrated PT intervention by effective communication with cardiopulmonary and orthopedic specialties is essential. Program development was performed based on history, radiologic findings, patient’s goals, and physical examination findings including specific postural assessment as well as thoracic wall excursion and pulmonary testing. The observed improvements in many of the outcome measures were likely a result of the fact that the patient was extremely motivated to improve his pectus excavatum and the symptoms he believed were related to pectus excavatum. The patient in this case study was motivated and attended every scheduled session and self-reported 100% compliance in performing all of the musculoskeletal strengthening and stretching exercises and 76% compliance with the cardiorespiratory interventions. This case report is important because it appears to be the first report of integrated cardiovascular, pulmonary, and musculoskeletal PT on a subject with pectus excavatum. Furthermore, a variety of novel outcome measures have been used to examine the effects of PT on pectus excavatum. Finally, pectus excavatum is the most common thoracic musculoskeletal anomaly of childhood and adolescence and deserves further attention. Many of the novel outcome measures were improved after 12 weeks of integrated PT including the anthropometric and volumetric measurements, chest wall and abdominal measurements, self-perception of pectus excavatum measurements, scapular measurements, and ventilatory muscle strength and endurance measurements. Few of these measurements have been previously reported in the literature in regard to pectus excavatum. The reliability and validity of several of the measurements have been reported, but reliability of several other measurements requires further investigation.

Chest Wall Excursion • Sternal Angle:

TV: 1 mm 10 mm VC: 2.5 cm 5.0 cm

• Xiphoid Process: TV: 3 mm VC: 7.5 cm

10 mm 7.5 cm

• Mid-Abdominal: TV: 8.5 mm VC: 4.5 cm

• Umbilicus: TV: 4.5 mm VC: -1 mm

10 mm 4.5 cm

8 mm 1.5 cm

Fig 6. Anthropometric measurements. Abbreviations: TV, tidal volume; VC, vital capacity breathing.

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BILATERAL SHOULDER PAIN AND PHYSICAL THERAPY, Canavan Table 4: Symptom-Limited Bruce Treadmill Exercise Test Results Variables

Heart rate Blood pressure

Borg RPE

Test

Rest

Minute 3

Minute 6

Minute 9

Pre Post Pre SBP DBP Post SBP DBP Pre Post

68 66 110 68 108 70 0 0

122 120 130 60 124 64 3 3

160 152 154 50 150 60 3 3

191 185 164 50 160 56 5 4

Minute 10

194

170 54 5

Minute 2 Postexercise

Minute 4 Postexercise

130 125 132 73 130 70 0.5 1

110 112 128 66 130 64 0.5 0.5

NOTE. Exercise test peak heart rate equal to 191 (96% of age-predicted heart rate); Exercise Test Electrocardiographic Interpretation equal to no arrhythmias or ST-segment abnormalities; Exercise Test Estimated Peak Oxygen Consumption: Pre-Exercise Training Test equal to 34.12mL·kg⫺1·min⫺1 (81% of age-predicted peak oxygen consumption); Postexercise Training Test equal to 37.48mL·kg⫺1·min⫺1 (88% of age-predicted peak oxygen consumption). Abbreviations: DBP, diastolic blood pressure; SBP, systolic blood pressure.

The rationale for the observed improvements in these novel measurements is needed to understand better the effects of integrated PT for the subject in this case and for other persons with pectus excavatum. The improvements in the anthropometric as well as the chest wall and abdominal measurements were likely a result of the effects of breathing exercises combined with the stretching and strengthening exercises shown in figure 4. Stretching and strengthening of the thoracic musculature combined with specific breathing exercises to the diaphragm, lateral costal areas, and site of pectus excavatum likely contributed to greater thoracic ROM and thoracic muscle strength (evidenced by a greater MIP and MEP). Greater thoracic ROM with improved thoracic and diaphragmatic muscle strength likely facilitated greater anthropometric, chest wall, and abdominal area motion. However, respiratory motion was observed to be most improved during tidal volume breathing. In fact, chest wall and abdominal motion during tidal volume breathing was found to improve at all sites after 12 weeks of integrated PT using 2 forms of measurement (RMMI, tape measurements). Respiratory motion during vital capacity breathing was performed only with a tape measure, which found the sternal angle and umbilicus sites to have greater motion after 12 weeks of integrated PT. Despite fewer sites showing greater respiratory motion during vital capacity breathing, the greater motion that was observed was substantial. The improvement at the sternal angle was 2 times greater and the improvement at the umbilicus was no longer negative (representing abdominal area retraction), but protruded 1.5cm versus a retraction of ⫺1.0mm. The improvements in respiratory rate and rise time are also important and demonstrate a greater duration of inspiratory rise in all but 1 site and more efficient respiration. Perhaps the most important finding from this case study is the greater amount of respiratory motion and mean rise time at RMMI sites 3 and 4 (the sites nearest the pectus excavatum) and the xiphoid process tape measure results. Observation of greater motion and inspiratory rise in these areas indicates that motion at the pectus excavatum site improved favorably, which may have potential to decrease the severity of pectus excavatum. The improvements may be a result of improved breathing control and biomechanics of breathing, which may also be valuable for persons with pectus excavatum. The improvement in the volumetric measurement (PSI H20) appears to indicate less severe pectus excavatum in this subject. However, the improvement in PSI H20 may be a result not only of less pectus excavatum but also of increased pectoral muscle mass or an altered depth of breathing, both of which could Arch Phys Med Rehabil Vol 89, November 2008

decrease the amount of water filling the pectus excavatum site. However, the depth and frequency of breathing were controlled, which makes the improvement in PSI H20 a result of either less pectus excavatum or increased pectoral muscle mass. The factor most responsible for the improvement in the volumetric measurement is the increase in pectoral muscle mass, because the depth of the pectus excavatum measured through ruler was unchanged. A more specific method to measure the severity of pectus excavatum is needed, such as a Breisky Pelvimeter, which may provide a measurement very similar to the traditional PSI. The potential for integrated PT to improve pectus excavatum has been previously alluded to and has been hypothesized to be caused by increased strength, tone, and mass of the pectoral muscles.28 Exercises very similar to those provided to the subject of this case have been suggested to have potential to decrease or maintain pectus excavatum through the concept of reverse origin and insertion of the pectoralis major muscle. It is hypothesized that fixing the upper extremities and contracting the pectoral muscles (or reversing a pectoral muscle contraction because of fixed upper extremities) has the potential to elevate the sternum with persons who have a nonfixed or semifixed pectus excavatum, especially with the pectoralis muscle positioned in its midrange of length.28 Further investigation of this effect in persons with pectus excavatum is needed. The rationale for improvement in the self-perception of pectus excavatum measurement is the improvement in posture, the improvement in thoracic appearance, and the subject’s perception that the pectus excavatum was less severe. Further investigations of such an effect on subjective outcome measures like the self-perception of pectus excavatum are needed. Additionally, the likely rationale for the improvements in the scapular measurements is an improved seated and standing posture because of improvements in the resting tone of posterior thoracic region musculature, but this requires further investigation. The methods used to determine the fixed versus nonfixed status of the pectus excavatum are novel and require further elaboration. The semifixed pectus excavatum status in this subject was unchanged after the integrated PT program, which was expected in this young adult with a relatively complete ossification of the chest wall. It seems likely that a nonfixed pectus excavatum may be more amenable to integrated PT than a fixed pectus excavatum. In fact, the specific methods used to measure the status of the pectus excavatum (nonfixed, semifixed, fixed) require further

BILATERAL SHOULDER PAIN AND PHYSICAL THERAPY, Canavan

discussion because they have never been previously described. Each of the methods appears to be a simple test with a biomechanic basis to examine the integrity of pectus excavatum. Examination of standing trunk flexion and changes in rib hump and thoracic spine may provide the therapist with information related to rib and spinal mobility and information related to prognosis for PT intervention. Optimal resting muscular tone and strength in the trunk and upper extremity muscles may be beneficial to help alleviate or diminish shoulder pain that may contribute to overall decreased activity level. The pectoralis major, rectus abdominus, rotator cuff, and scapula stabilizing muscle strength are important muscles related to posture and people with pectus excavatum. The traditional measurements made in this case study included posture assessment, leg length measurements MMT, and exercise testing. MMT of the rotator cuff muscles improved modestly; however, MMT assessment for grades over 3 is not as reliable as for grades 3 or below. The reasons for less significant improvements in the measurements include (1) measurements that were insensitive to the subtle changes in the pectus excavatum resulting from the integrated PT, (2) inappropriate measurements for the integrated PT program and the changes that were likely to be associated with the PT program provided to the patient, or (3) lack of substantial change in these measurements from the integrated PT. The most likely reason for the modest improvements in these measures is inappropriate measurements for the integrated PT program and the changes that were likely to be associated with the PT program provided to the patient in this case study. However, further investigation of these measurements and the effects of integrated PT are needed. The minimal-to-moderate improvement we observed in exercise tolerance is similar to the improvement observed in patients after corrective surgery for pectus excavatum. One investigator found a 7.9% increase in peak oxygen consumption in a 30-year-old who underwent corrective surgery for pectus excavatum,2 and another investigator examined the exercise tolerance of 15 postoperative adolescents who were found to exercise for a slightly longer duration with a slightly greater oxygen consumption.29 The subject of our study was observed to increase exercise duration by 1 minute, which represented a 9.8% increase in maximal oxygen consumption after 12 weeks of integrated PT. A progressive exercise training program was not provided in either of the studies referenced, which may have produced the minimal to modest improvements in exercise tolerance observed in subjects receiving surgical correction of pectus excavatum.2,29 Nonetheless, integrated PT provided to the young man in this case study improved exercise tolerance and oxygen consumption similarly to the results obtained with corrective surgery for pectus excavatum. Therefore, integrated PT should likely be provided to pectus excavatum patients prior to surgery to improve exercise tolerance and symptoms and to determine subsequently the need for corrective surgery. If corrective surgery is still warranted, integrated PT should likely be provided to patients with pectus excavatum after corrective surgery to improve exercise and functional tolerance as well as symptoms. Based on the available literature examining cardiorespiratory function in adolescents and young adults with pectus excavatum, integrated PT appears to have an important role in conservative and surgical correction of pectus excavatum.2,28,29 It is also important to note that another conservative nonsurgical method of managing pectus excavatum has recently been introduced. The conservative method essentially uses a vacuum to pull the pectus excavatum forward. This novel

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method has been studied in a total of 60 children, adolescents, and young adults (median age, 14.8y) who wore a suction cup over the pectus excavatum site for a median duration of 90 minutes daily for a median of 10 months. After 5 months, 12 subjects were observed to have no pectus excavatum.8 Exercises to facilitate the reduction in pectus excavatum were not used in this study. Knowledge of the attachment sites for the muscles and their function and development of an individualized PT rehabilitation program and patient compliance are paramount to improve the efficacy of PT intervention. Integrated PT provided to the patients in the study may have facilitated a more expedient correction of the pectus excavatum in more of the subjects and better maintained the correction of the pectus excavatum. Investigation of such combined therapy is needed. Other forms of nonsurgical correction of pectus excavatum have been reported on various websites with the most comprehensive methods employed by Sydney A. Haje, MD, PhD, an orthopedic pediatrician in Brazil.30 Unfortunately, much of the supportive literature for these nonsurgical techniques comes through patient testimonies and not scientific research. However, several of the methods are similar to the methods provided to the patient in this case study. Investigation of 1 or more of the nonsurgical techniques discussed for pectus excavatum is needed. CONCLUSIONS Future research on PT intervention is needed to determine the efficacy of an integrated PT program for patients with pectus excavatum and to identify optimal outcome measures for persons with pectus excavatum receiving PT. Integration of various PT approaches including cardiopulmonary and orthopedic appears to be warranted in persons with pectus excavatum. APPENDIX 1: PT INTERVENTIONS Cardiorespiratory Interventions Breathing Exercises Diaphragmatic breathing exercises Gentle breath-holding exercises Aerobic Exercise Training Mode of Exercise: Walk or jog routine and cycling. Walk or jog routine with several minutes of jogging followed by several minutes of walking within the target heart rate range described below. Cycling at a cadence of approximately 60rpm within the target heart rate range. Duration of Exercise: All exercise should be performed for 10 to 20 minutes with a goal of 20 to 30 minutes of total continuous aerobic exercise. Frequency of Exercise: Once a day, 3–5 times a week. Intensity of Exercise: Target heart rate range: 130 –150 beats/min (70%– 80% of maximal heart rate). Target Borg RPE: 3/10 (moderate exercise sensation with moderate dyspnea). Musculoskeletal Strengthening and Stretching Program Mid to low back stretches were performed to improve flexibility in this region. Standing with arms overhead, the subject was instructed to side-bend to the left and hold for 30 seconds to be performed with 4 repetitions, 3 times a day. The following exercise program was developed to address the identified weaknesses of the subject. The patient was instructed to add .45kg (1lb) of resistance when he was able to perform 20 repetitions with good technique and start back to 10 repetitions. Arch Phys Med Rehabil Vol 89, November 2008

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On All Fours. Cat/camel followed by shoulder flexion hold 3⫺6 seconds and repeat. On other upper extremity, 2 sets 10 –20 repetitions 1 time/day 3 times/wk. a) Hands directly under body (shoulder flexed 90°), 1 set 10 –20 reps. b) Hands in front of body (shoulder flexed ⬇120°), 1 set 10⫺20 reps. Supine. Knees flexed, shoulders in full shoulder. Elevation (bilateral shoulder extension half range to 90° of flexion) versus Theraband. Two sets 10⫺20 reps, 1 time/day 3 times/wk. Increasing the color of resistance after completion of 20 reps. Prone Bilateral Scapula Adduction with hands resting on buttocks. Hold 3⫺6s, 1 set 10⫺20 reps. Start at 10 reps, increase by 1⫺2 reps until 20 reps (fig 4D). Bilateral Horizontal Abduction with External Rotation. Prone arms abducted at 90°. Lift arms to the ceiling thumbs pointed up. Squeezing scapulas together, hold 3⫺6s. One set 10⫺20 reps (fig 4A). Bilateral Shoulder Elevation, Arms Abducted at ⬇130°. Lift arms to the ceiling with thumbs pointed to the ceiling. Squeezing scapulas together, hold 3⫺6s. One set 10⫺20 reps (fig 4C). Bilateral Shoulder Flexion. Arms held in maximal shoulder. Abduction with thumbs pointed toward the ceiling and lifting both arms up into the air while squeezing scapulas together. Hold 3⫺6s (fig 4B). References 1. Fonkalsrud EW. Current management of pectus excavatum. World J Surg 2003;27:502-8. 2. Malek M, Fonkalsrud EW, Cooper B. Ventilatory and cardiovascular responses to exercise in patients with pectus excavatum. Chest 2003;124:870-82. 3. Koumbourlis AC, Stolar CJ. Lung growth and function in children and adolescents with idiopathic pectus excavatum. Pediatr Pulmonol 2004;38:339-43. 4. Schoenmakers MA, Gulmans VA, Bax NM, Heiders PJ. Physiotherapy as an adjunct to the surgical treatment of anterior chest wall deformities: a necessity? a prospective descriptive study in 21 patients. J Pediatr Surg 2000;35:1440-5. 5. Culham EG, Jimenez HA, King CE. Thoracic kyphosis, rib mobility and lung volumes in normal women and women with osteoporosis. Spine 1994;19:1250-5. 6. Behrakis PK, Baydur A, Jaeger MJ, Milic-Emili J. Lung mechanics in sitting and horizontal body positions. Chest 1983;83:643-6. 7. Huddleston CB. Pectus excavatum. Semin Thorac Cardiovasc Surg 2004;16:225-32. 8. Schier F, Bahr M, Klobe E. The vacuum chest wall lifter: an innovative, non-surgical addition to the management of pectus excavatum. J Pediatr Surg 2005;40:496-500. 9. Borg G. Psychophysical bases of perceived exertion. Med Sci Sports Exerc 1982;14:377-81. 10. Killian KG, Jones NL. The use of exercise testing and other methods in the investigation of dyspnea. Clin Chest Med 1984;5: 99-108. 11. American Thoracic Society. Standardization of spirometry—1987 update. Am Rev Respir Dis 1987;136:1285-9. 12. Crapo RO, Morris AH, Gardner RM. Reference spirometric values using techniques and equipment that meet ATS recommendations. Am Rev Respir Dis 1981;123:659-64. 13. Cotes JE. Lung function assessment and application in medicine. 4th ed. Oxford: Blackwell; 1979.

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14. ACC/AHA guidelines for the clinical application of echocardiography. A report of the American College of Cardiology/American Heart Association Task Force on Assessment of Diagnostic and Therapeutic Cardiovascular Procedures (Subcommittee to Develop Guidelines for the Clinical Application of Echocardiography). J Am Coll Cardiol 1990;16:1505-28. 15. Black LF, Hyatt RE. Maximal respiratory pressures: normal values and relationship to age and sex. Am Rev Respir Dis 1969;99: 696-702. 16. Martyn JB, Moreno RM, Pare PD, Pardy RL. Measurement of inspiratory muscle performance with incremental threshold loading. Am Rev Respir Dis 1987;135:919-23. 17. Ragnarsdóttir M, KristjAnsdóttir A, Ingvarsdóttir I, Hannesson P, Torfason B, Cahalin L. Short term changes in pulmonary function and respiratory movements after cardiac surgery via median sternotomy. Scand Cardiovasc J 2004;38:46-52. 18. Moll JM, Wright V. An objective clinical study of chest expansion. Ann Rheum Dis 1972;31:1-8. 19. Harris J, Johansen J, Pedersen S, Kinney-LaPier T. Site of measurement and subject position affect chest excursion measurements. Cardiopulm Phys Ther 1977;8:12-7. 20. Bruce RA, Kusumi F, Hosmer D. Maximal oxygen intake and nomographic assessment of functional aerobic impairment in cardiovascular disease. Am Heart J 1973;85:546-60. 21. Plafcan DM, Canavan PK, Sebastianelli WJ, Swope KM. Reliability of a new instrument to measure scapula position. J Manual Manipulative Ther 2000;8:183-92. 22. Plafcan DM, Turczany PJ, Guenin BA, Kegerreis S, Worrell TW. An objective measurement technique for posterior scapular displacement. J Orthop Sports Phys Ther 1997;25:336-41. 23. Palmer ML, Epler ME. Fundamentals of musculoskeletal assessment techniques. 2nd ed. Philadelphia: Lippincott, Williams & Wilkins; 1998. 24. American College of Sports Medicine. ACSM’s guidelines for exercise testing and prescription. 5th ed. Baltimore: Lippincott, Williams & Wilkins; 1995. 25. Rigo M, Reiter CH, Weiss H. Effect of conservative management on the prevalence or surgery in patients with adolescent idiopathic scoliosis. Pediatr Rehabil 2003;6:209-14. 26. Maruyama T, Kitagawa T, Takeshita K, Mochizuki K, Nakamura K. Conservative treatment for adolescent idiopathic scoliosis: can it reduce the incidence of surgical treatment? Pediatr Rehabil 2003;6:215-9. 27. Bayar B, Uygur F, Bayar K, Bek N, Yakut Y. The short term effects of an exercise programme as an adjunct to an orthosis in neuromuscular scoliosis. Prosthet Orthot Int 2004;28:273-7. 28. Cheung SY. Exercise therapy in the correction of pectus excavatum. J Pediatr Respir Crit Care 2005;1:10-3. 29. Quigley PM, Haller JA, Jelus KL, Loughlin GM, Marcus CL. Cardiorespiratory function before and after corrective surgery in pectus excavatum. J Pediatr 1996;128:638-43. 30. Centro Clinico Orthopectus. Available at: http://www.orthopectus. com.br. Accessed July 18, 2008. Suppliers a. PK Morgan Instruments, Two Dundee Pk, Level One, Andover, MA 01810. b. Micro Medical, Quayside, Chatham Maritime, Chatham, Kent, ME4 4QY, UK. c. ReMo Inc, Keldnaholt, 113 Reykjavík, Iceland. d. Senix Ultra-U revision B; Senix Corp, 52 Maple St, Bristol, VT 05443.