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Treatment of Infantile Blount's Disease (Infantile Tibia Vara) with a Novel Brace: Preliminary Results

Lehman, Wallace B.; Taha, Abdel Majid Sheikh; Sala, Debra A.; Sanchez, Jessica; Chu, Alice
Introduction: Over the years, several types of braces have been used to treat the proximal tibial varus deformity of infantile Blount's disease. The purpose of this study was to evaluate the effectiveness of the JMMR Blount's KAFO (JMMR Inc, Ridgewood, NJ, USA) in correcting infantile tibia vara. Methods: From November 2011 to June 2014, four patients with Blount's disease completed treatment with JMMR Blount's KAFOs. The dynamic quality of the brace differentiates it from the standard orthotic treatment of Blount's disease. The active movement of the swivel hinge located on the posterior of plastic lower leg cuff and the telescoping lateral ankle hinge facilitate the application of the valgus stress force below the knee. This, in turn, permits full knee and ankle motion. As the corrective forces are produced during weight bearing, the orthosis is worn only during waking hours. Prebracing and postbracing anterioposterior radiographs were reviewed by an examiner who was blinded to patient's name and date of the radiograph. Results: The four patients (two males, two females) had a mean age of 23 months (range, 21"“27) at initiation of bracing. Two were bilateral and two were unilateral. Prebracing to postbracing comparison demonstrated Langenskiold stage improved by at least one stage and metaphyseal-diaphyseal angle decreased from a mean of 15° to 4.3°. The mean tibiofemoral angle was 19.2° varus prebracing and 0.7° valgus postbracing. The mean duration of brace use was 11 months (range, 7"“15). Conclusions: When the treatment of Blount's disease was started before 3 years of age, the JMMR Blount's KAFO corrected the tibia vara.
SCOPUS:85133158602
ISSN: 1040-8800
CID: 5315682

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

Masrouha, Karim Z; Moses, Michael J; Sala, Debra A; Litrenta, Jody; Lehman, Wallace B; Chu, Alice
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/METHODS:Level II-prospective comparative study.
PMID: 30913133
ISSN: 1539-2570
CID: 3776952

The validity of promis parent proxy instruments to assess function in children with talipes equinovarus [Meeting Abstract]

Chu, A; Masrouha, K; Sala, D; Litrenta, J; Lehman, W
The Patient-Reported Outcomes Measurement Information System (PROMIS) was developed to examine the quality of life across various medical conditions. No studies have evaluated the use of the PROMIS in children with idiopathic talipes equinovarus (ITEV). The construct validity of the PROMIS Parent Proxy Profile short forms version was assessed by comparing its domains of Mobility, Fatigue and Pain Interference to the Function domain of the Disease Specific Instrument (DSI) for ITEV. The two questionnaires were mailed out to the parents of 93 patients, aged 5-17 years, with ITEV. We received a total of 21 complete responses (22.6% response rate). The patients' current mean age was 8.0 years (SD, 3.0 years). Seventeen were males and 4 were females. Nine had unilateral ITEV and 12 had bilateral ITEV. Bivariate correlation analysis, using Pearson correlation coefficients, demonstrated a moderate positive correlation between the DSI Function domain and the PROMIS Mobility domain (r=0.67) as well as a moderate negative correlation between the DSI Function domain and the PROMIS Pain Interference domain (r=-0.64). A fair negative correlation was found between the DSI Function domain and the PROMIS Fatigue domain (r=-0.35). These results provide support for the validity of the PROMIS Mobility Item in this population, however further studies are needed to assess the feasibility and reliability of these instrument items as well as other items within PROMIS
EMBASE:628084470
ISSN: 1473-5865
CID: 3944502

Recurrent clubfoot after Ponseti treatment can be managed with minimally invasive surgery: A mean 7-year follow-up [Meeting Abstract]

Masrouha, K; Schoenfeld, K; Sala, D; Litrenta, J; Lehman, W; Chu, A
The Ponseti method for the correction of idiopathic talipes equinovarus (ITEV) produces excellent results, however recurrence is common, ranging from 11-37%. Recurrences require repeat manipulation and casting, frequently followed by surgical procedures. We hypothesized that at a minimum 4-year follow-up, the rate of invasive surgical interventions such as posteromedial release (PMR) or osteotomy, is low. A retrospective chart review of patients with ITEV treated by the Ponseti method from January 1999 to June 2017 was conducted. An invasive procedure was defined as a PMR or osteotomy, whereas minimally invasive procedures involved tendon lengthening and transfers. Inclusion criteria were patients with ITEV, less than 90 days old at initial visit, no previous series of casts, and potential of being 4 years old at last visit. A total of 150 feet (102 patients) were studied. Mean age at initial visit and last follow-up were 19.5 days and 7.6 years, respectively. For the initial series of casts, mean number of casts was 5.4 and 67.3% of feet had percutaneous Achilles tenotomies. Fifty-eight percent did not undergo any form of surgical procedure, while 33% underwent a tibialis anterior tendon transfer alone or in combination with a gastrocnemius recession, tendo-Achilles lengthening, or plantar fascia release. Early in the series, three feet in two patients underwent a PMR. The mean age at time of surgery was 55.3 months. In contrast to those treated for recurrence after PMR, the vast majority (98%) of feet with recurrence after Ponseti-treated ITEV had either no surgery or minimally invasive surgery
EMBASE:628084447
ISSN: 1473-5865
CID: 3944512

Perfusion is Not Measurably Decreased in Idiopathic Clubfoot

Forrester, Lynn Ann; Grissom, Helyn E; Shakked, Rachel J; Danna, Natalie R; Sala, Debra A; Lehman, Wallace B; Otsuka, Norman Y; Chu, Alice
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.
PMID: 31513525
ISSN: 2328-5273
CID: 4088342

Age of Recurrence in Idiopathic Clubfoot Treated with the Ponseti Method

Goldstein, Rachel; Chu, Alice; Sala, Debra; Lehman, Wallace
OBJECTIVES: While studies of patients undergoing the Ponseti method for idiopathic clubfoot demonstrate excellent correction after initial treatment, recurrence can occur in more than half of the cases. Few studies have demonstrated the age at which recurrence typically occurs or if age at discontinuation of the foot ankle orthosis (FAO) is associated with risk for surgical intervention. METHODS: Patients with idiopathic clubfoot treated with the Ponseti method with greater than 3 years of follow-up were evaluated. Age at presentation, need for percutaneous Achilles tenotomy, age of initiation of foot abduction orthosis, adherence with FAO, and need for additional treatment were recorded. Severity scores were recorded at initial presentation and yearly throughout follow-up. RESULTS: 110 patients were followed for an average of 5.9 years, and 32 patients required surgical intervention. Patients who eventually required surgery had significantly higher severity scores at presentation. Though they maintained higher scores throughout the follow-up period, they did not demonstrate significantly higher scores until 3 years of age. FAO was used until an average age of 2.6 years (range: 0.4 to 5.1 years). There was a significant difference in the age at which the FAO was stopped in those patients who eventually required surgery versus those who did not (2.2 years versus 2.8 years). CONCLUSIONS: Patients with recurrence after initially successful Ponseti treatment generally do not become distinguishable by our current classification systems until 3 years of age. Patients who used the FAO for more than 2 years were 2.77 times less likely to require surgery than those who used it less than 2 years. FAO use should be continued until at least 3 years of age in patients undergoing Ponseti treatment for idiopathic clubfoot.
PMID: 28902604
ISSN: 2328-5273
CID: 2709682

Perfusion is not measurably decreased in idiopathic clubfoot [Meeting Abstract]

Chu, A; Forrester, L; Shakked, R; Lehman, W; Otsuka, N
Purpose: Abnormal vasculature has been observed in as much as 85% of children with clubfoot. The purpose of this study was to compare foot perfusion between clubfeet and unaffected feet. Methods: A Masimo Radical 7 Pulse Oximeter (Masimo Corporation, Irvine, CA) was used to measure the foot perfusion index (PI) of children five years of age and younger. Patients with clubfoot undergoing nonoperative treatment, and control patients undergoing treatment for orthopedic concerns not involving the foot, were tested. Exclusion criteria for patients with clubfoot were previous clubfoot surgery and non-idiopathic etiology. The PI of the affected clubfeet was compared to the PI of the unaffected feet of the controls. Results: Sixty-six patients were enrolled, 34 with clubfoot (16 bilateral and 18 unilateral), and 32 controls. No significant difference in PI was found between the affected feet and the unaffected feet (3.1 vs. 2.5; P = 0.200). Conclusion: This analysis showed no difference in PI between clubfeet and unaffected feet of controls. Significance: Although abnormal vasculature in clubfoot has been reported, the perfusion to a non-operative treated clubfoot is similar to an unaffected foot
EMBASE:620636174
ISSN: 1473-5865
CID: 2958882

Treatment of Idiopathic Clubfoot in the Ponseti Era and Beyond

Chu, Alice; Lehman, Wallace B
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.
PMID: 26589078
ISSN: 1558-1934
CID: 1856242

Potocki-Lupski syndrome in conjunction with bilateral clubfoot

Dhanaraj, Dinesh; Chu, Alice; Pappas, John G; Moran, Ellen; Lehman, Wallace B
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.
PMID: 25768679
ISSN: 1060-152x
CID: 1495202

Predicting the Need for Surgical Intervention in Patients With Idiopathic Clubfoot

Goldstein, Rachel Y; Seehausen, Derek A; Chu, Alice; Sala, Debra A; Lehman, Wallace B
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.
PMID: 25075887
ISSN: 0271-6798
CID: 1090182