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Table of Contents
REVIEW ARTICLE
Year : 2022  |  Volume : 15  |  Issue : 4  |  Page : 171-175

Neonatal screening for developmental dysplasia of the hip Ortolani and Barlow sign accuracy review


Department of Pediatrics and Orthopedics, Hatta Hospital, DHA, Dubai, United Arab Emirates

Date of Submission20-Jul-2022
Date of Acceptance08-Aug-2022
Date of Web Publication22-Dec-2022

Correspondence Address:
Ammar Mohammed Haidar Shehadeh
Hatta Hospital, DHA, Dubai
United Arab Emirates
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/hmj.hmj_61_22

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  Abstract 


Introduction: Developmental dysplasia of the hip (DDH) joint is a preventable cause of disability. If detected early, the result is expected to be a complete improvement. Ortolani and Barlow clinical test is performed early to detect DDH. However, the authors have different opinions regarding the accuracy of the Ortolani and Barlow sign and the need for an ultrasound scan (USS) as a screening tool for DDH. Methods: This review involves a comprehensive search on Medline through PubMed and OVID, then from Google Scholar and article references for any paper or clinical trial that studies the accuracy of Ortolani and Barlow sign against the gold standard USS. Results: Four studies have been included. Sensitivity lied between 7% and 28.3%, specificity between 94.5% and 99.8%, positive predictive value (PPV) 13.6%–85.7% and negative predictive value (NPV) 88.4%–97.7%. However, when the clinical examination was repeated more than twice and the USS at 6 months of age was considered the gold standard, sensitivity improved significantly to reach 84%, specificity 82%, PPV 14% and NPV 99%. The relative risk of DDH in patients with positive OB was between 6.11 and 7.4. Discussion: The OB could be considered a risk factor for DDH. It did not show enough sensitivity to be considered a valid screening test for DDH. Low sensitivity and PPV devalue OB for DDH screening. Alternatively, repeated clinical examination improves the test sensitivity and could be adopted in settings where universal USS is not possible due to lack of funds or experience.

Keywords: Congenital hip dislocation, developmental dysplasia of the hip, Ortolani and Barlow sign


How to cite this article:
Haidar Shehadeh AM, Al Haroun MA, Sammak AK. Neonatal screening for developmental dysplasia of the hip Ortolani and Barlow sign accuracy review. Hamdan Med J 2022;15:171-5

How to cite this URL:
Haidar Shehadeh AM, Al Haroun MA, Sammak AK. Neonatal screening for developmental dysplasia of the hip Ortolani and Barlow sign accuracy review. Hamdan Med J [serial online] 2022 [cited 2023 Feb 1];15:171-5. Available from: http://www.hamdanjournal.org/text.asp?2022/15/4/171/364692




  Introduction Top


Developmental dysplasia of the hip (DDH) is the incomplete, improper or abnormal development of the hip affecting 1.5 of every 1000 life new-borns.[1] It may involve the acetabulum, the proximal femur, the labrum, the capsule and other soft tissue. DDH covers a wide spectrum of hip abnormalities ranging from hip dysplasia to reducible and irreducible hip joint dislocation.

DDH is a treatable cause of disability that should be discovered early. Although conservative treatment is effective in early detected DDH, more complicated surgical management is required in most of the cases detected after the age of 10 months.[2] Moreover, missed DDH may lead to functional disability and Cox arthritis.[3]

The detection of DDH clinical examination in new-born infants was described in 1937 by Ortolani.[4] The routine use of such an examination in all new-born babies, plus the beginning of immediate conservative management in all detected cases promised an end to missed late DDH.[5] Unfortunately, this did not prove to be the case, and although most screens provided an improvement in diagnostic success, late DDH continued to present a diagnostic challenge. This experience was reported by Barlow in 1962 when he described a supplementary clinical test to add to the Ortolani manoeuvre to improve the accuracy of clinical examination.[6]

Combining Ortolani and Barlow procedures is a very useful way for screening. However, ultrasound scan (USS) is considered the gold standard for detecting DDH and is needed as a screening for high-risk babies or as a confirmatory diagnostic method.

This article will review the best available evidence about the value and accuracy of OB in detecting DDH.


  Methods Top


This review focussed on a PICO question (P: neonates, I: Barlow Ortolani, C: Ultrasound, O: DDH). The search was done for any observational study or clinical trial that matched our PICO question on Medline through PubMed and OVID, then from Google Scholar. Mesh terms used: Hip dislocation, congenital, infant and newborn. Free text search for congenital hip dislocation or DDH AND Barlow Ortolani OR hip clinical examination performed. Consequently, references to the found papers were searched for any missing articles. The search was done by the three authors independently; then, the results were matched to assure reliability. Preferred Reporting Items for Systematic Reviews and Meta-Analyses search chart included [Figure 1].
Figure 1: PRISMA flow chart. PRISMS: Preferred Reporting Items for Systematic Reviews and Meta-Analyses, USS: Ultrasound scan, RCT: Randomised controlled trial

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  Results Top


Seven articles were shortlisted comparing USS and clinical examination.[7],[8],[9],[10],[11],[12],[13] However, three studies were excluded;[11],[12],[13] one old study had no clear data,[11] and another two studies compared universal and selective USS without a specific clinical examination group[12],[13] [Table 1].
Table 1: Search results

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Four studies have been included.[7],[8],[9],[10] These studies compared USS and clinical examination either in the same group[7],[9],[10] or between the intervention and the control group.[8] As our review is concerned with the accuracy of the OB examination, the control arm of the Roovers et al. study will be considered alone against the gold standard USS at the age of 6 months.[8]

Study population and design

The first two papers were multicentre studies on all babies born in six hospitals in Italy[7] or in a catchment area in the Netherlands.[8] While the other two studies involved babies born in a single hospital in Turkey[9] or Norway.[10]

All papers described clinical trials comparing clinical examination and USS results on the same group except for Roovers et al. which had an intervention group with USS and a control retrospective group with the standard clinical examination.[8]

Age at ultrasound scan and clinical examination

Clinical examination and OB were done in most papers during the 1st week of life.[7],[9],[10] However, Roovers et al.'s study involved repeated physical examinations as a part of the standard screening program, which extends until the age of 6 months.[8]

Baronciani et al. and Olsen et al. performed USS during the 1st month of life.[7],[10] While Dogruel et al. waited till the age of 4–6 weeks.[9] However, as Roover et al. aimed to follow the outcome of patients in the clinical examination arm, this study performed USS on these control arm patients at the age of 6 months.[8]

Examiners and ultrasound scan radiologist

Clinical examination and OB were performed by an experienced paediatrician in all papers. However, no specific training was mentioned in the studies except for Baronciani et al. where OB screening was done by two doctors after a specific short training course.[7]

USS was performed by an experienced radiologist in the studies by Baronciani et al. and Olsen et al.[7],[10] On the other hand, Roover et al. performed USS by technicians who were trained by Professor Graf or by the project team.[8] This was not clear in Dogruel et al.[9] Hips were classified according to the Graf principle in all studies.

Findings

Baronciani et al.[7] included 4648 neonates. Positive OB results were reported in (233) 5% of newborns. The USS was abnormal in 50% of the cases. The accuracy of the OB was calculated, with the USS the gold standard. The sensitivity of the test was 0.07; the specificity was 0.99 and the positive predictive value (PPV) was 0.35.

The relation between the results of the OB and USS was analysed. Subjects with a positive OB had a relative risk of 7.2 (confidence interval [CI] 4.9–9.6) for type II hip and 27.1 (CI 18.5–38.7) for type III-IV hip when compared to subjects with a negative manoeuvre. The exact number of subjects in relation to OB and USS outcomes was not mentioned in the study.

Roover et al.[8] [Table 2] the control group of clinical examination (2066 neonates) included and calculated accuracy against USS at age of 6 months, which is considered the gold standard. The control group was screened by routine physical examination as part of the programme for child health surveillance at the child health care centre. Babies received USS after 6 months of age to detect any abnormality that might have been missed by the screening. The sensitivity of the clinical examination screening was 84%, specificity 82%, positive predictive value PPV 14% and negative predictive value (NPV) was 99%.
Table 2: Roover et al. results

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Dogruel et al. and Olsen et al.[9],[10] [Table 3], [Table 4], [Table 5] recruited a total of 3541 infants and 4245, respectively. All infants underwent clinical examination and USS. Results were summarised and added. The sensitivity of the OB screening was between 5.7% and 28.3% and the specificity was between 94.5% and 99.8%.
Table 3: Dogruel et al. results

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Table 4: Olsen et al. results

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Table 5: Dogruel + Olsen results

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If positive OB was considered as a risk factor for DDH, then the relative risk respiratory rate (RR) for DDH in cases with positive OB is between 6.11[9] and 7.4[10] this is consistent with the calculated risk of 7.2 in Baronciani et al.'s study.[7] However, when USS was done at the age of 6 months, the relative risk of abnormal USS in patients with positive OB was very high, reaching 23.[8] Accuracy calculations of all papers were summarised [Table 6].
Table 6: All papers accuracy calculations

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  Discussion Top


DDH affects about 1.5 of every 1000 babies.[1] The prognosis of DDH depends on early diagnosis and treatment. Wirth et al. reported that USS for DDH led to a reduction in late presentations, inpatient treatment and surgical treatment.[11] However, the OB examination is an easier, faster and readily available screening tool. It requires shorter time and less training to master. This article reviewed the best available evidence on the reliability and accuracy of early OB screening.

Although highly specific, clinical examination showed low sensitivity and low PPV when compared with USS in most studies. OB failed to detect most of the positive cases by USS, especially mild cases, and positive OB was not a good predictive test for DDH. PPV was only 35% 14% and 13.6% in Baronciani et al., Roover et al. and Dogruel et al., respectively.[7],[8],[9] However, clinical examination accuracy depends on the examiner's experience, training, age at examination, risk factors and the prevalence of DDH.

After summing results from Dogruel et al. and Olsen et al. studies,[9],[10] prevalence of DDH by USS was around 6.4% in concordance with Von Kries et al. findings (5%−7%).[12] On the other hand, the clinical prevalence of abnormal OB was only 466/11327 (4.1%).[9],[10]

Dogruel et al. and Olsen et al. studies were standard single-centre studies with early OB and USS applied to the same group of patients.[9],[10] OB in Dogruel et al. showed low sensitivity of 28.3% and low PPV of 13.6%.[9] However, in Olsen et al.'s study, the examination was more reliable with 85% PPV. This could be explained by the extremely low sensitivity in Olsen et al.'s study (5.7%), as the examination was performed by the only paediatrician in the hospital. This could be explained by a very high threshold for positive OB that was set by this paediatrician depending on his training and experience which has led to higher PPV and lower sensitivity. Nevertheless, OB in both papers had high specificity (94.5%–99.8%) and NPV (88.4%–97.7%).

Baronciani et al.'s study was a relatively old study (1989–1990), with multiple examinations by trained physicians for each baby. OB was positive in 15/1000 of the babies only. The OB was inconsistent and not reproducible between hospitals and different examiners. OB had low sensitivity (7%) and high specificity (99%) in concordance with other studies. Nevertheless, considering OB as a risk factor will increase the risk of DDH 7.2 times (RR).[7]

The method of Roover et al. was totally different as it compared USS with clinical examination in two different groups, then did a USS at the age of 6 months to be considered a gold standard. On average, the children were examined 3.6 times. The high sensitivity of clinical examination in this study could be attributed to repeated examinations at different ages in contrast with the single early OB in other studies. In addition, the late gold standard USS may have omitted the early false-positive USS. This was apparent in the sensitivity of early USS, which was 88.5% only.[8]

Several studies have compared clinical examination and USS as methods of screening infants for DDH. Marks et al. reported that USS for DDH can detect cases of instability not diagnosed at birth by routine clinical examination and in infants who have no risk factors for DDH.[13] Tönnis et al.[14] and Rosenberg et al.[15] reported, respectively, that 52.2% and 50% of the USS pathological hips in their studies had no clinical signs of instability. Similarly, more than 70% of infants with abnormal USS had no clinical findings in this review selected papers (149/208 in Dogruel et al.[9] and 488/518 in Olsen et al.[10]).

On the other hand, Rosenberg et al. reported that in their series of 9,199 newborns, three hips appeared normal on the USS, but were unstable clinically.[16] Riboni et al. reported 12 newborn babies whose hips appeared USS normal at birth but appeared abnormal on USS at 3 months of age.[17] Our papers had a significant number of patients who had abnormal OB with normal USS (372/431 in Dogruel et al.[9] and 5/35 in Olsen et al.[10]).

Authors have different opinions regarding the routine use of the USS to screen all neonates for DDH. Castelein and Sauter reported that 82 hips in their series had falsely negative clinical examination (13%); USS was abnormal, and clinical examination findings were normal. However, after 6 months, although none were treated, DDH developed only in four hips. This indicates that USS may be too sensitive because it also identifies clinically unimportant instability and leads to overtreatment of DDH.[18] Moreover, another study comparing universal with selective USS screens showed nonsignificant differences between the two groups (0.13 and 0.65 per 1000, respectively, P = 0.22).[19] Therefore, this study did not recommend universal USS.

On the other hand, although it may be associated with overtreatment, universal USS reduced the rate of operative procedures for DDH.[12] because the treatment is benign, it is more reasonable to risk overtreatment associated with universal USS than to risk underdiagnosis associated with the clinical examination.[17]

Other authors advised the use of USS in infants with a risk factor or positive DDH clinical examination findings.[20] Similarly, Rosenberg et al. recommended a combined approach of clinical examination and USS to screening for DDH.[15]

In conclusion, according to our findings, OB could be considered a risk factor for DDH, as the RR of DDH in patients with positive OB was between 6.11[9] and 7.4.[10] However, the OB did not show enough sensitivity to be considered a valid screening test for DDH. Low sensitivity and PPV that depend on the setting, risk factors and the training of the examiner, devalue OB for DDH screening. Therefore, it is highly recommended to perform a USS screen at the age of 4–6 weeks, especially for infants with positive OB or risk factors for DDH. Alternatively, repeated clinical examination improves the test sensitivity and could be adopted in settings where universal USS is not possible due to the lack of funds or experience.[8]

Statement of ethics

This is a review article data search that was performed with proper referencing of all cited articles. Ethical approval was not required in accordance with the Dubai Scientific Research Ethics Committee policies.

Data availability statement

All data generated or analysed during this study are included in this article. Further enquiries can be directed to the corresponding author.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Karmazyn BK, Gunderman RB, Coley BD, Blatt ER, Bulas D, Fordham L, et al. ACR Appropriateness Criteria® on developmental dysplasia of the hip-child. J Am Coll Radiol 2009;6:551-7.  Back to cited text no. 1
    
2.
Price KR, Dove R, Hunter JB. Current screening recommendations for developmental dysplasia of the hip may lead to an increase in open reduction. Bone Joint J 2013;95-B: 846-50.  Back to cited text no. 2
    
3.
Wedge JH, Wasylenko MJ. The natural history of congenital dislocation of the hip: A critical review. Clinical orthopaedics and related research 1978;(137):154-62.  Back to cited text no. 3
    
4.
Ortolani M. Un segno poco noto e la sua importanza per la diagnosi precoce di pre lUSGsazione congenita dell'anca. Pediatria 1937;45:129-36.  Back to cited text no. 4
    
5.
Von Rosen S. Diagnosis and treatment of congenital dislocation of the hip joint in the new-born. J Bone Joint Surg Br 1962;44B: 284-91.  Back to cited text no. 5
    
6.
Barlow TG. Early diagnosis and treatment of congenital dislocation of the hip. J Bone Joint Surg Br 1962;44:292-301.  Back to cited text no. 6
    
7.
Baronciani D, Atti G, Andiloro F, Bartesaghi A, Gagliardi L, Passamonti C, et al. Screening for developmental dysplasia of the hip: From theory to practice. Collaborative Group DDH Project. Pediatrics 1997;99:E5.  Back to cited text no. 7
    
8.
Roovers EA, Boere-Boonekamp MM, Castelein RM, Zielhuis GA, Kerkhoff TH. Effectiveness of ultrasound screening for developmental dysplasia of the hip. Arch Dis Child Fetal Neonatal Ed 2005;90:F25-30.  Back to cited text no. 8
    
9.
Dogruel H, Atalar H, Yavuz OY, Sayli U. Clinical examination versus ultrasonography in detecting developmental dysplasia of the hip. Int Orthop 2008;32:415-9.  Back to cited text no. 9
    
10.
Olsen SF, Blom HC, Rosendahl K. Introducing universal ultrasound screening for developmental dysplasia of the hip doubled the treatment rate. Acta Paediatr 2018;107:255-61.  Back to cited text no. 10
    
11.
Wirth T, Stratmann L, Hinrichs F. Evolution of late presenting developmental dysplasia of the hip and associated surgical procedures after 14 years of neonatal ultrasound screening. J Bone Joint Surg Br 2004;86:585-9.  Back to cited text no. 11
    
12.
von Kries R, Ihme N, Altenhofen L, Niethard FU, Krauspe R, Rückinger S. General ultrasound screening reduces the rate of first operative procedures for developmental dysplasia of the hip: A case-control study. J Pediatr 2012;160:271-5.  Back to cited text no. 12
    
13.
Marks DS, Clegg J, al-Chalabi AN. Routine ultrasound screening for neonatal hip instability. Can it abolish late-presenting congenital dislocation of the hip? J Bone Joint Surg Br 1994;76:534-8.  Back to cited text no. 13
    
14.
Tönnis D, Storch K, Ulbrich H. Results of newborn screening for CDH with and without sonography and correlation of risk factors. J Pediatr Orthop 1990;10:145-52.  Back to cited text no. 14
    
15.
Rosendahl K, Markestad T, Lie RT. Ultrasound screening for developmental dysplasia of the hip in the neonate: The effect on treatment rate and prevalence of late cases. Pediatrics 1994;94:47-52.  Back to cited text no. 15
    
16.
Rosenberg N, Bialik V, Norman D, Blazer S. The importance of combined clinical and sonographic examination of instability of the neonatal hip. Int Orthop 1998;22:185-8.  Back to cited text no. 16
    
17.
Riboni G, Bellini A, Serantoni S, Rognoni E, Bisanti L. Ultrasound screening for developmental dysplasia of the hip. Pediatr Radiol 2003;33:475-81.  Back to cited text no. 17
    
18.
Castelein RM, Sauter AJ. Ultrasound screening for congenital dysplasia of the hip in newborns: Its value. J Pediatr Orthop 1988;8:666-70.  Back to cited text no. 18
    
19.
Holen KJ, Tegnander A, Bredland T, Johansen OJ, Saether OD, Eik-Nes SH, et al. Universal or selective screening of the neonatal hip using ultrasound? A prospective, randomised trial of 15,529 newborn infants. J Bone Joint Surg Br 2002;84:886-90.  Back to cited text no. 19
    
20.
Clarke NM, Clegg J, Al-Chalabi AN. Ultrasound screening of hips at risk for CDH. Failure to reduce the incidence of late cases. J Bone Joint Surg Br 1989;71:9-12.  Back to cited text no. 20
    


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