Trends in congenital clubfoot prevalence and co-occurring anomalies during 1994-2021 in Denmark: a nationwide register-based study.

BACKGROUND: Congenital talipes equinovarus (clubfoot) is a common musculoskeletal anomaly, with a suspected multifactorial etiopathogenesis. Herein, we used publicly available data to ascertain liveborn infants with clubfoot delivered in Denmark during 1994-2021, and to classify co-occurring congenital anomalies, estimate annual prevalence, and compare clubfoot occurrence with maternal smoking rates, a commonly reported risk factor. Characterizing this nationwide, liveborn cohort provides a population-based resource for etiopathogenic investigations and life course surveillance. METHODS: This case-cohort study used data from the Danish National Patient Register and Danish Civil Registration System, accessed through the publicly available Danish Biobank Register, to identify 1,315,282 liveborn infants delivered during 1994-2021 in Denmark to Danish parents. Among these, 2,358 infants (65.1% male) were ascertained with clubfoot and classified as syndromic (co-occurring chromosomal, genetic, or teratogenic syndromes) and nonsyndromic (isolated or co-occurring multiple congenital anomalies [MCA]). Annual prevalence estimates and corresponding 95% confidence intervals (CIs) for children with nonsyndromic clubfoot were estimated using Poisson regression and compared with population-based, maternal annual smoking rates obtained from publicly available resources. RESULTS: Infants most often presented with nonsyndromic clubfoot (isolated = 88.6%; MCA = 11.4%); limb and heart anomalies were the most frequently identified MCAs. Prevalence (per 1,000 liveborn infants) was 1.52 (CI 1.45-1.58) for isolated and 0.19 (CI 0.17-0.22) for MCA clubfoot. Prevalence estimates for both isolated and MCA clubfoot remained relatively stable during the study period, despite marked decreases in population-based maternal smoking rates. CONCLUSIONS: From 1994 to 2021, prevalence of nonsyndromic clubfoot in Denmark was relatively stable. Reduction in population-level maternal smoking rates did not seem to impact prevalence estimates, providing some support for the suspected multifactorial etiopathogenesis of this anomaly. This nationwide, liveborn cohort, ascertained and clinically characterized using publicly available data from the Danish Biobank Register, provides a population-based clinical and biological resource for future etiopathogenic investigations and life course surveillance.

[Clinical characteristics analysis of 22 062 patients of foot and ankle deformity from QIN Sihe Orthopaedic Surgery Database between May 25, 1978 and December 31, 2020].

Objective: Based on the clinical data of patients with foot and ankle deformities in the QIN Sihe Orthopaedic Surgery Database, to analyze the characteristics and treatment strategies of foot and ankle deformities, and provide a basis for clinical decision-making. Methods: A total of 22 062 patients with foot and ankle deformities who received orthopedic surgery between May 25, 1978 and December 31, 2020 were searched in the QIN Sihe Orthopedic Surgery Database. The gender, age at operation, regional distribution, etiology, type of deformity, operation method, postoperative fixation method, and other information were collected. Results: Among the 22 062 patients, there were 13 046 males (59.13%) and 9 016 females (40.87%); the age at operation ranged from 1 to 77 years, with a median of 17 years, and 20 026 cases (90.77%) were aged 5 to 40 years. The patients came from 32 provinces, municipalities, and autonomous regions across the China and 5 countries including India and the United States, et al. The etiology and diseases type covered 154 kinds (of which sequelae of poliomyelitis, cerebral palsy, spina bifida and tethered spinal cord, congenital equinovarus foot, post-traumatic foot and ankle deformity, and Charcot-Marie-Tooth disease accounted for the highest proportion). The types of deformities included varus foot, equinus foot, valgus foot, talipes calcaneus, equinocavus, high arched foot, claw toe, and flail foot. Surgical methods included tendon lengthening, soft tissue release, tendon transposition, osteotomy orthopedics, and ankle arthrodesis. The 36 620 operations were performed, including 11 561 cases of hip, knee, and lower leg operations to correct the foot and ankle deformities. Postoperative fixation methods included Ilizarov external fixator in 2 709 cases (12.28%), combined external fixator in 3 966 cases (17.98%), and plaster or brace fixation in 15 387 cases (69.74%). Conclusion: Male patients with foot and ankle deformities account for a large proportion, and the population distribution is mainly adolescents, with a wide distribution of regions, causes and diseases, and talipes equinovarus and varus foot are the main types of deformities. Foot and ankle deformities are often combined with deformities of other parts of the lower limb, which requires a holistic treatment concept. The application of foot soft tissue and bone surgery combined with Ilizarov external fixator and combined external fixators provides a guarantee for the correction of complex foot and ankle deformities.

Comparative evaluation and analysis of outcomes in non-idiopathic and idiopathic clubfeet with Ponseti method at a tertiary care centre of a developing country.

BACKGROUND: Idiopathic clubfoot (congenital talipes equinovarus, CTEV) is being managed worldwide by Ponseti method with high success rates, while for non-idiopathic clubfoot surgical interventions is being widely used with variable results. This study evaluated the effectiveness of Ponseti method in non-idiopathic clubfoot and compared the results with idiopathic clubfoot. METHODS: The paper evaluated the epidemiological incidence and demographic profile of non-idiopathic clubfoot in a tertiary centre of developing country. A total of 108 subjects with 85 having idiopathic (group I;125 feet) and 23 having non-idiopathic variety (group II;34 feet) were managed with Ponseti method and were followed for a mean duration of 38.33 (12-62) and 36.27 months (12-58) in group I and II respectively. The most common associations were meningomyelocele (MMC/spina bifida,5), arthrogryposis multiplex congenita (AMC,4), developmental dysplasia of hip (DDH,3) and Down's syndrome and amniotic band syndrome (2 each). RESULTS: Primary correction was achieved in both the groups in 98% and 87% in group I and II respectively, while recurrences of at least one deformity was observed in 11 (9%) and 12 (40%) feet in group I and II respectively. Favourable outcomes were noticed in 22 (65%) feet in non-idiopathic group and 12 feet (35%) underwent extensive soft tissue release as compared to 3 feet (2.4%) in idiopathic variety. CONCLUSION: Deformities improved significantly in non-idiopathic clubfeet with Ponseti methods although complete correction was not possible. Extensive surgical interventions were reduced in up to 35% feet in non-idiopathic variety and hence, it is recommended as primary treatment for all variety of clubfeet, irrespective of their etiology.

Impact of the volume of the myelomeningocele sac on imaging, prenatal neurosurgery and motor outcomes: a retrospective cohort study.

To investigate the association of the myelomeningocele (MMC) volume with prenatal and postnatal motor function (MF) in cases who underwent a prenatal repair. Retrospective cohort study (11/2011 to 03/2019) of 63 patients who underwent a prenatal MMC repair (37 fetoscopic, 26 open-hysterotomy). At referral, measurements of the volume of MMC was performed based on ultrasound scans. A large MMC was defined as greater than the optimal volume threshold (ROC analysis) for the prediction of intact MF at referral (2.7 cc). Prenatal or postnatal intact motor function (S1) was defined as the observation of plantar flexion of the ankle based on ultrasound scan or postnatal examination. 23/63 participants presented a large MMC. Large MMC lesions was associated with an increased risk of having clubfeet by 9.5 times (CI%95[2.1-41.8], p < 0.01), and reduces the chances of having an intact MF at referral by 0.19 times (CI%95[0.1-0.6], p < 0.01). At birth, a large MMC reduces the chance of having an intact MF by 0.09 times (CI%95[0.01-0.49], p < 0.01), and increases the risk of having clubfeet by 3.7 times (CI%95[0.8-18.3], p = 0.11). A lower proportion of intact MF and a higher proportion of clubfeet pre- or postnatally were observed in cases with a large MMC sac who underwent a prenatal repair.Trial registration: Clinicaltrials.gov NCT02230072 and NCT03794011 registered on September 3rd, 2014 and January 4th, 2019.

Cross-cultural adaptation, validity and reliability of Turkish version of Oxford Ankle Foot Questionnaire for children with congenital talipes equinovarus.

BACKGROUND: The purpose of the study was to evaluate the reliability and validity of the Turkish version of the Oxford Ankle Foot Questionnaire (OxAFQ) to provide cultural adaptation. METHODS: This study involved translation, back translation, and cross-cultural adaptation. Forty-nine patients with congenital talipes equinovarus were evaluated using the Turkish version of OxAFQ. Turkish version of the Childhood Health Assessment Questionnaire (CHAQ) was used as a gold standard to validate the Turkish version of the OxAFQ. The validation was assessed with Spearman correlation analysis by using CHAQ. The reliability of the questionnaire was assessed with Cronbach alpha (internal consistency) and exploratory factor analysis. RESULTS: High validity was found between OxAFQ and CHAQ (r = -0.422-0.292) (p < 0.01). Reliability analysis showed that OxAFQ had a high level of Cronbach alpha (α = 0.88-0.96) and internal consistency (ICC = 0.90-0.96). CONCLUSION: The Turkish version of OxAFQ is a valid, reliable and useful quality of life questionnaire in patients with congenital talipes equinovarus and it is proper for use by health professionals and researchers.

The role of clubfoot training programmes in low- and middle-income countries: a systematic review.

While adoption of the Ponseti method has continued gradually, its use to manage patients with congenital talipes equinovarus (CTEV) has been limited in low- and middle-income countries (LMICs) for a number of reasons including a lack of clinical training on technique and lack of appropriate clinical equipment. There are a frequent number of emerging studies that report on the role of clubfoot training programmes; however, little is known in regard to cumulative benefits.A systematic review was undertaken through Medline, the Cochrane Library and Web of Science for studies analysing clubfoot training programmes. There were no limitations on time, up until the review was commenced on January 2020. The systematic review was registered with PROSPERO as 165657. Ten articles complied with the inclusion criteria and were deemed fit for analysis. Training programmes lasted an average of 2-3 days. There was a reported increase in knowledge of applying the Ponseti method in managing clubfoot by participants (four studies P < 0.05). Skill retention was examined by multiple choice (MCQ) examination style questions before and after the training programme in two studies; both showed an improvement (MCQ answers improved from 59% to 73%). All studies showed an improvement in participants' self-reported understanding of the Ponseti method and confidence in its use in future practice (P < 0.05). There were improved benefits of knowledge and clinical application of the Ponseti method by participants in the programmes in all studies examined. However, there was a significant lack of follow-up and exploration of long-term effects of these programmes. Implementing training programmes based on perceived benefits rather than actual long-term benefits may have a negative impact on healthcare delivery and patient management in LMICs.

Prevalence of Hip Dysplasia and Associated Conditions in Children Treated for Idiopathic Early-onset Scoliosis-Don't Just Look at the Spine.

BACKGROUND: Hip dysplasia, congenital muscular torticollis, plagiocephaly, and metatarsus adductus are known to be associated. The etiology of infantile idiopathic scoliosis and its association with the aforementioned conditions is unknown. This study reviews a series of infantile scoliosis patients to address this gap. METHODS: The medical records of all patients treated with casting for early-onset scoliosis (EOS) from 2001 to 2016 were retrospectively reviewed. Inclusion criteria were a diagnosis of idiopathic EOS and age below 4 years at the time of the first cast. Demographic information, comorbid conditions, and radiographic measurements including Cobb angle and acetabular index (AI) were collected. The first acceptable anteroposterior pelvis radiograph for each patient was measured. An AI≥30 degrees was defined as hip dysplasia. A measurement between 25 and 30 degrees was defined as a "hip at risk." RESULTS: Between 2001 and 2016, 142 patients were treated with casting. Eighty-one patients met the inclusion criteria. The mean age at the first cast was 19.3 (±7.5) months and the mean Cobb angle was 53.6 (±18.8) degrees. There was no significant correlation between Cobb angle and AI. Nine patients met radiographic criteria for hip dysplasia (11.1%), only 4 of whom had been previously diagnosed. Thirty-six patients (44.4%) met the criteria of having at least 1 hip "at risk" of hip dysplasia. Ten patients (12.3%) had been diagnosed with torticollis and 13 patients (16.0%) with plagiocephaly. Three patients (3.7%) had been diagnosed with metatarsus adductus or clubfoot. In total, 30.9% of patients (25/81) had at least one of the above comorbid conditions. CONCLUSIONS: In a large group of children treated for idiopathic EOS, we found a high prevalence of commonly associated conditions-hip dysplasia, torticollis, plagiocephaly, metatarsus adductus, and clubfoot. In 6.2% of our sample, a diagnosis of hip dysplasia was not made in a timely manner despite routine radiographic spine follow-up. With increasing subspecialization within pediatric orthopaedics, surgeons need to maintain vigilance in assessing the entire child. LEVEL OF EVIDENCE: Level IV.

HOXA9 rs3801776 G>A polymorphism increases congenital talipes equinovarus risk in a Chinese population.

BACKGROUND: Congenital talipes equinovarus (CTEV) is the most common congenital deformity in children, and muscular dysplasia plays a potential role in the etiology of CTEV. Notably, previous studies have found that HOXA9 rs3801776 and TPM2 rs2025126 genetic polymorphisms play important roles in regulating muscle development in Caucasian children; however, there is a lack of investigations conducted in Chinese children. METHODS: We conducted a hospital-based, case-control study of 189 children with CTEV and 457 CTEV-free children aiming to examine the associations between these two polymorphisms and CTEV susceptibility. The rs3801776 (G>A) and rs2025126 (G>A) polymorphisms were genotyped using TaqMan. Odds ratios (ORs) and 95% confidence intervals (CIs) were calculated to measure the associations between the selected polymorphisms and CTEV susceptibility. RESULTS: We found that rs3801776A was associated with increased CTEV risk (GA versus GG: adjusted OR = 1.81, 95% CI = 1.22-2.69, p = 0.0031; AA versus GG: adjusted OR = 2.19, 95% CI = 1.28-3.73, p = 0.0041; GA/AA versus GG: adjusted OR = 1.89, 95% CI = 1.29-2.76, p = 0.0010). In a stratified analysis, the risk effect of rs3801776 GA/AA was observed in both unilateral and bilateral patients. CONCLUSIONS: The present study suggests that the rs3801776 G>A polymorphism is associated with CTEV risk in Chinese children; however, this conclusion should be validated in larger studies.

Relapse Rates in Patients with Clubfoot Treated Using the Ponseti Method Increase with Time: A Systematic Review.

BACKGROUND: The Ponseti method is the preferred technique to manage idiopathic clubfoot deformity; however, there is no consensus on the expected relapse rate or the percentage of patients who will ultimately require a corrective surgical procedure. The objective of the present systematic review was to determine how reported rates of relapsed deformity and rates of a secondary surgical procedure are influenced by each study's length of follow-up. METHODS: A comprehensive literature search using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines was performed to identify relevant articles. The definition of relapse, the percentage of patients who relapsed, the percentage of feet that required a surgical procedure, and the mean duration of follow-up of each study were extracted. Pearson correlations were performed to determine associations among the following variables: mean follow-up duration, percentage of patients who relapsed, percentage of feet that required a joint-sparing surgical procedure, and percentage of feet that required a joint-invasive surgical procedure. Logarithmic curve fit regressions were used to model the relapse rate, the rate of joint-sparing surgical procedures, and the rate of joint-invasive surgical procedures as a function of follow-up time. RESULTS: Forty-six studies met the inclusion criteria. Four distinct definitions of relapse were identified. The reported relapse rates varied from 3.7% to 67.3% of patients. The mean duration of follow-up was strongly correlated with the relapse rate (Pearson correlation coefficient = 0.44; p < 0.01) and the percentage of feet that required a joint-sparing surgical procedure (Pearson correlation coefficient = 0.59; p < 0.01). Studies with longer follow-up showed significantly larger percentages of relapse and joint-sparing surgical procedures than studies with shorter follow-up (p < 0.05). CONCLUSIONS: Relapses have been reported to occur at as late as 10 years of age; however, very few studies follow patients for at least 8 years. Notwithstanding that, the results indicated that the rate of relapse and percentage of feet requiring a joint-sparing surgical procedure increased as the duration of follow-up increased. Longer-term follow-up studies are required to accurately predict the ultimate risk of relapsed deformity. Patients and their parents should be aware of the possibility of relapse during middle and late childhood, and, thus, follow-up of these patients until skeletal maturity may be warranted. LEVEL OF EVIDENCE: Therapeutic Level IV. See Instructions for Authors for a complete description of levels of evidence.

The incidence of biphalangeal fifth toe: Comparison of normal population and patients with foot deformity.

BACKGROUND: Pedal biphalangism, which was also defined as symphalangism, is seen at a frequency that cannot be ignored; however, no study can be found in the literature evaluating biphalangism in normal population in comparison to those who have foot disorders. The aim of this study was to evaluate the incidence of the pedal fifth toe symphalangism in normal population and in patients with foot deformity including hallux valgus, pes planus, pes cavus, and pes equinovarus. We hypothesized that pedal fifth toe symphalangism may be a predisposing factor or an accompanying structural variation for foot deformity. MATERIALS: Patients admitted to the emergency department of our center in October and November 2016 were defined as the control group, and patients with the diagnosis of hallux valgus, pes planus, pes cavus, and pes equinovarus treated between 2011 and 2016 in our department were defined as the foot deformity group. Individuals who had anteroposterior, oblique, and lateral radiographs of foot were included in the study. RESULTS: One thousand and four patients participated in the cross-sectional observational study. Biphalangeal fifth toe was found in 328 of 1004 (32.7%) patients. In foot deformity group ( n = 672), 222 patients (33%) had biphalangeal fifth toe. In the control group, 106 (31.9%) of the 332 patients had biphalangeal fifth toe. There was no statistically significant difference in the incidence of biphalangeal fifth toe between the two groups ( p = 0.72). CONCLUSIONS: According to the results of this study, biphalangeal fifth toe is a common pedal anatomical variant seen approximately in one-third of the population who have either foot deformity or not. This information may be valuable for podiatrist undertaking the conservative or surgical treatment of fifth toe-related disorders.

Clubfoot Etiology: A Meta-Analysis and Systematic Review of Observational and Randomized Trials.

BACKGROUND: Clubfoot is a common congenital anomaly with multiple potential risk factors. Identification of modifiable risk factors may minimize future incidence of clubfoot. The aim of this meta-analysis was to systematically review and analyze the best clinical evidence regarding risk factors associated with clubfoot. METHODS: Medline, Embase, and Cochrane databases were systematically searched from 1967 to May 11, 2016 for studies reporting risk factors for clubfoot. Randomized trials and observational studies were eligible for inclusion, and assessed in duplicate. Study quality was assessed with the Newcastle-Ottawa Scale or Cochrane risk of bias tool; low quality studies were excluded, all randomized trials were included. Two reviewers extracted data independently. This meta-analysis was conducted in accordance with PRISMA guidelines. Pooled effect estimates for the odds of clubfoot were calculated using random or fixed-effects models based on heterogeneity. RESULTS: Forty-two studies (28 case-control, 10 cohort, 4 randomized trials) comprising 31,844 clubfoot cases and 6,604,013 controls were included. Risk factors associated with increased odds of clubfoot included maternal smoking [odds ratio (OR)=1.65; 95% confidence interval (CI), 1.54-1.78], paternal smoking (OR=1.72; 95% CI, 1.05-2.84), maternal body mass index >30 (OR=1.46; 95% CI, 1.29-1.65), family history (OR=7.80; 95% CI, 4.04-15.04), amniocentesis (OR=2.08; 95% CI, 1.34-3.21), selective serotonin reuptake inhibitor exposure (OR=1.78; 95% CI, 1.34-2.37) maternal single status (OR=1.17; 95% CI, 1.11-1.23), gestational diabetes (OR=1.40; 95% CI, 1.13-1.72), nulliparity (OR=1.32; 95% CI, 1.19-1.45), male sex (OR=1.68; 95% CI, 1.48-1.94), and aboriginal Australian race (OR=2.35; 95% CI, 1.63-3.38). CONCLUSIONS: Smoking, maternal obesity, family history, amniocentesis, and some selective serotonin reuptake inhibitor exposures are the most clinically relevant exposures associated with increased odds of clubfoot, with family history representing the greatest risk. Recognition of modifiable risk factors may help in counseling patients, and minimizing clubfoot incidence. LEVEL OF EVIDENCE: Level II.

Descriptive epidemiology of clubfoot in Romania: a clinic-based study.

OBJECTIVE: Congenital clubfoot affects 1 per 1000 live births per year in Romania. To date, no epidemiological studies have been conducted in this country to assess risk factors associated with the deformity. The aim of this study was to evaluate specific environmental and socio-demographic factors that may increase the risk of an infant to be born with clubfoot. PATIENTS AND METHODS: A descriptive clinic-based study over a twelve-week period was conducted using structured questionnaires given to biological parents of clinically confirmed clubfoot and control subjects. 62 parents of probands and 66 parents of control patients were enrolled for risk factor questionnaires. Phenotypic data from clubfoot children was also collected. RESULTS: We found that males were twice as likely to have clubfoot and half of clubfoot subjects were affected bilaterally. There was no significant difference in the rate of left versus right clubfoot. Infant and maternal characteristics showing a strong association with clubfoot included breech presentation and old maternal age at conception. CONCLUSIONS: Our results support reported literature data that males are two times as likely to have clubfoot which indicates a genetic influence. Previous reports suggest clubfoot babies are born to young mothers but in Romania advanced maternal age (≥ 35 years) was an indicator which may suggest genetic influence. This clinic-based study does not support previously recorded data of a positive association for maternal or household smoking. Data from this Romanian population also does not support previous data suggesting strong associations with maternal diabetes.

Incidence and type of foot deformities in patients with spina bifida according to level of lesion.

AIM: The previously suggested association between the incidence of high-level foot deformity and muscle imbalance is no longer supported, when evaluated independent from motor and sensory loss and level of lesion, by current studies. The purpose of this study was to evaluate the association between level of lesion and foot deformity. METHODS: Of 545 patients, a total of 136 (272 feet) patients admitted to the spina bifida clinic between 2010 and 2014 were included in this study. Levels of all lesions were evaluated using initial operation data, the motor-sensory exams, and direct radiography. All patients were categorized into four different groups: Thoracic region (group 1), high-level lumbar-L1-2 region (group 2), mild and lower lumbar regions (L3-4-5) (group 3), and Sacral region (group 4). RESULTS: The mean follow-up time was 34.9 months (range 8-176 months). Group 1, group 2, group 3, and group 4 included 24 (17.6%), 14 (10.3%), 19 (14%), and 79 (58.1%) patients with regards to level of lesion, respectively. The incidences of foot deformity were 85.4, 85.7, 81.5, and 50.6% in groups 1, 2, 3, and 4, respectively. Of all patients, 22% (61 feet) had clubfoot, 16% (44 feet) pes cavus, 10% (26 feet) pes valgus, 6% (17 feet) isolated equinus, 6 % (17 feet) pes calcaneus, and 5% (13 feet) metatarsus adductus. Patients without a foot deformity (81% of normal feet) usually had a lesion at the sacral level (p ≤ 0.05). On the other hand, isolated equinus (70%) and clubfoot (49%) deformities were mostly observed in spinal lesions (p > 0.05). The incidence of pes calcaneus, pes valgus, and adductus deformities inclined as the lesion level decreased (p > 0.05). CONCLUSION: In this study, it was concluded that foot deformities were directly related to the level of lesion. The comparison of higher and lower level lesions revealed that the types of foot deformity differed significantly. The muscle imbalance due to spina bifida was not sufficient to explain the pathology. On the other hand, the level of spinal lesion is an important factor for the type of deformity.

Factors Associated With Increased Risk of Clubfoot: A Norwegian National Cohort Analysis.

BACKGROUND: Previous studies investigating the causes of clubfoot have shown conflicting results, potentially because of retrospective study designs or incomplete assessment of causative factors. The study aim was to examine risk factors for clubfoot in a large prospective Norwegian cohort. METHODS: Exposures prior and during pregnancy were identified through the Norwegian Mother and Child Cohort Study (MoBa) conducted by the Norwegian Institute of Public Health. This was linked to the Norwegian Medical Birth Registry, which provided clubfoot diagnosis through ICD-10 code Q66.0. Logistic regression analysis investigated associations between potential risk factors and development of clubfoot. RESULTS: A total of 121 clubfoot cases were identified; 1.1 per 1000 births. Parental diagnosis of clubfoot [odds ratio (OR): 31.5; 95% confidence interval (CI):9.61-103.3] and cigarette smoking, both in the three months prior to pregnancy (OR:1.82; 95%CI:1.05-3.18) and the first trimester (OR:2.67; 95% CI:1.28-5.55) were associated with clubfoot. Infants with clubfoot had greater solvent exposure (OR:1.66; 95% CI:1.00-2.76). Oligohydramnios, parental age, parental education, parity, maternal anxiety or depression, alcohol use, season of birth did not have statistically significant associations. CONCLUSIONS: In addition to parental diagnosis of clubfoot, results confirm the previously reported association between clubfoot and smoking, and counter previous evidence supporting season of birth, parental education, and other risk factors. Further studies are needed to investigate solvent exposure as a risk factor for clubfoot. Exposure to smoke and solvents can be controlled; this study highlights the importance of public health initiatives to limit these exposures both during pregnancy and in those considering conceiving in the future.

Comparison of web versus interview participants in a case-control study.

PURPOSE: To evaluate whether selection bias might be introduced because of differential self-selection into an internet-based case-control study. METHODS: We compared sociodemographic factors and behavioral factors for cases and controls who opted for their first entrée into the study be via the internet (Web group) versus the telephone (Phone group), using data from a study on clubfoot. RESULTS: Overall, 200 of 1,825 mothers were in the Web group and were more likely to be non-Hispanic White, nonsmokers, more educated, wealthier, and took ≥7 medications/vitamins in pregnancy. These differences were greater for case mothers, introducing the potential for selection bias. Maternal smoking is an established risk factor for clubfoot and was confirmed in the Phone group (OR = 1.61) but was not observed in the Web group (OR = 1.15). CONCLUSION: Our results suggest there is potential for selection bias if enrollment is solely internet based for a case-control study.

Kite versus Ponseti Method in the Treatment of 235 Feet With Idiopathic Clubfoot: Results of a Single Romanian Medical Center.

Congenital idiopathic clubfoot (CC) represents the fifth common most congenital malformation which may be treated conservatively or by surgery. In this article, we present the results obtained in our clinic after conservative therapy performed with 2 methods.A total of 235 consecutive feet (161 patients) were conservatively treated using Kite (n = 129) and Ponseti method (n = 106). The Dimeglio score was determined before and at 6 months after treatment to compare the 2 methods. All of the patients were treated in their first week of life.CC was more frequently diagnosed in males (n = 93; 57.76%), bilaterality being seen in 45.96% of the patients (n = 74). Although before therapy the Dimeglio score was similar in both groups (P = 0.85), it was significantly improved in patients treated by Ponseti method (P = 0.005). Duration of therapy was also longer in patients from Kite versus Ponseti group (20 vs 11 weeks). Failure of orthopedic treatment was more frequent in Kite group (30.32% vs 8.49% of the patients) and the relapses rate at 6 months was also higher (35.65% vs 11.32%).The conservative method used to treat the CC should be adapted on the patient's age and Ponseti method seems to be the most effective type of treatment used for patients treated in their first week of life. Dimeglio score can be successfully used for evaluation of these children. This is the eighth published study that compare the efficacy of Kite versus Ponseti method.

The prevalence of developmental dysplasia of the hip in idiopathic clubfoot: a systematic review and meta-analysis.

PURPOSE: Developmental dysplasia of the hip (DDH) and idiopathic clubfoot are both common paediatric orthopaedic conditions. There is conflicting evidence in the literature as to whether the prevalence of DDH is higher in children with idiopathic clubfoot. The aim of our study was to systematically review the literature and determine the prevalence of DDH in idiopathic clubfoot. METHODS: We searched several databases from 1946 to 2014 for observational studies that reported the prevalence of DDH in idiopathic clubfoot. We performed a meta-analysis using random effects model to pool the prevalence of DDH in idiopathic clubfoot. We also investigated the imaging modality utilised to diagnose and treat DDH in idiopathic clubfoot. Descriptive, quantitative and qualitative data were extracted. RESULTS: Twelve studies (ten case series, one retrospective cohort and one retrospective case-control) were eligible for the meta-analysis, with a total of 2,549 children with idiopathic clubfoot including 83 with DDH. The pooled prevalence estimate of DDH in children with idiopathic clubfoot was 4.1% (95% CI, 2.1-7.7%). There was a substantial amount of between study heterogeneity (I (2) = 87.1%, p < 0.001) due to variability between studies rather than chance variability. Sensitivity analyses showed that the pooled prevalence estimate was 3.5% (95% CI, 1.1-10.8 %) and 3.8% (95% CI, 1.0-13.7%) for studies utilising ultrasound as the imaging modality of diagnosis and treatment with a Pavlik harness respectively. CONCLUSIONS: The overall pooled prevalence of DDH in idiopathic clubfoot is similar to the normal population. The cumulative evidence at present does not indicate an association between DDH and idiopathic clubfoot. Therefore, we do not recommend routine screening for DDH in children with idiopathic clubfoot. However, future retrospective and/or prospective cohort studies of better quality will be able to answer this question with more certainty.

Maternal cigarette, alcohol, and coffee consumption in relation to risk of clubfoot.

BACKGROUND: Clubfoot is associated with maternal cigarette smoking in several studies, but it is not clear if this association is confined to women who smoke throughout the at-risk period. Maternal alcohol and coffee drinking have not been well studied in relation to clubfoot. METHODS: The present study used data from a population-based case-control study of clubfoot conducted in Massachusetts, New York, and North Carolina from 2007 to 2011. Mothers of 646 isolated clubfoot cases and 2037 controls were interviewed about pregnancy events and exposures, including the timing and frequency of cigarette smoking, alcohol intake, and coffee drinking. RESULTS: More mothers of cases than controls reported smoking during early pregnancy (28.9% vs. 19.1%). Of women who smoked when they became pregnant, those who quit in the month after a first missed period had a 40% increase in clubfoot risk and those who continued to smoke during the next 3 months had more than a doubling in risk, after controlling for demographic factors, parity, obesity, and specific medication exposures. Adjusted odds ratios for women who drank >3 servings of alcohol or coffee per day throughout early pregnancy were 2.38 and 1.77, respectively, but the numbers of exposed women were small and odds ratios were unstable. CONCLUSIONS: Clubfoot risk appears to be increased for offspring of women who smoke cigarettes, particularly those who continue smoking after pregnancy is recognisable, regardless of amount. For alcohol and coffee drinkers, suggested increased risks were only observed in higher levels of intake.

Congenital talipes equinovarus: frequency of associated malformations not identified by prenatal ultrasound.

OBJECTIVES: To establish the frequency of prenatally undetected associated malformations (identified at birth) in infants with apparent "isolated" club foot deformity. METHODS: A cohort study of all infants with unilateral or bilateral club foot deformity identified at birth among 311 480 infants surveyed between 1972 and 2012 at Brigham and Women's Hospital in Boston. Those with talipes equinovarus were divided into "isolated" and "complex", based on the findings in examination and by chromosome analysis. RESULTS: One hundred and forty-two infants had "isolated" talipes equinovarus (TEV), and 66 had the "complex" type. Six (4.2%) of the 142 infants with "isolated" TEV were found at birth to have associated malformations that had not been identified by imaging during pregnancy. These abnormalities included hip dislocation (n = 2), bilateral post-axial polydactyly of the feet (n = 1), penile chordee (n = 1), and hypospadias (n = 2). CONCLUSION: In this consecutive series of infants with isolated talipes equinovarus, 95.8% had no additional malformations identified by examination at birth. None of the additional findings were severe enough to affect the medical prognosis of the affected infant. © 2014 John Wiley & Sons, Ltd.

Outcomes of children exposed in utero to chemotherapy for breast cancer.

INTRODUCTION: The incidence of breast cancer diagnosed during pregnancy is expected to increase as more women delay childbearing in the United States. Treatment of cancer in pregnant women requires prudent judgment to balance the benefit to the cancer patient and the risks to the fetus. Prospective data on the outcomes of children exposed to chemotherapy in utero are limited for the breast cancer population. METHODS: Between 1992 and 2010, 81 pregnant patients with breast cancer were treated in a single-arm, institutional review board-approved study with 5-fluorouracil, doxorubicin, and cyclophosphamide (FAC) in the adjuvant or neoadjuvant setting. Labor and delivery records were reviewed for each patient and neonate. In addition, the parents or guardians were surveyed regarding the health outcomes of the children exposed to chemotherapy in utero. RESULTS: In total, 78% of the women (or next of kin) answered a follow-up survey. At a median age of 7 years, most of the children exposed to chemotherapy in utero were growing normally without any significant exposure-related toxicity or health problems. Three children were born with congenital abnormalities: one each with Down syndrome, ureteral reflux or clubfoot. The rate of congenital abnormalities in the cohort was similar to the national average of 3%. CONCLUSIONS: During the second and third trimesters, pregnant women with breast cancer can be treated with FAC safely without concerns for serious complications or short-term health concerns for their offspring who are exposed to chemotherapy in utero. Continued long-term follow-up of the children in this cohort is required. TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT00510367. Other Study ID numbers: ID01-193, NCI-2012-01578. Registration date: 31 July 2007.