The Validity of Patient-reported Outcome Measurement Information System (PROMIS) Parent Proxy Instruments to Assess Function in Children With Talipes Equinovarus.

BACKGROUND: Idiopathic talipes equinovarus (ITEV) is a prevalent structural deformity of the foot and ankle in children. The disease-specific instrument (DSI) has commonly been used as an outcomes metric in these patients. The Patient-Reported Outcomes Measurement Information System (PROMIS) was developed to examine the quality of life across various medical conditions. PROMIS has been validated for multiple conditions; however, no studies have evaluated the use of the PROMIS in children with ITEV. METHODS: A prospective analysis was performed whereby the 2 questionnaires were mailed out to the parents of 91 patients, aged 5 to 17 years, with ITEV. Construct validity of the PROMIS Parent Proxy Profile short forms version was assessed by comparing its domains of Mobility, Fatigue, Pain Interference, and Pain Intensity to the DSI Function domain and PROMIS domains of Anxiety, Depressive Symptoms, Peer Relationships, and Pain Intensity to DSI Satisfaction domain. RESULTS: Thirty-one complete responses (34% response rate) were returned. The patients' current mean age was 8.8 years, 7 were female individuals, and 12 had unilateral ITEV. Bivariate correlation analysis, using Spearman correlation coefficients, demonstrated a moderate positive correlation between the DSI Function domain and the PROMIS Mobility domain (rs=0.52) and a moderate negative correlation between the DSI Function domain and the PROMIS Pain Interference domain (rs=-0.56), as well as the PROMIS Pain Intensity domain (rs=-0.75). A fair negative correlation (rs=-0.38) with PROMIS Fatigue domain was found. Correlations between the DSI Satisfaction domain and the PROMIS domains were fair or had little relationship. CONCLUSIONS: These results provide support for the validity of the PROMIS Mobility, Pain Interference, and Pain Intensity domains in this population, however there are no items in PROMIS that can assess overall satisfaction, as with the DSI. LEVEL OF EVIDENCE: Level II-prospective comparative study.

Predicting the need for surgical intervention in patients with idiopathic clubfoot.

BACKGROUND: The purpose of this study was to determine the early factors associated with the need for surgical interventions in patients with idiopathic clubfoot treated with the Ponseti method. METHODS: All patients with idiopathic clubfoot treated with Ponseti method at our institution with >3 years of follow-up were evaluated. Age at presentation, history of previous treatment, number of casts used, need for percutaneous Achilles tenotomy (PAT), age of initiation of foot abduction orthosis (FAO), compliance with FAO, and need for additional casts were recorded. Dimeglio/Bensahel and Catterall/Piriani scores were recorded at initial presentation, at initiation of FAO, at 1, 2, 3 years of follow-up, and at the most recent follow-up. RESULTS: Since 2000, 86 patients (134 feet) had >3 years of follow-up from time of initial presentation, and 43 of these feet (32%) had undergone surgery beyond a PAT. Patients who were noncompliant with the FAO were 7.9 times more likely to need surgery than those who were compliant [confidence interval (CI), 2.8-22.0; P<0.001]. Female patients were 5.4 times more likely to need surgery than male patients (CI, 1.8-16.6; P=0.003). For every 1 point increase in Dimeglio/Bensahel score at presentation, patients were 1.3 times more likely to need surgery (CI, 1.0-1.5; P=0.033). For every 1 point increase in Dimeglio/Bensahel score at initiation of the FAO, patients were 1.5 times more likely to need surgery (1.1-2.0, P=0.005). Moreover, for each additional cast required before the initiation of the FAO, patients were 1.5 times less likely to need surgery (CI, 1.1-2.7; P=0.030). No other variable significantly contributed to predicting the need for surgery. CONCLUSIONS: There are early factors that can be used to predict increased risk for surgical intervention in patients undergoing treatment for idiopathic clubfoot. Female patients and those patients with higher Dimeglio/Bensahel scores at presentation and at initiation of the FAO are at increased risk for needing surgical intervention. Noncompliance with the FAO is associated with the highest risk for surgical intervention. LEVEL OF EVIDENCE: Level III.

Achievement of gross motor milestones in children with idiopathic clubfoot treated with the Ponseti method.

BACKGROUND: The Ponseti method of idiopathic clubfoot treatment involves a series of weekly casts, a percutaneous Achilles tenotomy if needed, followed by wearing a foot abduction orthosis (FAO). Gross motor development of children with idiopathic clubfoot has not been examined. The purposes of this study were to determine the ages of achievement of gross motor milestones in children with clubfoot treated with Ponseti method and to compare those ages with historical normative developmental data. METHODS: In this prospective study, 51 children with idiopathic clubfoot who had their first cast applied when ≤90 days old, were full-term with no other orthopaedic conditions or previous clubfoot treatment, and were compliant with wearing FAO were enrolled. Parents were interviewed repeatedly to acquire the ages of achievement of 8 gross motor milestones. RESULTS: Fifteen children were excluded for reasons such as noncompliance with FAO, and not returning for follow-up. Thirty-six children, mean age of 15.2 days at first casting, achieved rolling prone to supine at a mean age of 5.1 months, rolling supine to prone at 5.1 months, sitting without support at 6.6 months, crawling on stomach at 7.1 months, crawling on hands and knees at 8.6 months, pull-to-stand at 9.0 months, cruising at 10.2 months, and ambulating independently at 13.9 months. When compared with previously published values for unaffected children, the mean ages of achievement for 6 of 8 milestones were significantly greater (P<0.05) for the children with clubfoot. The preambulatory milestones were achieved from 0.7 to 1.5 months later and independent ambulation up to 2.2 months later. Fifty percent of children with clubfoot were ambulating at 13.8 months; 90% at 17.7 months. CONCLUSIONS: Minimal delays in gross motor milestone achievement were found in children with idiopathic clubfoot treated with the Ponseti method. Delays were, at most, 1.5 months, except for independent ambulation, which was approximately 2 months. These findings should enable pediatric clinicians to alleviate the concerns of parents of children with idiopathic clubfoot regarding gross motor milestone achievement. LEVEL OF EVIDENCE: Therapeutic Level II.

Clubfoot classification: correlation with Ponseti cast treatment.

BACKGROUND: Many different clubfoot classification systems have been proposed, but no single one is universally accepted. Two frequently cited systems, developed by Dimeglio/Bensahel and Catterall/Pirani, are often used for evaluation purposes in the treatment of idiopathic clubfoot. Our hypothesis was that the initial scores would be positively correlated with the number of casts required for clubfoot correction, indicating to us that the more severe score would require more casts, and therefore truly show the accuracy and usefulness of the scoring system. METHODS: From May 2000 to April 2008, 123 patients (185 feet) with idiopathic clubfeet were treated. All patients were below 60 days of age (mean 15.3 d, range: 2 to 57 d) at the time of their initial evaluation, and had not received prior clubfoot treatment. All cast placements were under the supervision of the same pediatric orthopedic surgeon. Initial correction was achieved in all patients. RESULTS: The mean number of casts required for correction was 5.1 (range: 2 to 8). On the basis of number of casts required, no significant differences were found in final total scores (Dimeglio/Bensahel P=0.14 and Catterall/Pirani P=0.44), indicating a similar level of correction for all feet. The Dimeglio/Bensahel and Catterall/Pirani classification systems were both similarly, poorly correlated with the number of casts needed [Spearman rank correlation coefficients (rs)=0.34 vs. 0.33]. The 2 components with the highest correlations were equinus (rs=0.39) and forefoot adduction (rs=0.35) for the Dimeglio/Bensahel system and coverage of the lateral head of the talus (rs=0.40) and rigid equinus (rs=0.39) for the Catterall/Pirani system. CONCLUSIONS: When using the initial scores, both the Dimeglio/Bensahel and Catterall/Pirani classification systems had a low correlation with the number of Ponseti casts required. Analysis of the individual components revealed variability in the coefficients, with some having low-to-moderate correlation and others having none. There was no difference between the Dimeglio/Bensahel and Catterall/Pirani classification systems when measuring their correlation with the number of Ponseti casts required for clubfoot correction. An improved classification system is needed to predict the length of treatment and, ultimately, the risk of recurrence. LEVEL OF EVIDENCE: Prognostic Level IV.

Correction of arthrogrypotic clubfoot with a modified Ponseti technique.

Surgical releases for arthrogrypotic clubfeet have high recurrence rates, require further surgery, and result in short, painful feet. We asked whether a modified Ponseti technique could achieve plantigrade, braceable feet. Ten patients (mean age, 16.2 months; range, 3-40 months), with 19 arthrogrypotic clubfeet, underwent an initial percutaneous Achilles tenotomy to unlock the calcaneus from the posterior tibia followed by weekly Ponseti-style casts. A second percutaneous Achilles tenotomy was performed in 53%. Mean number of casts was 7.7 (range, 4-12). From pretreatment to completion of initial series of casts, mean scores of Dimeglio et al. improved from 16 to 5 (ranges, 12-18 and 2-9, respectively), Catterall scores (as modified by Pirani and colleagues) from 4.8 to 0.9 (ranges, 1.5-6.0 and 0.0-2.0), and maximum passive dorsiflexion from -45 degrees (range, -75 degrees to -20 degrees ) to 10 degrees (range, 0 degrees to 40 degrees ). Ankle-foot orthoses maintained correction. At the minimum followup of 13 months (mean, 38.5 months; range, 13-70 months), the mean maximum dorsiflexion was 5 degrees (range, -20 degrees to 20 degrees ), two patients had posterior releases and no patient's ambulatory ability was compromised by foot shape. Arthrogrypotic clubfeet can be corrected without extensive surgery during infancy or early childhood. Limited surgery may be required as the children age.

Perfusion is Not Measurably Decreased in Idiopathic Clubfoot.

BACKGROUND: Vascular aberration has been accepted as a potential etiology of clubfoot, and abnormal vasculature has been observed in as high as 85% of children with severe clubfoot. The perfusion index (PI) corresponds to the ratio of pulsatile to nonpulsatile blood flow at a monitoring site and can be used to quantify perfusion of the extremities. The purpose of this study was to use PI to compare the perfusion of clubfeet to controls in order to further assess the role of abnormal vasculature in clubfoot. METHODS: A Masimo Radical 7 Pulse Oximeter (Masimo Corporation, Irvine, California) was used to measure the PI and oxygen saturation (SpO2) of the feet of children 5 years of age and younger with and without clubfoot. The sensor was placed on the great toe. Patients with clubfoot undergoing non-operative treatment and control patients undergoing treatment in a clinic for orthopedic concerns not involving the foot and with no known vascular issues were assessed. The PI and SpO2 for the following three groups were compared: 1. affected feet of patients with bilateral or unilateral clubfoot, 2. unaffected feet of patients with unilateral clubfoot, and 3. control feet. RESULTS: One hundred and twenty-eight patients were enrolled, 64 with clubfoot (31 bilateral and 33 unilateral) and 64 controls. No significant differences in PI or SpO2 were found between: 64 clubfeet and 64 feet of controls (PI of 2.9 vs. 2.9, p = 0.984; SpO2 of 97.1 vs. 98.1, p = 0.192); unaffected feet of 30 patients with unilateral clubfoot and 64 controls (PI of 3.0 vs.2.9, p = 0.907; SpO2 of 96.9 vs. 98.1, p = 0.224); and affected and unaffected feet of 30 patients with unilateral clubfoot (PI of 3.3 vs.3.0, p = 0.500; SpO2 of 97.4 vs. 96.9, p = 0.527). CONCLUSIONS: No difference was observed in the PI or SpO2 when comparing affected clubfoot limbs with unaffected limbs, suggesting that vascular anomalies cannot fully explain the development of clubfoot.

Treatment of idiopathic clubfoot using the Ponseti method: minimum 2-year follow-up.

One hundred and thirty-seven idiopathic clubfeet were treated by the Ponseti technique and followed for at least 2 years. Nine feet (7%) were not corrected with initial casting and required early surgery. Recurrence after correction was related to compliance with bracing. At latest follow-up, two-thirds of those noncompliant with brace had recurrences with one-third of these recurrences requiring more extensive surgery than Achilles tenotomy and anterior tibial tendon transfer while only 14% of those compliant with brace had recurrences with none requiring more than Achilles tenotomy and anterior tibial tendon transfer. Early failures and recurrences constituted about 20% of our 137 feet by 2 years of follow-up. When the Ponseti method was fully followed, including initial casting, compliance with brace and treatment of recurrences by recasting, Achilles tenotomy and/or anterior tibial tendon transfer, our success rate was 93%.

Interobserver and intraobserver reliability of radiographic evidence of bone healing at osteotomy sites.

Plain radiographs are commonly used to evaluate the degree of bone healing after an osteotomy and the application of an external fixator. The purpose of the study was to assess intraobserver and interobserver reliability in determining bone healing, defined as bridging callus across three of four cortices, of osteotomy sites on radiographs. Substantial intraobserver reliability and a high intraobserver percentage agreement were found. Interobserver reliability was moderate and interobserver percentage agreement was less than half for agreement between all involved orthopaedic surgeons. The lower reliability across surgeons suggests that the determination of the extent of the bone healing is subjective.

Reminder: radiopacities from metal-containing substances.

A 17-month-old boy with a toed-out left lower extremity was presented for evaluation. Before coming to the doctor, the mother had applied a zinc oxide lotion to treat a simple diaper rash in the groin. The radiologist did not remove the boy's diaper for the radiographic examination and so did not notice the lotion. The first radiograph showed what looked like multiple soft-tissue calcifications in the groin, but the radiopacities had been produced by the zinc oxide. This case serves as a reminder to be aware that metal-containing substances both on and in the body can produce radiopacities.

Treatment of Idiopathic Clubfoot in the Ponseti Era and Beyond.

The initial treatment of idiopathic clubfoot was mostly surgical for the 1980s/1990s. In the latter half of the 1990s, there was a surge of interest in the Ponseti method of casting after the publication of Dr. Ponseti's 30-year results. Many authors have since shown correction rates in the high 90th percentile, rendering posteromedial release surgery almost obsolete. The success of the Ponseti method has been brought internationally and extrapolated to more and more difficult cases, such as idiopathic or syndromic, primary or recurrence. This new trend will create a different subset of complications.

Potocki-Lupski syndrome in conjunction with bilateral clubfoot.

Potocki-Lupski syndrome (PTLS) is a rare chromosomal microduplication syndrome resulting in multiple congenital abnormalities including developmental delays, autistic features, and certain structural anomalies, with cardiovascular being the most common. The phenotype of this contiguous gene duplication syndrome is quite variable and may include musculoskeletal abnormalities. Given the infrequency and novelty of this disorder, full phenotypic characterization of PTLS has not yet been fully elucidated. We present a case of severe bilateral clubfoot in a patient with PTLS. Diagnosis was made by array-based comparative genomic hybridization and confirmed by fluorescence in-situ hybridization. Because clubfoot was also present in an apparently unaffected brother, the presence of PTLS may have acted as a modifier of the phenotype. This report highlights the complex interaction of chromosomal and familial factors that contribute to musculoskeletal birth defects.

Congenital clubfoot.

Although the etiology of congenital clubfoot remains unknown, reproducible pretreatment grading now seems possible. However, the lack of an agreed-on and reproducible posttreatment evaluation system still hinders outcome studies of the treatment of clubfoot. The literature from about 1970 to 1990 contains enthusiastic reports on the correction of congenital clubfoot through extensive surgical release procedures. Over time, we have come to recognize the complications of such surgery, including recurrence, overcorrection, stiffness, and pain (WJ Shaughnessy, MD, P Dechet, MD, HB Kitaoka, MD, Vancouver, BC, Canada, unpublished data, 2000). Perhaps because of these findings, there is a renewed interest in nonsurgical techniques for the correction of congenital clubfoot. Recent studies have documented the effectiveness of the two leading techniques involving serial manipulation and cast treatment. The Ponseti technique appears to be effective and requires only a reasonable amount of time out of the lives of the patient and his or her parents. The technique frequently includes some minimally invasive surgery. The Kite and Lovell technique requires minimally invasive surgery less often but is more time consuming. French investigators and others have introduced new ideas that may reduce the need to immobilize the foot. The French approach requires fairly extensive physical therapy and demands substantial parental time and attention. It is not yet clear that the French technique is more successful in obviating the need for surgery than is expertly applied serial manipulation and cast immobilization. It also has not been proved that the long-term results of the French technique are better than those of serial manipulation and cast immobilization. It is probably that unless the French technique is found to substantially decrease the need for surgery, it will prove to be less cost effective than serial manipulation and cast immobilization. It is likely that a small number of clubfeet will require surgery even after expertly applied nonsurgical treatment. However, it is hoped that such surgery will be less extensive than procedures commonly performed in the recent past.

Percutaneous fixation in clubfoot surgery: a radiographic and gait study.

Twenty patients with Type II clubfeet were evaluated an average of 3.1 years after complete surgical soft tissue release. A pinned group had fixation of the talonavicular and calcaneocuboid joints and a non-pinned group had no fixation. The control group consisted of 10 children without foot ailments. Radiographic measurements of talocalcaneal and talus-first metatarsal angles and frequencies of dorsal and medial navicular subluxation were not different for the two treatment groups. Foot progression angle was not significantly different for the two treatment groups, but was significantly different between all clubfoot patients (3.6 degrees of in-toeing) and the control group (5.0 degrees of out-toeing). Tourniquet time was significantly greater for the pinned than for the non-pinned group, but the duration of surgery was similar. Similar radiographic and gait measurement findings suggest that clubfoot surgery with and without percutaneous fixation have comparable outcomes.

A method for the early evaluation of the Ponseti (Iowa) technique for the treatment of idiopathic clubfoot.

The Ponseti casting technique is reported to have a high success rate in the treatment of idiopathic clubfoot. Non-operative treatment of clubfoot provides a lower complication rate, less pain, and higher function as the patient ages than operative treatment. To demonstrate serial post-treatment change in clubfeet over time, three clubfoot rating systems were utilized in the current study. Patients compliant with the Ponseti technique and treated before the age of 7 months, had a 92% success rate at an early follow-up after casting was completed. It is not the purpose of this article to analyze the long-term clubfoot treatment result but to establish tools which can be used to judge initial success with the Ponseti technique. Complications are few and minor, limited to equipment used and cast technique.

Calcaneocuboid arthrodesis for recurrent clubfeet: what is the outcome at 17-year follow-up?

PURPOSE: Calcaneocuboid arthrodesis was used during revision clubfoot surgery in order to maintain midfoot correction. The purposes of this study were to determine: (1) functional level at 17-year follow-up compared to 5-year follow-up; (2) patients' current functional level, satisfaction, and pain; and (3) current arthropometric measurements. METHODS: Twenty patients (27 clubfeet) with clubfoot relapse underwent revision soft tissue release and calcaneocuboid fusion between 1991 and 1994. They were previously evaluated at a mean follow-up of 5.5 years. Ten out of 20 patients (13 clubfeet), mean age of 24 years, were reevaluated at mean follow-up of 17.5 years. The Hospital for Joint Diseases Functional Rating System (HJD FRS) for clubfoot surgery, Outcome Evaluation in Clubfoot developed by the International Clubfoot Study Group, the Clubfoot Disease-Specific Instrument, American Academy of Orthopaedic Surgeons (AAOS) Foot and Ankle Outcomes Questionnaire, Laaveg and Ponseti's functional rating system for clubfoot and pain scale were completed by patient and/or surgeon to assess function, patient satisfaction and pain. Foot and ankle radiographs and anthropometric measurements were reviewed. For HJD FRS, scores from original follow-up were compared to current ones. RESULTS: The HJD FRS score of all feet was 65.9, demonstrating a significant decline from the original mean score of 77.8 (p = 0.03). Excellent/good HJD FRS scores went from 85 to 38 %. Mean AAOS Foot Ankle Outcomes Questionnaire standardized core and shoe comfort scores were 84.6 and 84.5, respectively. Average foot pain was 1.8 on a scale of 1-10. Patients were very/somewhat satisfied with status of foot in 76 % of feet and appearance of foot in 46 % of feet, based on Clubfoot Disease-Specific Instrument questions. CONCLUSIONS: Revision clubfoot surgery with calcaneocuboid fusion in patients 5-8 years of age showed an expected decline in functional outcome measures over a 17-year follow-up period. It still produced comparable results to other studies for a similar population of difficult, revision cases, and should have a place in current surgical treatment techniques.

Idiopathic clubfoot treated with the Ponseti method: factors associated with patient follow-up.

BACKGROUND: Follow-up after treatment with the Ponseti method is important because of the known association be- tween lack of brace wear and recurrence. This study was designed to ascertain factors associated with patients who did not return for the recommended follow-up versus those who did. METHODS: Between January 1, 2000, and December 31, 2009, 222 patients were treated for idiopathic clubfoot at the New York Ponseti Clubfoot Center at NYU Hospital for Joint Diseases, of which 93 patients (143 feet) were potentially available for follow-up (i.e., ≤ 7 years of age, had not moved, or transferred care to another institution). Attempts were made to contact all patients' parents or guardians by telephone to respond to a survey, which included questions from the Disease Specific Instrument and the Oxford Ankle Foot Questionnaire. Forty-two of the 93 patients (45%) responded. The responder group, those who answered the survey, was compared to the group of non-responders. The responder group was further divided into a returning group (35 out of 42, 83%) and a lost to follow-up group (17%, not followed-up in over a year). A chart review was performed for demographics, Dimeglio/Bensahel and Catterall/Pirani scores, and treatment. RESULTS: When comparing the responder and non- responder groups, the responder group had significantly lower (p < 0.05) Catterall/Pirani scores at initial visit than the non-responder group (5.0 versus 5.5), but otherwise these two groups were similar. Among the responders, 91% or more were very satisfied/satisfied with status and appearance of foot in both lost to follow-up and returning groups. The lost to follow-up group was significantly (p < 0.05) older at the time of the survey (5 years versus 3.7 years), required significantly fewer casts (4.4 versus 5.5), had significantly lower Dimeglio/Bensahel scores at time of the start of foot adbuction orthoses (FAO) (0.0 versus 2.0), and trended toward greater footwear limitations (p = 0.051) compared with the returning group. CONCLUSIONS: Number of casts, severity scores at the start of FAO, and footwear limitations are possible factors to differentiate between lost to follow-up and returning patients. This information may help other clubfoot centers provide vigilant outreach and therefore decrease recurrence rate.

Persistent clubfoot deformity following treatment by the Ponseti method.

The Ponseti method of clubfoot correction is now widely practiced worldwide. Initial correction rates are nearly 100%, but subsequent relapses may occur in up to one-third of patients. Very little has been written by anyone other than Dr Ponseti about the characterization and treatment of recurrent clubfoot deformity following use of the Ponseti method. This review paper is the first one which draws together the current literature on the topic.

Progression of idiopathic clubfoot correction using the Ponseti method.

The Ponseti technique involves sequential clubfoot correction by abduction, supination, and finally dorsiflexion. Although shown to be effective, correction progression has not been examined. The Dimeglio/Bensahel classification system was used to analyze heel equinus, varus, midfoot rotation, adduction, posterior crease, medial crease, and cavus initially and after each casting. From 2000 to 2008, 123 patients (185 feet) with idiopathic clubfeet, aged below 60 days, without prior treatment were grouped by number of casts required. Successive castings achieved goals of reducing cavus and medial crease first, then gradually correcting midfoot rotation, adduction, and heel varus. Unexpectedly, heel equinus improved simultaneously with midfoot variables, as well as with final casting.

Correction of clubfoot deformity associated with Weber type I tibial hemimelia using the Ponseti method.

The clubfoot deformity associated with Weber type I tibial hemimelia, a rare congenital disorder, is rigid and difficult to correct. Surgeons have utilized a variety of treatment methods. Since the 1960s, some adopted the Syme amputation to produce a weightbearing lower limb. Others began to explore alternatives such as the Ilizarov technique, ankle reconstruction, and casting, which salvage the foot but have produced mixed results. The current investigators suggest that the Ponseti method, a minimally invasive technique, can produce a functional weightbearing foot. Two cases were treated with the Ponseti method, including a percutaneous Achilles tenotomy and post-cast bracing. After a minimum of 2-years follow-up, both are ambulatory.