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The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study
Journal of Orthopaedic Science xxx (xxxx) xxx
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Original Article
The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study Yasuhiko Takegami*, Taisuke Seki, Yusuke Osawa, Taiki Kusano, Naoki Ishiguro Department of Orthopaedic Surgery, Nagoya University Graduate School of Medicine, Nagoya, Japan
a r t i c l e i n f o
a b s t r a c t
Article history: Received 3 June 2019 Received in revised form 20 August 2019 Accepted 4 September 2019 Available online xxx
Introduction: This study aimed to clarify the relationship of individual radiographic parameters and features with the patient-reported outcome measurements (PROMs) of patients with developmental dysplasia of the hip (DDH) in a hospital-referred cross-sectional analysis. Methods: This cross-sectional study included 108 Japanese DDH patients (female, n ¼ 88; male, n ¼ 20; mean age, 43.4 years). We recorded the CE angle, angle of Sharp, ARO, ADR, AHI, crossover sign, pistol grip deformity, and femoral head-neck ratio, which were measured on the AP view, and the a-angle, which was measured on the Lauenstein view. We also recorded cyst formation and osteophytes on either view. We used the Japanese Orthopaedic Association hip disease evaluation questionnaire (JHEQ) to assess the PROMs. The PROMs of patients with and without cysts or osteophytes and those of patients with or without cam or pincer deformity were compared. The relationships between specific PROMs and radiographic features were evaluated with a linear regression analysis and independent associations between PROMs and radiographic features were assessed with a multiple regression analysis using various independent variables. Results: The JHEQ movement and mental values in patients with cyst formation were significantly lower in comparison to those in patients without cyst formation. The JHEQ subscale values did not differ according to the presence or absence of osteophytes, cam deformity or pincer deformity. The CE angle was found to be associated with the JHEQ movement score in the linear regression analysis. To identify radiographic features that were independently associated with each of the JHEQ subscale scores, we performed a multiple regression analysis with age, body mass index, sex and the number of affected hip joints (bilateral or unilateral) included as independent variables. The CE angle was found to be independently associated with JHEQ movement. Conclusion: The CE angle was independently associated with the JHEQ movement value. © 2019 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.
1. Introduction Developmental dysplasia of the hip (DDH) is known to cause hip pain and functional impairment, so-called acetabular rim syndrome [1]. In a hospital-based cross-sectional study, 77% of the patients with DDH felt moderate or severe pain. Eighty-five percent of those had a limp when walking a short distance [2]. In another study, patients with pre-arthritic hip disorders, including mild DDH
* Corresponding author. Department of Orthopaedic Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-Cho, Showa-Ku, Nagoya, 466-8550, Japan. Fax: þ81 52 744 2260. E-mail address: [email protected] (Y. Takegami).
demonstrated a moderate degree of pain and lower physical and mental scores, as assessed by the Medical Outcomes Study Short Form-12 (SF-12) [3]. DDH is defined according to the following radiographic parameters: the center-edge angle (CE angle) [4], the angle of Sharp [5], the acetabular roof oblique (ARO) [6], the acetabular depth ratio (ADR) [7], and the acetabular head index (AHI) [8]. A number of studies have referred to the relationships between these parameters [9,10]. In addition, in advanced osteoarthritis of the hip, the radiographic features, which are characterized by cyst and osteophyte formation, are associated with patient-reported outcome measurements (PROMs) of patients with osteoarthritis of the hip [11]. Radiographic femoroacetabular impingement (FAI), which is
https://doi.org/10.1016/j.jos.2019.09.007 0949-2658/© 2019 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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demonstrated by cam or pincer type deformities, was also found to be common in a population of healthy young adults in a large crosssectional study [12]. To the best of our knowledge, the relationship between pain and impairment (as assessed by PROMs) and the radiographic parameters and features have scarcely been mentioned in the literature. This study aimed to clarify the relationship between each radiographic parameters and features and their effect on the PROMs of patients with DDH in a hospital-referred cross-sectional analysis.
1.1. Patients and methods This study was approved by the institutional review board of our institution. All participants provided their written informed consent at the time of the first consultation.
1.2. Subjects We reviewed 510 patients who newly visited a universityhospital outpatient clinic between April 2014 and April 2015. The study population included 366 women (460 symptomatic hips) and 144 men (258 symptomatic hips). First, we selected the 243 patients with radiographically demonstrated Tonnis grade 0 or 1 [13] and with radiographic findings of DDH in at least one of the hip joints, which was defined as follows: CE angle <25 , angle of Sharp >45 , ARO >13 , ADR <43%, and AHI <75% [14,15]. The following exclusion criteria were applied: 1) patients who have undergone hip arthroplasty or rotational acetabular osteotomy on the contralateral side of the affected hip (n ¼ 77); 2) former hip surgery of any kind including osteotomy in their affected hip (n ¼ 20); 3) dislocated hip on the contralateral side of the affected hip (n ¼ 13); 4) patients with Perthes-like deformities of their femoral head despite conservative treatment for DDH (n ¼ 12); 5) patients with lumber spine disease such as spina bifida, spinal caries and lumber canal stenosis with posterior fusion operation (n ¼ 3); 6) patients with neurological disorder such as cerebral palsy and congenital muscular dystrophy (n ¼ 2); 7) patients with systemic bone disease such as achondroplasia (n ¼ 2) and 8) incomplete answers to a questionnaire (n ¼ 6). After the exclusion of these patients, 108 patients (male, n ¼ 20; female, n ¼ 88) were included in the analysis (Fig. 1). In addition, 10 patients out of 108 patients were received any conservative treatment in their early childhood. Thus, we showed the comparison of patient demographics and PROM between the patients who received conservative treatments in their early childhood and the patients who had not any treatment for their DDH in the supplemental Tables 1 and 2 (Available Online).
Fig. 1. Flowchart showing the application of the inclusion and exclusion criteria.
1.3. Radiographic methods We assessed the pelvic radiographs in both the anteroposterior (AP) and Lauenstein views. Pelvic radiographs were taken with the patients in the supine position with a tube-to-film distance of 130 cm and the tube perpendicular to the table. The central beam in the AP view was directed to the midpoint between the upper border of the pubic symphysis and a horizontal line connecting both anterior iliac spines. The CE angle, angle of Sharp, ARO, ADR, AHI, crossover sign, pistol grip deformity, and femoral head-neck ratio were measured on the AP view. The a-angle was measured on the Lauenstein view. Hips with either an a-angle of >50 , pistol grip deformity or a femoral head-neck ratio of <1.27 were defined as the cam deformity group. Patients whose hips showed the cross-over sign were classified into the pincer deformity group. These are described in previous reports [14,15]. In addition, we recorded the cyst formation and osteophyte formation as radiographic features of osteoarthritis of the hip according to previous reports [11,16]. We defined the bone formation located in the marginal of acetabular rim or femoral head as osteophyte. A bone cyst is defined as radiolucent lesion with bone sclerosis in the subchondral bone in the acetabulum or femoral head [16]. Radiographic measurements were obtained using a computerized medical information system (Neochart Hospital Information System; Fujitsu, Tokyo, Japan). One orthopedic physician (TY) performed all radiographic evaluations. To investigate intraobserver reliability, all radiographs were re-measured at a 2-week interval. The average of the two values was used for the analysis. 1.4. PROMs instruments We assessed the PROMs with the Japanese Orthopaedic Association hip disease evaluation questionnaire (JHEQ). The JHEQ has been established as one of the PROMs for patients with hip disease [17]. The JHEQ consists of pain (28 points), movement (28 points), and mental (28 points) subscales, with higher scores indicating a better outcome. The JHEQ has been validated for use in the clinical evaluation of patients with hip disease [18]. 1.5. Statistical analysis First, we compared the correlation between each radiographic measurement using Pearson's correlation coefficient. Second, we compared patient reported outcomes with or without each radiographic feature (cyst formation, osteophytes, cam deformity, and pincer deformity). The normality of the distribution of each subscale of the JHEQ was tested for using the ShapiroeWilk test. As a result, no subscales showed a normal distribution. Thus, we used the ManneWhitney test. Third, we performed a linear regression analysis to assess the relationship between the JHEQ subscales and each radiographic feature. We then performed a multiple regression analysis to assess the independent associations of radiographic measurements and radiographic features with the JHEQ subscales with age, body mass index (BMI), sex, radiographic features and radiographic measurements as independent variables. Finally, we used the interclass correlation coefficient to assess the reproducibility of the data. The data on intraobserver reliability of the radiographic interpretations are summarized in Supplemental Table 3 (Available Online). Based on the 95% confidence interval of the ICC estimate, values of <0.5, 0.5e0.75, 0.75e0.9, and >0.90 are indicative of poor, moderate, good, and excellent reliability, respectively. Based on the data, the intraobserver agreement was acceptable. P values of <0.05 were considered to indicate statistical
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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significance. All statistical analyses were performed using EZR (Saitama Medical Center, Jichi Medical University) [19].
Table 1 Patient demographics. Number of patients Female/Male, no. (%) Age, mean ± SD years, (range) Body mass index, mean ± SD kg/m2 Hip involvement, bilateral/Unilateral, no (%) Minimum joint space, mean ± SD mm, (range) Tonnis grading of each hip: 0/1 no. of hips. (%) Presence of the radiographic feature of HOA Cyst (None/Ace/FH/both), no (%) Osteophyte (None/Ace/FH/both), no (%) The presence of the radiographic feature of FAI None/Pincer deformity/Cam deformity/Combined, no (%) Duration of symptom, median (month) (range)
108 88 (91)/20 (19) 43.7 ± 12.0 (15e69) 22.6 ± 3.0 102 (95)/6 (5) 4.6 ± 1.3 (1.9e7.5) 156(74)/54(26) 29(16)/42(23) 73 (68)/25 (23)/6(6)/4 (4) 79 (73)/24 (22)/1 (1)/4 (4) 87 (81)/14 (13)/7 (6)/0 (0) 7 (1e228)
*SD, Standard deviation; Ace, radiographic feature exists on the acetabular side; FH, radiographic feature exists on the femoral head; both, radiographic feature exists on both the acetabular side and on the femoral head; FAI, femoroacetabular impingement.
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2. Results The characteristics of the study patients are shown in Table 1. Ninety-five percent of the patients had bilateral DDH. Table 2 shows the correlation coefficients between the radiographic measurements. Significant correlations were observed between all groups (P < 0.001). The CE angle had the highest correlation with other radiographic measurements. The JHEQ movement values of patients with cyst formation were significantly lower in comparison to the JHEQ movement values of patients without cyst formation. The JHEQ mental values of patients with cyst formation were also significantly lower in comparison to the JHEQ mental values of patients without cyst formation. The JHEQ pain values of patients with and without cyst formation did not differ to a statistically significant extent. The JHEQ subscale values did not differ according to the presence or absence of osteophytes (Fig. 2). Fig. 3 demonstrates that the JHEQ subscale values were not significantly influenced by the presence or absence of cam or pincer deformity. The relationships between each JHEQ subscale value and the radiographic features were evaluated by a linear regression analysis. We decided to deal with the CE angle as a representative
Table 2 The correlation coefficients (95% confidence intervals) between radiographic measurements.
AHI ADR ARO CEA
ADR
ARO
CEA
Sharp
0.58 (0.43, 0.70) e e e
0.56 (0.67, 0.41) 0.68 (0.78, 0.56) e e
0.86 (0.79, 0.92) 0.71 (0.60, 0.80) 0.76 (0.84, 0.67) e
0.59 (0.70, 0.49) 0.32 (0.49, 0.12) 0.53 (0.36, 0.66) 0.68 (0.78, 0.56)
AHI, Acetabular head index; ADR, Acetabular depth ratio; ARO, Acetabular roof obliquity; CEA, Center-edge (CE) angle; Sharp, Angle of Sharp. All P values < 0.05.
Fig. 2. The JHEQ subscale values for the hip with or without cysts and osteophytes. (a) The JHEQ subscale values in patients with or without cyst formation. (b) The JHEQ subscale values in patients with or without osteophytes. A, radiographic features absent; P, radiographic features present. *P value < 0.05.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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Fig. 3. The JHEQ subscale values hips with or without femoroacetabular impingement. (a) The JHEQ subscale values in patients with or without cam deformity. (b) The JHEQ subscale values in patients with or without pincer deformity A, radiographic features absent; P, radiographic features present.
radiological measurement in both the linear regression analysis and the multiple regression analysis because it showed the highest correlation among the radiographic measurements (Table 2). The CE angle was found to be associated with the JHEQ movement score in the linear regression analysis (Table 3). To determine the independent associations of each radiographic feature with the scores on each of the JHEQ subscales, we performed a multiple regression analysis with age, body mass index, sex and the number of affected hip joints (bilateral or unilateral) included as independent variables. The CE angle was found to be independently associated with JHEQ movement (b coefficient 1.72; adjusted R^2 ¼ 0.12) (Table 3).
3. Discussion Our data demonstrated that the CE angle was independently associated with the JHEQ movement value. In a previous large cross-sectional study in Norway, there is no association between CE angle and patient-based assessments such as WOMAC or EQ-5D [20]. However, the participants' background was different from our study. This large cross-sectional study was conducted with healthy men and women aged 19 years with/without hip pain. While, we did this research at a university hospital, the patients who had visited a medical institution at least once with some of complaint about the hip joint. Yasunaga et al. reported that patients
Table 3 Association of each JHEQ subscale with each radiographic evaluation, adjusted for age and gender. Univariate regression coefficient
CEA Cyst Osteophyte Pincer Cam
JHEQ pain (95%CI)
P value
JHEQ movement (95%CI)
P value
JHEQ mental (95%CI)
P value
0.27 (1.7,1.3) 0.59 (2.4,0.91) 0.79 (2.3,0.76) 0.35 (1.83,1.21) 0.01 (1.5,1.5)
0.70 0.42 0.36 0.72 0.98
2.1 (3.6,0.51) 1.71 (3.3,0.19) 1.33 (2.9,0.26) 1.0 (2.6,0.61) 0.2 (1.8,1.6)
0.009 0.030 0.10 0.20 0.82
0.07 (2.1,0.9) 1.69 (3.3,0.17) 0.81 (2.6,0.72) 0.50 (2.1,1.1) 0.30 (1.5,1.9)
0.40 0.03 0.28 0.54 0.75
Multiple regression coefficient
CEA Cyst Osteophyte Pincer Cam
JHEQ pain (95%CI)
P value
JHEQ movement (95%CI)
P value
JHEQ mental (95%CI)
P value
VIF
0.29 (1.7, 1.2) 0.60 (2.4, 0.8) 0.88 (2.4, 0.74) 0.33 (1.8, 1.2) 0.01 (1.5, 1.5)
0.72 0.43 0.31 0.70 0.99
1.71 (3.35, 0.11) 0.92 (2.43, 0.66) 0.48 (2.2, 1.20) 1.4 (2.9, 0.15) 0.31 (1.90, 1.35)
0.04* 0.29 0.66 0.09 0.73
1.2 (2.8, 0.5) 0.96 (2.52, 0.62) 0.11 (1.42, 1.64) 1.1 (2.35, 0.76) 0.56 (1.2, 2.1)
0.20 0.24 0.89 0.44 0.53
1.14 1.12 1.09 1.03 1.33
CI, confidence interval; CEA, center-edge angle; Pincer, presence of pincer deformity; Cam, presence of cam deformity; VIF, variance inflation factor. *P value < 0.05.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
Y. Takegami et al. / Journal of Orthopaedic Science xxx (xxxx) xxx
with severe DDH (preoperative mean CE angle: 1.6 ) who underwent rotational acetabular hip osteotomy had preoperative pain that was tolerable with limited daily activity [21]. Another large retrospective study demonstrated that patients with severe DDH (<5 ) had less activity, as assessed by the UCLA score [22]. These reports implied that the activity of patients with severe DDH might be reduced, which would have led to a decrease in hip pain along with a decline in their daily activity. In a finite element study, it was reported that the von Mises stress on the acetabular side and the femoral head side increased as the CE angle decreased [23]. An evaluation of dGEMRIC for early osteoarthritis of hip using MRI in patients with DDH also showed a correlation between CE angle and signal changes of the dGEMERIC, which suggested cartilage degeneration [24,25]. Thus, the CE angle is involved in the early development of coxarthrosis. Patients who develop pain due to DDH over time to avoid osteoarthritis may limit activities of daily living. It is expected that all patients felt some pain from the time they had went to the local medical institution to the time they visited the university hospital. Moreover, it is known that behavioral restrictions were effective as a conservative treatment for coxalgia in patients with various hip diseases [26e28]. By the time they visited our university hospital, the patients who develop pain due to DDH may limit activities of daily living. There was a significant difference in the JHEQ movement scores of the patients with and without cyst formation. Cyst formation was associated with the initiation and progression of osteoarthritis of the hip [29]. A previous report revealed the cyst formation significantly affected hip pain and impairment [11]. Radiographic cyst formation might indicate the progression of osteoarthritis of the hip. There was no relationship between the FAI findings and PROMs. A retrospective study compared the radiographs of symptomatic patients to those of asymptomatic patients, and found no difference in the prevalence of FAI between the groups [30]. That is, FAI-like morphologic changes may not be related to pain or dysfunction in patients with DDH. This present study is associated with several limitations. False profile views [31], might have been obtained when the anterior coverage of the femoral head was evaluated in the standing position. To prevent excessive radiation exposure, we minimized the imaging studies that were performed to those that were necessary. Second, we did not evaluate labrum tear, bone marrow edema or cartilage degeneration on MRI. These are also causes of hip pain. Third, patients who were referred to our hospital were introduced by local hospitals as patients who required specialized treatment. They might have received conservative treatments including the behavioral restrictions at the local hospitals before visiting our hospital. Details of the previous hospital's conservative treatment were not available, but these may have affected the results. Fourth, this was a hospital-referred cross-sectional study that was performed in a single university hospital. Thus, the causal relationship could not be determined. In conclusion, the CE angle was independently associated with the JHEQ movement value. While no factors were independently associated with JHEQ pain, the JHEQ movement values of DDH patients with cyst formation were lower than those of patients without cyst formation. The FAI findings were not related to the PROMs in DDH patients. These radiographic features may have an application in the assessment of pain and functional impairment in DDH patients. Conflicts of interest None.
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Acknowledgement This work was supported by Grant-in-Aid for Scientific Research C. Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi.org/10.1016/j.jos.2019.09.007. References [1] Klaue K, Durnin CW, Ganz R. The acetabular rim syndrome. A clinical presentation of dysplasia of the hip. J Bone Joint Surg Br 1991 May;73(3):423e9. [2] Nunley RM, Prather H, Hunt D, Schoenecker PL, Clohisy JC. Clinical presentation of symptomatic acetabular dysplasia in skeletally mature patients. J Bone Joint Surg Am 2011 May;93(Suppl. 2):17e21. [3] Hunt D, Prather H, Harris M, Clohisy JC. Clinical outcomes analysis of conservative and surgical treatment of patients with clinical indications of prearthritic, intra-articular hip disorders. PM R 2012 July;4(7):479e87. [4] Fredensborg N. The results of early treatment of typical congenital dislocation €. J Bone Joint Surg Br 1976 Aug;58(3):272e8. of the hip in Malmo [5] Sharp IK. Acetabular dysplasia: the acetabular angle. J Bone Joint Surg Br 1961 May;43(2):268e72. [6] Massie WK. Howorth MB Congenital dislocation of the hip. Part I. Method of grading results. J Bone Joint Surg 1950 July;32(3):519e31. [7] Cooperman DR, Wallensten R, Stulberg SD. Acetabular dysplasia in the adult. Clin Orthop Relat Res 1983 May;175:79e85. [8] Heyman CH, Herndon CH. Legg-Perthes disease; a method for the measurement of the roentgenographic result. J Bone Joint Surg 1950 Sep;32(4): 767e78. [9] Daysal GA, Goker B, Gonen E, Demirag MD, Haznedaroglu S, Ozturk MA, Block JA. The relationship between hip joint space width, center-edge angle and acetabular depth. Osteoarthr Cartil 2007 Dec;15(12):1446e51. [10] Lequesne M. The normal hip joint space: variations in width, shape, and architecture on 223 pelvic radiographs. Ann Rheum Dis 2004 Aug;63(9): 1145e51. [11] Takegami Y, Seki T, Higuchi Y, Komatsu D, Nishida Y, Ishiguro N. Independent association of joint space narrowing, cyst formation and health-related quality of life of patients with hip osteoarthritis in Japan. J Orthop Sci 2017 Nov;22(6):1096e101. [12] Laborie LB, Lehmann TG, Engesæter IØ, Eastwood DM, Engesæter LB, Rosendahl K. Prevalence of radiographic findings thought to Be associated with femoroacetabular impingement in a population-based cohort of 2081 healthy young adults. Radiology 2011 Aug;260(2):494e502. €nnis D, Legal H, Graf R. Congenital dysplasia and dislocation of the hip in [13] To children and adults. Berlin: GermanySpringer; 1987. [14] Mori R, Yasunaga Y, Yamasaki T, Nakashiro J, Fujii J, Terayama J, Ohshima S, Ochi M. Are cam and pincer deformities as common as dysplasia in Japanese patients with hip pain? Bone Joint J 2014 Feb;96(2):172e6. [15] Nehme A, Trousdale R, Tannous Z, Maalouf G, Puget J, Telmont N. Developmental dysplasia of the hip: is acetabular retroversion a crucial factor? Orthop Traumatol Surg Res 2009 Nov;95(7):511e9. [16] Altman RD, Gold GE. Atlas of individual radiographic features in osteoarthritis, revised. Osteoarthr Cartil 2007;15(Suppl A):1e56. [17] Matsumoto T, Kaneuji A, Hiejima Y, Sugiyama H, Akiyama H, Atsumi T, Ishii T, Izumi K, Ichiseki T, Ito H, Okawa T, Ohzono K, Otsuka H, Kishida S, Kobayashi S, Sawaguchi T, Sugano N, Nakajima Y, Nakamura S, Hasegawa Y, Fukuda K, Fujii G, Mawatari T, Mori S, Yasunaga Y, Yamaguchi M. Japanese orthopaedic association hip disease evaluation questionnaire (JHEQ): a patient-based evaluation tool for hip-joint disease. The subcommittee on hip disease evaluation of the clinical outcome committee of the Japanese orthopaedic association. J Orthop Sci 2012 Jan;17(1):25e38. [18] Seki T, Ishiguro N, Hasegawa Y, Ikeuchi K, Hiejima. Reliability and validity of the Japanese Orthopaedic Association hip disease evaluation questionnaire (JHEQ) for patients with hip disease. J Orthop Sci 2013 Sep;18(5):782e7. [19] Kanda Y. Investigation of the freely available easy-to-use software ‘EZR’ for medical statistics. Bone Marrow Transplant 2013 Mar;48(3):452e8. [20] Engesæter IØ, Laborie LB, Lehmann TG, Lie SA, Engesæter LB, Rosendahl K. Prevalence of radiographic findings associated with hip dysplasia in a population-based cohort of 2081 19-year-old Norwegians. Bone Joint J 2013 Feb;95-B(2):279e85. [21] Yasunaga Y, Ochi M, Yamasaki T, Shoji T, Izumi S. Rotational acetabular osteotomy for pre- and early osteoarthritis secondary to dysplasia provides durable results at 20 years. Clin Orthop Relat Res 2016 Oct;474(10):2145e53. [22] Matheney T, Zaltz I, Kim Y-J, Schoenecker P, Mills M, Podeszwa D, Zurakowski D, Ckohisy J. Activity level and severity of dysplasia predict age at bernese periacetabular osteotomy for symptomatic hip dysplasia. J Bone Joint Surg 2016 April;98(8):665e71.
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Original Article
The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study Yasuhiko Takegami*, Taisuke Seki, Yusuke Osawa, Taiki Kusano, Naoki Ishiguro Department of Orthopaedic Surgery, Nagoya University Graduate School of Medicine, Nagoya, Japan
a r t i c l e i n f o
a b s t r a c t
Article history: Received 3 June 2019 Received in revised form 20 August 2019 Accepted 4 September 2019 Available online xxx
Introduction: This study aimed to clarify the relationship of individual radiographic parameters and features with the patient-reported outcome measurements (PROMs) of patients with developmental dysplasia of the hip (DDH) in a hospital-referred cross-sectional analysis. Methods: This cross-sectional study included 108 Japanese DDH patients (female, n ¼ 88; male, n ¼ 20; mean age, 43.4 years). We recorded the CE angle, angle of Sharp, ARO, ADR, AHI, crossover sign, pistol grip deformity, and femoral head-neck ratio, which were measured on the AP view, and the a-angle, which was measured on the Lauenstein view. We also recorded cyst formation and osteophytes on either view. We used the Japanese Orthopaedic Association hip disease evaluation questionnaire (JHEQ) to assess the PROMs. The PROMs of patients with and without cysts or osteophytes and those of patients with or without cam or pincer deformity were compared. The relationships between specific PROMs and radiographic features were evaluated with a linear regression analysis and independent associations between PROMs and radiographic features were assessed with a multiple regression analysis using various independent variables. Results: The JHEQ movement and mental values in patients with cyst formation were significantly lower in comparison to those in patients without cyst formation. The JHEQ subscale values did not differ according to the presence or absence of osteophytes, cam deformity or pincer deformity. The CE angle was found to be associated with the JHEQ movement score in the linear regression analysis. To identify radiographic features that were independently associated with each of the JHEQ subscale scores, we performed a multiple regression analysis with age, body mass index, sex and the number of affected hip joints (bilateral or unilateral) included as independent variables. The CE angle was found to be independently associated with JHEQ movement. Conclusion: The CE angle was independently associated with the JHEQ movement value. © 2019 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.
1. Introduction Developmental dysplasia of the hip (DDH) is known to cause hip pain and functional impairment, so-called acetabular rim syndrome [1]. In a hospital-based cross-sectional study, 77% of the patients with DDH felt moderate or severe pain. Eighty-five percent of those had a limp when walking a short distance [2]. In another study, patients with pre-arthritic hip disorders, including mild DDH
* Corresponding author. Department of Orthopaedic Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-Cho, Showa-Ku, Nagoya, 466-8550, Japan. Fax: þ81 52 744 2260. E-mail address: [email protected] (Y. Takegami).
demonstrated a moderate degree of pain and lower physical and mental scores, as assessed by the Medical Outcomes Study Short Form-12 (SF-12) [3]. DDH is defined according to the following radiographic parameters: the center-edge angle (CE angle) [4], the angle of Sharp [5], the acetabular roof oblique (ARO) [6], the acetabular depth ratio (ADR) [7], and the acetabular head index (AHI) [8]. A number of studies have referred to the relationships between these parameters [9,10]. In addition, in advanced osteoarthritis of the hip, the radiographic features, which are characterized by cyst and osteophyte formation, are associated with patient-reported outcome measurements (PROMs) of patients with osteoarthritis of the hip [11]. Radiographic femoroacetabular impingement (FAI), which is
https://doi.org/10.1016/j.jos.2019.09.007 0949-2658/© 2019 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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demonstrated by cam or pincer type deformities, was also found to be common in a population of healthy young adults in a large crosssectional study [12]. To the best of our knowledge, the relationship between pain and impairment (as assessed by PROMs) and the radiographic parameters and features have scarcely been mentioned in the literature. This study aimed to clarify the relationship between each radiographic parameters and features and their effect on the PROMs of patients with DDH in a hospital-referred cross-sectional analysis.
1.1. Patients and methods This study was approved by the institutional review board of our institution. All participants provided their written informed consent at the time of the first consultation.
1.2. Subjects We reviewed 510 patients who newly visited a universityhospital outpatient clinic between April 2014 and April 2015. The study population included 366 women (460 symptomatic hips) and 144 men (258 symptomatic hips). First, we selected the 243 patients with radiographically demonstrated Tonnis grade 0 or 1 [13] and with radiographic findings of DDH in at least one of the hip joints, which was defined as follows: CE angle <25 , angle of Sharp >45 , ARO >13 , ADR <43%, and AHI <75% [14,15]. The following exclusion criteria were applied: 1) patients who have undergone hip arthroplasty or rotational acetabular osteotomy on the contralateral side of the affected hip (n ¼ 77); 2) former hip surgery of any kind including osteotomy in their affected hip (n ¼ 20); 3) dislocated hip on the contralateral side of the affected hip (n ¼ 13); 4) patients with Perthes-like deformities of their femoral head despite conservative treatment for DDH (n ¼ 12); 5) patients with lumber spine disease such as spina bifida, spinal caries and lumber canal stenosis with posterior fusion operation (n ¼ 3); 6) patients with neurological disorder such as cerebral palsy and congenital muscular dystrophy (n ¼ 2); 7) patients with systemic bone disease such as achondroplasia (n ¼ 2) and 8) incomplete answers to a questionnaire (n ¼ 6). After the exclusion of these patients, 108 patients (male, n ¼ 20; female, n ¼ 88) were included in the analysis (Fig. 1). In addition, 10 patients out of 108 patients were received any conservative treatment in their early childhood. Thus, we showed the comparison of patient demographics and PROM between the patients who received conservative treatments in their early childhood and the patients who had not any treatment for their DDH in the supplemental Tables 1 and 2 (Available Online).
Fig. 1. Flowchart showing the application of the inclusion and exclusion criteria.
1.3. Radiographic methods We assessed the pelvic radiographs in both the anteroposterior (AP) and Lauenstein views. Pelvic radiographs were taken with the patients in the supine position with a tube-to-film distance of 130 cm and the tube perpendicular to the table. The central beam in the AP view was directed to the midpoint between the upper border of the pubic symphysis and a horizontal line connecting both anterior iliac spines. The CE angle, angle of Sharp, ARO, ADR, AHI, crossover sign, pistol grip deformity, and femoral head-neck ratio were measured on the AP view. The a-angle was measured on the Lauenstein view. Hips with either an a-angle of >50 , pistol grip deformity or a femoral head-neck ratio of <1.27 were defined as the cam deformity group. Patients whose hips showed the cross-over sign were classified into the pincer deformity group. These are described in previous reports [14,15]. In addition, we recorded the cyst formation and osteophyte formation as radiographic features of osteoarthritis of the hip according to previous reports [11,16]. We defined the bone formation located in the marginal of acetabular rim or femoral head as osteophyte. A bone cyst is defined as radiolucent lesion with bone sclerosis in the subchondral bone in the acetabulum or femoral head [16]. Radiographic measurements were obtained using a computerized medical information system (Neochart Hospital Information System; Fujitsu, Tokyo, Japan). One orthopedic physician (TY) performed all radiographic evaluations. To investigate intraobserver reliability, all radiographs were re-measured at a 2-week interval. The average of the two values was used for the analysis. 1.4. PROMs instruments We assessed the PROMs with the Japanese Orthopaedic Association hip disease evaluation questionnaire (JHEQ). The JHEQ has been established as one of the PROMs for patients with hip disease [17]. The JHEQ consists of pain (28 points), movement (28 points), and mental (28 points) subscales, with higher scores indicating a better outcome. The JHEQ has been validated for use in the clinical evaluation of patients with hip disease [18]. 1.5. Statistical analysis First, we compared the correlation between each radiographic measurement using Pearson's correlation coefficient. Second, we compared patient reported outcomes with or without each radiographic feature (cyst formation, osteophytes, cam deformity, and pincer deformity). The normality of the distribution of each subscale of the JHEQ was tested for using the ShapiroeWilk test. As a result, no subscales showed a normal distribution. Thus, we used the ManneWhitney test. Third, we performed a linear regression analysis to assess the relationship between the JHEQ subscales and each radiographic feature. We then performed a multiple regression analysis to assess the independent associations of radiographic measurements and radiographic features with the JHEQ subscales with age, body mass index (BMI), sex, radiographic features and radiographic measurements as independent variables. Finally, we used the interclass correlation coefficient to assess the reproducibility of the data. The data on intraobserver reliability of the radiographic interpretations are summarized in Supplemental Table 3 (Available Online). Based on the 95% confidence interval of the ICC estimate, values of <0.5, 0.5e0.75, 0.75e0.9, and >0.90 are indicative of poor, moderate, good, and excellent reliability, respectively. Based on the data, the intraobserver agreement was acceptable. P values of <0.05 were considered to indicate statistical
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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significance. All statistical analyses were performed using EZR (Saitama Medical Center, Jichi Medical University) [19].
Table 1 Patient demographics. Number of patients Female/Male, no. (%) Age, mean ± SD years, (range) Body mass index, mean ± SD kg/m2 Hip involvement, bilateral/Unilateral, no (%) Minimum joint space, mean ± SD mm, (range) Tonnis grading of each hip: 0/1 no. of hips. (%) Presence of the radiographic feature of HOA Cyst (None/Ace/FH/both), no (%) Osteophyte (None/Ace/FH/both), no (%) The presence of the radiographic feature of FAI None/Pincer deformity/Cam deformity/Combined, no (%) Duration of symptom, median (month) (range)
108 88 (91)/20 (19) 43.7 ± 12.0 (15e69) 22.6 ± 3.0 102 (95)/6 (5) 4.6 ± 1.3 (1.9e7.5) 156(74)/54(26) 29(16)/42(23) 73 (68)/25 (23)/6(6)/4 (4) 79 (73)/24 (22)/1 (1)/4 (4) 87 (81)/14 (13)/7 (6)/0 (0) 7 (1e228)
*SD, Standard deviation; Ace, radiographic feature exists on the acetabular side; FH, radiographic feature exists on the femoral head; both, radiographic feature exists on both the acetabular side and on the femoral head; FAI, femoroacetabular impingement.
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2. Results The characteristics of the study patients are shown in Table 1. Ninety-five percent of the patients had bilateral DDH. Table 2 shows the correlation coefficients between the radiographic measurements. Significant correlations were observed between all groups (P < 0.001). The CE angle had the highest correlation with other radiographic measurements. The JHEQ movement values of patients with cyst formation were significantly lower in comparison to the JHEQ movement values of patients without cyst formation. The JHEQ mental values of patients with cyst formation were also significantly lower in comparison to the JHEQ mental values of patients without cyst formation. The JHEQ pain values of patients with and without cyst formation did not differ to a statistically significant extent. The JHEQ subscale values did not differ according to the presence or absence of osteophytes (Fig. 2). Fig. 3 demonstrates that the JHEQ subscale values were not significantly influenced by the presence or absence of cam or pincer deformity. The relationships between each JHEQ subscale value and the radiographic features were evaluated by a linear regression analysis. We decided to deal with the CE angle as a representative
Table 2 The correlation coefficients (95% confidence intervals) between radiographic measurements.
AHI ADR ARO CEA
ADR
ARO
CEA
Sharp
0.58 (0.43, 0.70) e e e
0.56 (0.67, 0.41) 0.68 (0.78, 0.56) e e
0.86 (0.79, 0.92) 0.71 (0.60, 0.80) 0.76 (0.84, 0.67) e
0.59 (0.70, 0.49) 0.32 (0.49, 0.12) 0.53 (0.36, 0.66) 0.68 (0.78, 0.56)
AHI, Acetabular head index; ADR, Acetabular depth ratio; ARO, Acetabular roof obliquity; CEA, Center-edge (CE) angle; Sharp, Angle of Sharp. All P values < 0.05.
Fig. 2. The JHEQ subscale values for the hip with or without cysts and osteophytes. (a) The JHEQ subscale values in patients with or without cyst formation. (b) The JHEQ subscale values in patients with or without osteophytes. A, radiographic features absent; P, radiographic features present. *P value < 0.05.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
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Y. Takegami et al. / Journal of Orthopaedic Science xxx (xxxx) xxx
Fig. 3. The JHEQ subscale values hips with or without femoroacetabular impingement. (a) The JHEQ subscale values in patients with or without cam deformity. (b) The JHEQ subscale values in patients with or without pincer deformity A, radiographic features absent; P, radiographic features present.
radiological measurement in both the linear regression analysis and the multiple regression analysis because it showed the highest correlation among the radiographic measurements (Table 2). The CE angle was found to be associated with the JHEQ movement score in the linear regression analysis (Table 3). To determine the independent associations of each radiographic feature with the scores on each of the JHEQ subscales, we performed a multiple regression analysis with age, body mass index, sex and the number of affected hip joints (bilateral or unilateral) included as independent variables. The CE angle was found to be independently associated with JHEQ movement (b coefficient 1.72; adjusted R^2 ¼ 0.12) (Table 3).
3. Discussion Our data demonstrated that the CE angle was independently associated with the JHEQ movement value. In a previous large cross-sectional study in Norway, there is no association between CE angle and patient-based assessments such as WOMAC or EQ-5D [20]. However, the participants' background was different from our study. This large cross-sectional study was conducted with healthy men and women aged 19 years with/without hip pain. While, we did this research at a university hospital, the patients who had visited a medical institution at least once with some of complaint about the hip joint. Yasunaga et al. reported that patients
Table 3 Association of each JHEQ subscale with each radiographic evaluation, adjusted for age and gender. Univariate regression coefficient
CEA Cyst Osteophyte Pincer Cam
JHEQ pain (95%CI)
P value
JHEQ movement (95%CI)
P value
JHEQ mental (95%CI)
P value
0.27 (1.7,1.3) 0.59 (2.4,0.91) 0.79 (2.3,0.76) 0.35 (1.83,1.21) 0.01 (1.5,1.5)
0.70 0.42 0.36 0.72 0.98
2.1 (3.6,0.51) 1.71 (3.3,0.19) 1.33 (2.9,0.26) 1.0 (2.6,0.61) 0.2 (1.8,1.6)
0.009 0.030 0.10 0.20 0.82
0.07 (2.1,0.9) 1.69 (3.3,0.17) 0.81 (2.6,0.72) 0.50 (2.1,1.1) 0.30 (1.5,1.9)
0.40 0.03 0.28 0.54 0.75
Multiple regression coefficient
CEA Cyst Osteophyte Pincer Cam
JHEQ pain (95%CI)
P value
JHEQ movement (95%CI)
P value
JHEQ mental (95%CI)
P value
VIF
0.29 (1.7, 1.2) 0.60 (2.4, 0.8) 0.88 (2.4, 0.74) 0.33 (1.8, 1.2) 0.01 (1.5, 1.5)
0.72 0.43 0.31 0.70 0.99
1.71 (3.35, 0.11) 0.92 (2.43, 0.66) 0.48 (2.2, 1.20) 1.4 (2.9, 0.15) 0.31 (1.90, 1.35)
0.04* 0.29 0.66 0.09 0.73
1.2 (2.8, 0.5) 0.96 (2.52, 0.62) 0.11 (1.42, 1.64) 1.1 (2.35, 0.76) 0.56 (1.2, 2.1)
0.20 0.24 0.89 0.44 0.53
1.14 1.12 1.09 1.03 1.33
CI, confidence interval; CEA, center-edge angle; Pincer, presence of pincer deformity; Cam, presence of cam deformity; VIF, variance inflation factor. *P value < 0.05.
Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007
Y. Takegami et al. / Journal of Orthopaedic Science xxx (xxxx) xxx
with severe DDH (preoperative mean CE angle: 1.6 ) who underwent rotational acetabular hip osteotomy had preoperative pain that was tolerable with limited daily activity [21]. Another large retrospective study demonstrated that patients with severe DDH (<5 ) had less activity, as assessed by the UCLA score [22]. These reports implied that the activity of patients with severe DDH might be reduced, which would have led to a decrease in hip pain along with a decline in their daily activity. In a finite element study, it was reported that the von Mises stress on the acetabular side and the femoral head side increased as the CE angle decreased [23]. An evaluation of dGEMRIC for early osteoarthritis of hip using MRI in patients with DDH also showed a correlation between CE angle and signal changes of the dGEMERIC, which suggested cartilage degeneration [24,25]. Thus, the CE angle is involved in the early development of coxarthrosis. Patients who develop pain due to DDH over time to avoid osteoarthritis may limit activities of daily living. It is expected that all patients felt some pain from the time they had went to the local medical institution to the time they visited the university hospital. Moreover, it is known that behavioral restrictions were effective as a conservative treatment for coxalgia in patients with various hip diseases [26e28]. By the time they visited our university hospital, the patients who develop pain due to DDH may limit activities of daily living. There was a significant difference in the JHEQ movement scores of the patients with and without cyst formation. Cyst formation was associated with the initiation and progression of osteoarthritis of the hip [29]. A previous report revealed the cyst formation significantly affected hip pain and impairment [11]. Radiographic cyst formation might indicate the progression of osteoarthritis of the hip. There was no relationship between the FAI findings and PROMs. A retrospective study compared the radiographs of symptomatic patients to those of asymptomatic patients, and found no difference in the prevalence of FAI between the groups [30]. That is, FAI-like morphologic changes may not be related to pain or dysfunction in patients with DDH. This present study is associated with several limitations. False profile views [31], might have been obtained when the anterior coverage of the femoral head was evaluated in the standing position. To prevent excessive radiation exposure, we minimized the imaging studies that were performed to those that were necessary. Second, we did not evaluate labrum tear, bone marrow edema or cartilage degeneration on MRI. These are also causes of hip pain. Third, patients who were referred to our hospital were introduced by local hospitals as patients who required specialized treatment. They might have received conservative treatments including the behavioral restrictions at the local hospitals before visiting our hospital. Details of the previous hospital's conservative treatment were not available, but these may have affected the results. Fourth, this was a hospital-referred cross-sectional study that was performed in a single university hospital. Thus, the causal relationship could not be determined. In conclusion, the CE angle was independently associated with the JHEQ movement value. While no factors were independently associated with JHEQ pain, the JHEQ movement values of DDH patients with cyst formation were lower than those of patients without cyst formation. The FAI findings were not related to the PROMs in DDH patients. These radiographic features may have an application in the assessment of pain and functional impairment in DDH patients. Conflicts of interest None.
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Please cite this article as: Takegami Y et al., The relationship between radiographic findings and patient-reported outcomes in adult hip dysplasia patients: A hospital cross-sectional study, Journal of Orthopaedic Science, https://doi.org/10.1016/j.jos.2019.09.007