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The Paley ulnarization of the carpus with ulnar shortening osteotomy for treatment of radial club hand
Abstract
Recurrent deformity from centralization and radialization led to the development in 1999 of a new technique by the author called ulnarization. This method is performed through a volar approach in a vascular and physeal sparing fashion. It biomechanically balances the muscle forces on the wrist by dorsally transferring the flexor carpi ulnaris (FCU) from a deforming to a corrective force. The previous problems of a prominent bump from the ulnar head and ulnar deviation instability were solved by acutely shortening the diaphysis and by temporarily fixing the station of the carpus to the ulnar head at the level of the scaphoid. This is the first report of this modified Paley ulnarization method, which the author considers a significant improvement over his original procedure.
... Ulnarization is more extensile and permits decompression of the ulnar and median nerves and radial and ulnar arteries. The caput ulnar artery, which is a branch of the ulnar artery, can be visualized and protected, preventing damage to the circulation of the distal ulnar physis and epiphysis [17]. ...
... Paley reported 0% recurrent wrist deformity or growth arrest after ulnarization [17]. In longer term follow-up, 10-15% of patients developed gravity-related dynamic ulnar deviation. ...
... For reference, the original ulnarization procedure [15,16] with the use of external fixation will be referred to as ulnarization G1 (Generation 1). To improve upon this, Paley modified the procedure by shortening the ulna [17]. The shortening did not eliminate the ulnar bump in every case and some cases continued to grow past the carpus as in G1. ...
(1) Background: Patients treated with the two previous generations of ulnarization developed a bump related to the ulnar head becoming prominent on the radial side of the hand. To finally remedy this problem, a third generation of ulnarization was developed to keep the ulnar head contained. While still ulnar to the wrist center, the center of the wrist remains ulnar to the ulnar head, with the ulnar head articulating directly with the trapezoid and when present the trapezium. (2) Methods: Between 2019 and 2021, 22 radial club hands in 17 patients were surgically corrected with this modified version of ulnarization. (3) Results: In all 17 patients, the mean HFA (hand–forearm-angle) correction was 68.5° (range 12.2°–88.7°). The mean ulna growth was 1.3 cm per year (range 0.2–2 cm). There were no recurrent radial deviation deformities more than 15° of the HFA. (4) Conclusions: This new version of ulnarization may solve the problem of the ulna growing past the carpus creating a prominent ulnar bump. The results presented are preliminary but promising. Longer-term follow-up is needed to fully evaluate this procedure.
... Any surgical attempts on RCH focus on creating a centralized wrist to improve the function and appearance of the upper limb [4]. The various surgical procedures currently employed for managing hands with RCH include preliminary soft tissue distraction and tendon transfer with or without ulnar osteotomy [7,8], surgical centralization of the wrist [9,10], use of external fixators [11], proximal fibular transplants, arthrodesis [12] and recently ulnarization of the carpus with ulnar shortening osteotomy [13]. ...
... This technique resulted in ulnar deviation of wrist due to gravity on the longer term. Recently, the same author put forward a revised technique called as ulnarization of the carpus with ulnar shortening osteotomy [13] to nullify the wrist ulnar deviation and has reported satisfactory results on a 2-year follow-up. ...
... The children in group 2 (ulnarization of the carpus with ulnar shortening osteotomy) were operated as per the guidelines of Paley [13]. Active elbow and finger exercises were initiated after the surgery with the hand in elevation. ...
Background
Radial club hand (RCH) is characterized by a wide array of hand and forearm anomalies. Various treatment approaches have been described depending upon the stages of RCH. The major drawback of these studies is that the effectiveness of these interventions was reported on clinical and radiological outcomes. With the increasing focus on patient-centered care nowadays, we wanted to identify the components associated with functioning and evaluate the effectiveness of two surgical procedures on functional outcomes using the International Classification of Functioning, Disability and Health (ICF)-based tools.
Materials and Methods
We identified 14 children from our records (nine boys, five girls) with a mean age of 5.6 years, classified as Bayne types III–IV and classified them into two groups; those who were operated by centralization (group 1) and ulnarization of the carpus with ulnar shortening osteotomy procedure (group 2). The outcomes were evaluated by the brief ICF core set for the child and youth with cerebral palsy up to the age of 5 and the brief ICF core set for hand conditions for a period of 1 year after surgery.
Results
The results showed that both the operative techniques showed improvement in the structure component (s730-structure of upper extremity). ICF categories of d445-hand and arm use, d530-toileting, and d880-engagement in play showed a change in frequencies of more than 40% after surgery and were maintained till follow-up. However, categories related to muscle power functions (b730), muscle tone (b735), fine hand use (d440), hand and arm use (d445) and engagement in play (d880) showed no significant improvement (p > 0.05). There were no differences between both the surgical procedures in improving the outcomes (p > 0.05).
Conclusion
We conclude that surgical techniques may be more appropriate to improve the cosmetic or structural appearance of the upper extremity than functioning.
... To restore proper hand alignment, various authors have proposed different techniques over the years. These include ulno-carpal arthrolysis associated with centralization or radialization of the distal ulna [3][4][5][6][7], progressive dorso-ulnar wrist distraction by the means of an external fixator [8][9][10], microvascular metatarsophalangeal joint transfer to the wrist radial side [11], distal ulna subperiosteal transposition to the wrist radial side [12], and index finger pollicization with its metacarpal base fused to the radial side of the distal ulna [13]. With the procedure performed in children between 1 and 4 years of age, satisfactory outcomes are most commonly observed during the initial followup; however, the long-term prognosis is compromised by recurrence of the deformity, regardless of the technique used. ...
... Indeed, in very young patients who have major growth potential at the time of surgery, sustainability of the correction requires flawless rebalancing of the forearm-wrist complex in the three planes of space, which is often not feasible in such complex regional deformities. In order to prevent recurrences, the majority of these authors recommend adding tendon transfers and/or internal fixation to stabilize the osteoarticular realignment [4,6,7,10]. ...
... As witnessed in early onset scoliosis deformities [16,17], such devices may prevent the need for repetitive surgery while keeping the hand aligned with the forearm through the child's growth. Previously described by different authors [3,7], the use of a radiopalmar approach seems adequate, since it makes it possible to perform radial soft tissue release and/or ulnar osteotomy as needed while implanting the device. In addition, since the ulna distal epiphysis is not traumatized surgically, full growth potential of the forearm as well as wrist motion are theoretically preserved. ...
... Гипоплазия лучевой кости обуславливает отклонение запястья и кисти в радиальную сторону, что требует проводить коррекцию лучевой девиации в первую очередь [3]. Наиболее популярные методы хирургического лечения -центрация и ее модифицикации (радиализация, ульнаризация), создание «вилки» лучезапястного сустава с помощью микрохирургической аутотрансплантации сустава стопы, расщепление дистального отдела локтевой кости в сагиттальном направлении [4,5,6]. Устранение деформации предплечья с помощью различных вариантов стабилизации кисти на локтевой кости позволяет улучшить функцию предплечья и кисти, а также придать эстетичный вид верхней конечности [7]. ...
Background. Congenital radial club hand is characterized by the underdevelopment of all forearm structures. Ulnar bone shortening ranges from 24.7% to 50.0% compared to the intact limb. The aim of the study was to evaluate the outcomes of ulnar lengthening by distraction osteogenesis in patients with congenital radial club hand type IV who underwent osteotomy with the formation of a bone-periosteal-muscle flap, and to compare these results with the treatment outcomes of the patients who had standard (oblique) ulnar osteotomy. Methods. The main group consisted of 20 patients who underwent osteotomy with the formation of a bone-periosteal-muscle flap during ulnar lengthening between 2019 and 2022. The control group included 19 patients (22 forearms) who underwent oblique ulnar osteotomy between 1998 and 2018. The following indicators were evaluated: length of the regenerate, distraction time, correction period, fixation index, osteosynthesis index, and complications. Results. A lengthening of 4.1 cm was achieved (30.7% of the initial ulnar bone length). The correction of angular deformity was 71.4%. Greater correction was achieved with osteotomy in the proximal ulna. In the subgroup with proximal segment osteotomy, the distraction and osteosynthesis indices were 25.6 and 25.7 days/cm, respectively. In the mid-third osteotomy group, these indices were 42.3 and 42.6 days/cm, respectively. Complications were limited to inflammatory phenomena in 30% of cases. All patients in the main group exhibited successful regenerate formation. Thus, the bone fragment with a periosteal-muscle pedicle serves as an additional source of osteogenesis during distraction. Conclusions. This study demonstrates the appropriateness of osteotomy with the formation of a bone-periosteal-muscle flap in children with congenital radial club hand. This technique allows for greater deformity correction, a shortened regenerate formation period, and a reduction in complications.
... Paley later modified this technique as modified Paley ulnarization by adding acute shortening of the diaphysis and temporary fixation of station of carpus to the ulnar head at the level of scaphoid to prevent prominent bump as seen in the previous technique. [14] Both radialization and ulnarization are widely known techniques, and one or the other is used extensively in the treatment of managing radial club hand disorder. ...
Various terms in orthopedics are formed using or incorporating the name of native bones and these terms then describe a clinical entity or a procedure. The term thus created, however, may or may not be directly related to the bone associated. There are a few interesting examples of such terms that are encountered in the orthopedic literature. A short recollection of those few notable terms that have the name of a known bone within their terminology is described here for general reading and knowledge.
Keywords: Acetabulization, bones, olecranization, radialization, skeletal system, tibialization, ulnarization
... Treatments include conservative procedures, which are reserved for patients with slight deformity and stable joints [1,3,7], and surgery, where centralization [1,5,8,9], radialization [10], or ulnarization [11] of the wrist at the distal end of the ulna is the standard procedures. Additional surgical procedures may include osteotomy and distraction osteogenesis of the bent ulna, which improve the aesthetic appearance and reduce the length of the forearm relative to a healthy extremity [12]. ...
Purpose: Publications evaluating the results of the ulna lengthening in congenital radial deficiency are based only on small groups of subjects which yield statistical studies of low scientific value. The aim was to examine the effectiveness of ulna lengthening in radial longitudinal deficiency and determine the number and quality of complications based on one of the most numerous study groups described in the literature.
Methods: The material consists of a study group with 31 upper limbs of unmatured patients diagnosed with type III and IV radial longitudinal deficiency. The study group was evaluated based on the parameters known from the literature. The difficulties during elongation were classified according to Paley's classification.
Results: The study group contained patients with a mean age of 9 years, and the number of boys and girls was comparable. Ulna length significantly increased after elongation compared to the initial bone length. The patient's age didn't affect the ulna lengthening, and the amount of elongation didn't significantly affect the total stabilization period. However, the total stabilization time increased with increasing patient age. Difficulties affected more than half of the cases.
Conclusions: Ulna elongation in congenital radial deficiency results in significant lengthening of the ulna, and thus the entire forearm, compared to the initial bone length. This technique has a high percentage of difficulty, so its use should be considered after cautious discussion with the parents and patients.
... Наибольшее количество работ, представленных в мировой и отечественной литературе, посвящено хирургическому лечению ВЛК. Предложены различные варианты оперативных вмешательств с целью устранения лучевой девиации кисти: центрация и радиализация [9], ульнаризация [10], микрохирургическая аутотрансплантация плюснефалангового сустава стопы с целью формирования «вилки» лучезапястного сустава [11]. Однако в связи с изменением нормальных анатомических соотношений мягких тканей и костей предплечья невозможно пассивно вывести кисть в среднее положение, что не позволяет произвести оперативное лечение одномоментно без укорочения локтевой кости. ...
Keywords. Congenital radial club hand, conservative treatment, centralization, children.
Background. The main method of correction of forearm deformities in сongenital radial club hand (CRCH) type IV according to the Bayne and Klug classification is operative. Changes in the normal anatomical proportions of the soft tissues and bones of the forearm does not allow to correct the deformity in one stage, second operation is usually needed. Conservative treatment is recommended from the first days of the child's life. Aim. To evaluate of the results of preoperative conservative treatment in patients with CRCH type IV according to the Bayne and Klug classification Methods. The results of treatment 50 patients with CRCH type IV from 2010 to 2020 years were analyzed. The assessment was carried out in 14 girls and 36 boys, aged 2,2 ± 1,3 years. In the main group (N=25), children received preoperative conservative treatment aimed at removing the hand to the center position. In the control group, this treatment was not performed. The analysis was carried out: 1) hand forearm angle (HFA°); 2) the period of distraction needed to correct the deformity was noted (days). Results. At the first examination, the HFA of the main group averaged 88.7±11.4°, after conservative treatment it was 41.8±21.3°. In 48% of cases, patients of the main group underwent one-stage hand centralization. With the two-stage method, the distraction period was 48,6% less than in the control group. Conclusion Preoperative conservative treatment improves the condition of soft tissues on the radial surface of the forearm, reduces the number of operations, shortens the distraction period and reduces the risk of complications
... All seven patients, who had a combined 8 temporary arthrodesis implants placed around the hip or knee at the index procedure, required planned implant removal. Temporary arthrodesis is a technique developed by the senior author (D.P.) in which a joint is temporarily spanned with plate, rod or external fixator in order hold a joint contracture in the corrected position to maintain correction or to augment fixation for healing of an osteotomy [19,[30][31][32][33] or to prevent development of contracture during lengthenings. This is particularly useful in the Paley-type rotationplasty as there is little room in the remnant femoral head to achieve solid fixation to the remnant femur with screws alone. ...
Rotationplasty is a reconstructive option for severe congenital femoral deficiency (CFD). The senior author (D.P.) developed five new rotationplasty techniques for use in CFD based on the Paley classification, including the Paley–Brown (fusion femur to pelvis), Paley (fusion femur to femoral head), Paley–Winkelman (insertion tibial condyle to acetabulum), PaleySUPERhip–Van Nes (hip osteotomy with knee fusion) and PaleySling–Van Nes (hip reconstruction with knee fusion revision) rotationplasty techniques. The purpose of this study is to retrospectively evaluate the complications, radiographic outcomes and need for secondary surgery in 19 rotationplasty cases performed by the senior author (D.P.) for severe CFD from 2009 to 2019. Rotationplasty comprised only 2% of the authors treated CFD cases during this period. Average age at surgery was 8.6 years old. Average follow-up was 3.3 years. Sixteen concomitant procedures were performed including temporary arthrodesis, tibial osteotomy and SUPERhip procedure. The most common complication was wound necrosis/dehiscence, which occurred in 52% of the cases related to the circumferential incision and required a total of 31 additional debridements. Additional complications were successfully treated and included sciatic nerve palsy decompressed by abducting the femur, a tibial delayed union that underwent bone grafting, two distal femur failed epiphysiodesis treated by revision with one osteotomy and a thigh compartment syndrome requiring debridement. Indication specific rotationplasty successfully addresses the severe degree of femoral deficiency, deformity, and discrepancy in patients with CFD, despite high rates of wound complications.
... Since 1997, I started using shortening as a means to increase the magnitude and complexity of acute deformity correction. Shortening became one of the steps incorporated into the SUPERhip procedure [ Figure 1], [5] SUPERankle procedure [ Figure 2], [6] tibial hemimelia reconstruction, [7] ulnarization [ Figure 3], [8] congenital pterygium, and knee flexion contracture correction [ Figure 4]. We also used shortening to eliminate bone defects instead of bone transport. ...
... Лучевая косорукость III и IV типов встречается наиболее часто, примерно в 79 % случаев. На первом этапе целью оперативного лечения является устранение лучевой девиации и восстановление возможности двустороннего схвата кисти [6,7,[10][11][12]. В последующем большинство родителей пациентов жалуются на укорочение предплечья. ...
Background. Congenital radial club hand is characterized by the radial deviation of the hand, the longitudinal underdevelopment of the forearm, and the dysfunction of the upper limb. The shortening of the ulna is observed in all types of congenital radial club hand. The average shortening of the ulna surgical treatment was 33.3% compared to the intact contralateral side. Aim. This study aimed to evaluate the results of ulna elongation by the method of external fixation, depending on the level of osteotomy, in patients with congenital radial club hand type IIIIV. Materials and methods. The treatment results of 36 patients with congenital radial club hand type IIIIV from 1998 to 2018 were analyzed. The average age of the patients was 7.4 years 3.5 years. The patients were divided into three groups, depending on the level of ulnar osteotomy. Shortening of the ulna, correction of the angle of deformity of the ulna, radial deviation of the hand, period of correction, elongation obtained, index of fixation and osteosynthesis, and associated complications were analyzed. Results. The observation period was an average of 5.8 years. Before surgical treatment, the ulna was 33.3% shorter, while after surgery, it was 16%. Before surgery, the angle of deformation was 20.5 14.8, while after surgery, it was 7.4 5.6; this gives an angle of deformity correction of 63.9%. The elongation of the ulna was 3.2 1.1 cm. In patients who underwent proximal osteotomy, the resulting elongation was 32% and 18.4% more, respectively, than in patients who underwent an osteotomy in the middle and distal sections of the ulna. In group 1, the correction period was 24.4% and 28.9% more than in groups 2 and 3, respectively. The index of fixation in group 1 was 53.6%, which was 45.7% less than in groups 1 and 3. Postoperative complications included a false joint (15%), inflammation (10%), and forearm deformities (7.5%). Conclusions. In patients with congenital radial club hand type IIIIV, the optimal part of an ulna osteotomy is the proximal section. With a hand deviation of more than 20, osteotomy is performed in the distal section with simultaneous correction of the deformity.
... Overgrowth of the distal ulna relative to the carpus and excessive ulnar deviation reported as the biggest problems of ulnarization. To prevent this complication, Paley recommended to reduce distraction force on the ulnar head by ulnar shortening [26]. ...
Introduction
Radial club hand (RCH) is a rare congenital deformity leading in several functional and psychological problems. However, our knowledge about the long-term functional outcomes of treating RCH is limited. In current study, we investigated the outcomes of centralization and pollicization using second or third metacarpal bone in RCH patients.
Methods
There were 15 hands (13 patients) with RCH underwent centralization and pollicization using second or third metacarpal bone or tendon transfer. The patients aged 1.2 ± 1 years at the time of the surgery. On early postoperative x-rays, the forearm-hand angle was measured. The patients were followed for 6.2 ± 2.3 years. At the final visit, disabilities of arm, shoulder and hand (DASH) score was completed. Furthermore, forearm-hand angle and range of motion of both wrists in sagittal and coronal planes were measured.
Results
The mean of forearm-hand angle increased significantly. In 11 wrists, forearm-hand angle increased only 10 degrees of less. The range of operated wrist was improved in sagittal and coronal planes. The relative range of wrist motion in patients with unilateral deformity in sagittal and coronal planes was 83 ± 11 percent and 61 ± 12 percent. Three patients developed skin necrosis.
Conclusion
Early centralization and pollicization using second or third metacarpal bone can significantly restore the range of motion and function in patients with RCH.
... При данных типах, когда имеется критическое недоразвитие лучевой кости либо ее полное отсутствие, реконструктивные операции направлены на стабилизацию кисти на локтевой кости. Операцией выбора в этом случае, по данным литературы, является центрация кисти [4][5][6]. Значительно реже встречаются I и II типы косорукости. Пациенты с лучевой косорукостью I типа не нуждаются в оперативном лечении на предплечье. ...
Введение. Врожденная лучевая косорукость — это порок развития верхней конечности, при котором наблю-дается продольное недоразвитие предплечья и кисти по лучевой поверхности. При данном пороке отмечается разная степень недоразвития лучевой кости — от незначительной гипоплазии до полного ее отсутствия. Су-ществует более 50 методов хирургического лечения деформаций предплечья в зависимости от степени недо-развития лучевой кости.Цель исследования — оценка результатов лечения пациентов с врожденной лучевой косорукостью II типа по классификации Bayne и Klug, пролеченных методом микрохирургической аутотрансплантации кровоснабжае-мого эпиметафиза второй плюсневой кости с ростковой зоной и методом удлинения лучевой кости с помощью компрессионно-дистракционного остеосинтеза.Материалы и методы. Проведен ретроспективный анализ результатов лечения 16 пациентов с врожденной лучевой косорукостью II типа по классификации Bayne и Klug (средний возраст пациентов — 4,6±0,9 года), которые наблюдались и получали лечение в отделении реконструктивной микрохирургии и хирургии кисти ФГБУ «НИДОИ им. Г.И. Турнера» Минздрава России в период с 1994 по 2017 г. Первая группа включала паци-ентов, которым производили восстановление дистального отдела лучевой кости методом микрохирургической аутотрансплантации кровоснабжаемого эпиметафиза второй плюсневой кости, включающего ростковую зону. Пациентам второй группы выполняли удлинение лучевой кости методом компрессионно-дистракционного остеосинтеза. Был произведен ретроспективный анализ результатов лечения пациентов в обеих группах.Результаты. Длительность наблюдения составила от 12 месяцев до 10 лет (в среднем — 3,8 года). У пациен-тов первой группы хороший результат был получен в 62,5% случаев. При рентгенологическом обследовании в отдаленном периоде отмечена функциональная активность ростковой зоны пересаженного трансплантата, что проявлялось в увеличении длины восстанавливаемой лучевой кости. У пациентов второй группы хорошего ре-зультата удалось добиться в 50% случаев. При клинико-рентгенологическом обследовании отмечался рецидив девиации кисти, укорочение лучевой кости, что в последующем потребует повторного удлинения последней.Заключение. Настоящее исследование показало, что при реконструкции лучевой кости у пациентов с врож-денной лучевой косорукостью II типа микрохирургическая аутотрансплантация второй плюсневой кости, включающей ростковую зону, имеет преимущества за счет создания зоны роста в дистальном отделе лучевой кости. Однако не стоит исключать удлинения лучевой кости методом компрессионно-дистракционного остео-синтеза при сохранении дистального эпиметафиза и нормально развитых поперечных размеров лучевой кости.Ключевые слова: врожденная лучевая косорукость; хирургическое лечение; микрохирургическая аутотранс-плантация; компрессионно-дистракционный остеосинтез.
... Ulnarization is the first treatment of RCH, which has demonstrated no recurrence or growth arrest. Although this method has its drawbacks such as overgrowth of the distal ulna relative to the carpus and excessive ulnar deviation, the promising results of this method might suggest substitution of this method with the traditional techniques in the near future (20). ...
Background: Congenital radial club hand (RCH), as a rare congenital deformity of the upper extremity, is characterized by a wide spectrum of malformations including radial deviation. Centralization surgery is the standard treatment for severe cases that have been associated with a high rate of recurrence. This study reports the long-term results and recurrence rate of radial deviation following the centralization surgery of RCH.
Methods: The medical records of 13 congenital RCH patients (16 hands), who underwent centralization surgery, were reviewed retrospectively. Hand-forearm angle (HFA), hand-forearm position (HFP), and ulnar bow (UB) were used to assess forearm angles.
Results: The mean age of the patients was 19.4±8.9 months, and their mean follow-up was 62.1±39.9 months. The mean HFA correction was 29.4°±23.9°, the mean HFA recurrence was 13.3°±13.7°, the mean correction of HFP was 13.4±7.3 mm, and the mean recurrence of HFP was 1.4±2.8 mm. The mean UB showed 7.6°±12.5° correction immediately after surgery and a further 3.6°±7.3° at the last follow-up (overall 11.2°±17.6°). A number of 12 out of 13 parents were completely satisfied with the results.
Conclusion: According to our results, an acceptable long-term result is expected after the centralization surgery of RCH. However, the risk of the recurrent radial deviation is high and needs to be optimized in future investigations.
... The first article "The Paley ulnarization of the carpus with ulnar shortening osteotomy for treatment of radial club hand " [1] is a milestone in the specialty, as Dror Paley from West Palm Beach, Florida, USA is publishing his new noble modification for his original technique about Ulnarization in radial club hand management. His technique gained more popularity among the specialists worldwide after it was published in 2008. ...
This article summarizes the content of this special issue of the journal about: deformity correction, limb lengthening and reconstruction.
Radial longitudinal deficiency (RLD) is a multidimensional congenital hand difference encompassing skeletal, musculotendinous, and joint components. Managing RLD remains challenging, with numerous surgical procedures over the past century failing to achieve a stable, mobile, growing wrist without recurrence of the deformity. This review investigates new therapeutic approaches for RLD, delving into genetic, embryological, and histological aspects, including proximal muscle involvement and causes of recurrence.
A notable association between RLD and preaxial polydactyly, linked to aberrations in the Sonic Hedgehog signaling pathway, is highlighted, suggesting a common embryonic origin. Experimental evidence indicates that ectopic Sonic Hedgehog signaling can result in radial aplasia and preaxial polydactyly. Histopathological studies revealed significant muscle abnormalities in RLD, contributing to deformities and recurrences.
Techniques such as preoperative soft tissue distraction show promise, but recurrence rates persist. Alternative surgical procedures, such as vascularized second metatarsophalangeal joint transfer and ulna cuff osteotomy, offer potential improvements by minimizing recurrence and optimizing limb length.
Despite advancements, effective management of RLD requires further research into the interplay between genetic factors, muscle abnormalities, and surgical outcomes. This review underscores the importance of early detection, genetic counseling, and a multidisciplinary approach to enhance long-term functional and aesthetic results for RLD patients.
Congenital radial longitudinal dysplasia remains an ‘unsolved problem' in hand surgery. The challenges presented by the skeletal deficiency of the distal radius and soft tissue dysplasia of the severe radial longitudinal deficiency have been addressed by a number of techniques that aim to stabilize the position of the hand relative to the forearm and optimize forearm growth and hand function. Analysis of hand function and position in these children is difficult because of the abnormal ‘wrist' mechanics, and the published results of the techniques used to date often lack a standardized approach and importantly the perception of function from the patient's perspective. The existing data is reviewed and compared with the results of cohorts from two major congenital upper limb centres. Soft tissue distraction prior to radialization or centralization may offer benefit in ulnar growth and forearm length but there is a need for further research into the long-term functional outcomes of the various techniques available to determine the optimal choice for these children.
Level of evidence: V
Introduction
Over the past 20 years, pediatric orthopaedics has gradually become increasingly sub-specialized. The rapid advancement of pediatric orthopaedic knowledge and surgical techniques has made it more difficult to remain proficient in all areas of the field. Surgeons have also started to identify and concentrate on personal areas of special interest within pediatric orthopaedics. As a result, specific disciplines within pediatric orthopaedics, such as spine, sports, hip, and hand, are now recognized as distinct sub-specialties.
Pediatric limb reconstruction, however, is not universally recognized as a separate sub-specialty. For example, it was not listed as one of the choices of primary clinical interest in the 2017 POSNA member needs assessment. The AAOS also does not provide limb lengthening and limb reconstruction as a separate designation for instructional course lectures or scientific presentations. These omissions may be explained by the fact that limb reconstruction appears to overlap with so many different aspects of orthopaedics. Nevertheless, developing a distinct limb reconstruction program as part of your institution’s orthopaedic strategy should be encouraged. The existence of such programs has been shown to have multiple benefits to the entire orthopaedic department.1
The purpose of this article is to outline the components necessary to build an ideal limb reconstruction practice including the surgeon(s), the clinic team, the operating room team, and practice recommendations (Figure 1). While it may not be possible to have every piece in place from the beginning, this guide will provide a fundamental structure that each surgeon should work towards creating at his/her institution...
Background:
The choice of surgical procedure in severe (Bayne and Klug types 3 and 4) radial longitudinal deficiency (RLD) is contentious. Existing studies have reported varying results with both centralization and radialization procedures. The purpose of this study was to compare the clinical and radiologic outcome of radialization and centralization procedures at a short-to-intermediate–term follow-up for the treatment of types 3 and 4 RLD.
Methods:
Fourteen patients with 17 affected limbs having types 3 or 4 RLD were recruited in this prospective, randomized, controlled trial. After initial application of successive casts for soft tissue distraction, patients were randomized to 2 wrist alignment procedures—centralization and radialization. Clinical and radiologic parameters recorded at stipulated intervals until a final follow-up of 24 months included hand-forearm angle, ulnar bow, forearm length, arm length, total angulation, and range of motion at elbow, wrist, and fingers.
Results:
Centralization was performed in 9 affected limbs, whereas radialization was performed in 8 affected limbs. Nine affected limbs had type 4 RLD, and 8 affected limbs had type 3 RLD. There was no significant difference in the hand-forearm angle in the immediate postoperative period. At 3 months, the radiologic hand-forearm angle increased to 19 degrees in the centralization group, while the radialization group showed an average increase to 4 degrees. This increase in the hand-forearm angle continued at 6-, 12-, and 24-month follow-up assessments. Worsening of the deformity was more in the centralization group, as compared with the radialization group. The forearm length also significantly differed in the 2 groups at 6-, 12-, and 24-month follow-up; however, when adjusted for preoperative lengths, the difference was significant only at 12- and 24-month follow-up.
Conclusions:
At a short-to-intermediate–term follow-up, radialization fares better than centralization in terms of recurrence of deformity and in terms of affecting the forearm length. Longer follow-up with a larger sample size is needed to draw definitive conclusions.
Level of Evidence:
Level I.
Radial club hand deformities are commonly treated with arthrolysis to allow centralization of the ulna. In this retrospective cohort study of 31 hands in 28 patients, we aimed to assess the outcomes of correction using progressive distraction and subsequent percutaneous pinning of the wrist with a corrective ulnar osteotomy. Mean follow-up time was 7 years (range 2 to 20). The angulation of the hand–forearm complex was decreased after each step of the procedure. Mean correction of the angulation was 64°, and the residual total forearm–hand angulation was 12° after completion of the surgery. At the time of bony maturity (four patients), all wrists had fused. Fifty-eight reoperations were required in 31 wrists because of pin migration or breakage, and in addition 18 secondary osteotomies of the ulna were performed. From this study we conclude that distraction and pinning provide satisfactory and stable realignment of the wrist to correct the deformity, but this treatment has drawbacks regarding the high number of reoperations and the loss of wrist mobility.
Level of evidence: IV
Treatment of radial clubhand has progressed over the years from no treatment to aggressive surgical correction. Various surgical methods of correction have been described; Centralization of the carpus over the distal end of the ulna has become the method of choice. Corrective casting prior to centralization is an easy and effective method of obtaining soft tissue stretching before any definitive procedure is undertaken. Moreover, it helps put the limb in a correct position. The outcome of deformity correction by serial casting / JESS distractor followed by centralization is discussed.
In a prospective study, of 17 cases with 18 radial clubhands of Heikel's Grade III and IV (with average age 11 months (range 20 days - 24 months) with M:F of 2.6:1, were treated by gradual soft tissue stretching using corrective cast (14 cases) and JESS distraction (4 cases), followed by centralization (16 cases) or radialization (2 cases) and tendon transfers.
The average correction attained during the study was 71 degrees of radial deviation and 31 degrees of volar flexion. The average third metacarpal to distal ulna angle in anteroposterior and lateral view at final follow-up was 7 degrees in both views. Angle of movement at elbow showed a small increase from 99 degrees to 101 degrees during the follow-up period. However, the range of movement at fingers showed increase in stiffness during the follow-up. No injury occurred to the distal ulnar epiphysis during the operative intervention. The results at the final follow-up, at the end of 2 years were graded on the basis of the criteria of F.W. Bora, and of Bayne and Klug. Considering the criteria of F.W. Bora, satisfactory result was shown by nine of the 18 hands (50%) while 16 out of 18 hands (89%) showed good or satisfactory result based on deformity criteria of Bayne and Klug.
The management of radial clubhand by gradual corrective cast or JESS distractor followed by centralization and tendon transfers in children is an acceptable method of treatment with consistently satisfactory results, both functional and cosmetic.
Radial longitudinal deficiency, also known as radial club hand, is a congenital deformity of the upper extremity which can present with a spectrum of upper limb deficiencies. The typical hand and forearm deformity in such cases consists of significant forearm shortening, radial deviation of the wrist and hypoplasia or absence of a thumb. Treatment goals focus on the creation of stable centralized and functionally hand, maintenance of a mobile and stable wrist and preservation of longitudinal forearm growth. Historically centralization procedures have been the most common treatment method for this condition; unfortunately centralization procedures are associated with a high recurrence rate and have the potential for injury to the distal ulnar physis resulting in a further decrease in forearm growth. Here we advocate for the use of a vascularized second metatarsophalangeal joint transfer for stabilization of the carpus and prevention of recurrent radial deformity and subluxation of the wrist. This technique was originally described by the senior author in 1992 and he has subsequently been performed in 24 cases with an average of 11-year follow-up. In this paper we present an overview of the technique and review the expected outcomes for this method of treatment of radial longitudinal deficiency.
Previous methods of treatment of radial club hand (RCH) have resulted in recurrent deformity and growth arrest of the distal ulna in a very high percent of cases. Ulnarization (procedure developed by Paley in 1999) is the first treatment of RCH to demonstrate no recurrence or growth arrest. Through a volar approach, the entire carpus is acutely transferred to the ulnar side of the ulna.
Die Arbeit enthlt zunchst eine Darstellung der Morphologie radialer Klumphnde und sttzt sich dabei auf frher mitgeteilte pathologisch-anatomische Befunde sowie eigene Beobachtungen an 89 Patienten mit Radiushypoplasien, partiellen und totalen Radiusaplasien einschlielich 19 operativ behandelter Flle. Bemerkenswert am eigenen Krankengut ist einmal die Hufung der Geburtsjahrgnge 1960–1962, dann die relativ hohe Rate von Synostosen beider Unterarmknochen sowie die groe Zahl von Daumenfehlbildungen, deren Schwere meistens auch mit dem Grade der Rckbildungsstrungen des Radius zunimmt. Die Verschmelzungen der Unterarmknochen bleiben in leichteren Fllen auf das proximale Radioulnargelenk lokalisiert (radio-ulnre Synostosen). Schwereren Prozessen dagegen liegen ausgedehnte, bis zum einknochigen Unterarm reichende Synostosen oder Knochenbrcken an zwei Unterarmabschnitten gleichzeitig zugrunde. Fr diese uerst seltenen Total- und Zweifachsynostosen wird je ein Beispiel angefhrt. Im brigen besttigen die weiteren Befunde an Knochen und Weichteilen erneut, da die radiale Klumphand trotz der Flle unterschiedlicher Erscheinungen als Hemmungsmibildung vorwiegend des radialen Armstrahles zu betrachten ist.Wesentliche Gesichtspunkte fr die Aufstellung des Behandlungsplanes sind frhzeitiger Behandlungsbeginn, Bestimmung des funktionellen Wertes und der weiteren Prognose der Klumphand, Vorrang konservativer Methoden und Bercksichtigung der Gesamtsituation des Patienten.Unter den konservativen Verfahren verdient die Behandlung der Ellbogenstrecksteifen durch stndige Fixation des Handgelenks in Korrekturstellung besondere Erwhnung. — Die Voraussetzungen zur operativen Therapie der Klumphand sind gegeben, wenn sich die Deformitt passiv vollstndig ausgleichen lt, wenn gute Beweglichkeit im Ellbogengelenk besteht und die Verlagerung der Handwurzel proximal-radial des distalen Ellenendes nicht mehr als ca. 1 cm betrgt, wenn zustzliche Daumenersatzoperationen geplant sind, der Operateur den besonderen Anforderungen der Handchirurgie gewachsen ist, wenn die Mglichkeiten einer speziellen postoperativen Behandlung gegeben sind und wenn durch Funktionsteste erwiesen ist, da aus der Einschrnkung der Handgelenks beweglichkeit keine Herabsetzung der Gebrauchsfhigkeit der Gliedmae resultiert. Ziel der eigenen Operationsmethode ist nmlich die Fixation der Hand in Funktionsstellung. Dies wird durch Implantation des distalen Ellenendes in den Carpus zu erreichen versucht.
Hauptgefahren des Eingriffes sind Schden an der Epiphysenscheibe der Elle. Mierfolge drohen aber auch insbesondere bei zu kurzfristiger oder ungengender Ruhigstellung der operierten Hand und Nichtbeachtung der Indikationsrichtlinien. Mit dem Hinweis auf unsere bisherigen, andernorts publizierten Ergebnisse — postoperative Beobachtungszeiten bis zu fast 4 Jahren — soll zum Ausdruck kommen, da eine endgltige Beurteilung des bisher berwiegend erfolgreich angewandten Verfahrens verfrht ist und da deshalb weiter Zurckhaltung mit Klumphandoperationen gebt werden soll.Initially in this paper the today known anatomo-pathological findings in congenital club-hands and the own observations at 89 patients with hypo- and aplasia of the radius are described: It is possible to place the developmental disturbances of the radius in a teratological order, reaching from the light hypoplasia over the partial to the total aplasia. Particularly striking in the own casuistics are the cases with processes of fusion between the two forearm bones. The synostoses also show different stages of severity. One case of double synostosis and one case of total synostosis are exceptionally rare observations. The developmental anomalies of the ulna, of the hand skeleton as well as the malformations in the soft tissues of the affected limb are demonstrating that the club-hand is no localized only the skeleton of the radius afflicting deformity.In the therapy at first are to regard the possibly existing further abnormalities of other limbs and organs as well as functional view points of the malformed extremity. On principle, conservative and operative measures are in discussion. The own operative methode, carried out in 19 cases, consists in the implantation of the distal end of the ulna into the carpus. By opening of the junction between the lunatum and triquetrum like a leaf of a door and by resection of the head of the capitatum a suitable gap for the capitulum ulnae is fitted. The carpalia put at both sides to the distal end of the ulna have to improve the stabilisation of the hand. Faults and risks of the operation mainly rest on a wrong indication, technique and postoperative care with the consequence of epiphyseal damage and relapse of the club-hand. On the base of the hitherto existing results disturbances of growth generally must be considered as avoidable consequences of operation. The paper finally contains references to the therapy of the hypo- and aplasies of the thumb as well as of the congenital contractures of the long fingers frequently accompanying the club-hand.Les rsultats des tudes anatomo-pathologiques de mains-botes congnitales connus jusqu' prsent et les propres observations de 89 patients atteints d'hypoplasies et d'aplasies du radius seront d'abord dcrites dans cette publication: Les dysontognses du radius peuvent tre classes dans une srie tratologique s'tendant d'une lgre hypoplasie une hypoplasie partielle et jusqu' une aplasie totale. Les cas accompagns de processus de fonte osseuse entre les deux os de l'avant-bras rapports dans nos observations sont particulirement frappants. Les synostoses montrent galement diffrents degrs de gravidit. En ce qui concerne les synostoses doubles, extrmement rares, ainsi que les synostoses totales une observation personnelle est communique pour chaque cas. Les dysontognses cubitales, du squelette de la main ainsi que les difformits des parties molles des membres atteints montrent que la main-bote est une difformit ne se rapportant pas seulement qu'au squelette du radius. Lors du traitement, il faut avant tout tenir compte des ventuelles difformits d'autres membres et organes ainsi que de la valeur fonctionnelle du membre malform. En principe, traitements conservateurs et interventions prteront discussion. Dans 19 cas, le type d'intervention excut consiste en une implantation de l'extrmit infrieure du cubitus dans le carpe. Aprs le relvement sous la forme d'une porte deux battants du semi-lunaire et du pyramidal et aprs avoir rsqu la tte du grand os, on aura ainsi cr une enclave pour y recevoir la tte du cubitus. Les os du carpe ainsi disposs de part et d'autre de l'extrmit infrieure du cubitus doivent apporter un meilleur appui de la main. Les erreurs et les risques opratoires seront avant tout le fait d'une mauvaise indication ou celui d'une mauvaise technique ou de soins postopratoires dfaillants avec tout ce que cela comporte comme consquencese, savoir des troubles piphysaires et des rcidives de main-botes. D'une facon gnrale, d'aprs les rsultats jusqu'alors obtenus, ces troubles de la croissance doivent tre considrs comme des suites opratoires vitables. En conclusion, cette publication comporte des indications concernant le traitement des hypoplasies et des aplasies du pouce qui accompagnent souvent la main-bote ainsi que celui des contractures congnitales des autres doigts.
Based on a review of the embryology, genetics, and anatomy of radial club hand, it is suggested that damage to the apical ectoderm on the anterior aspect of a developing limb bud leads to the deformity. Study of the families of thirty-five children with radial club hand suggested that the condition is not genetically patterned. The anatomical findings and associated congenital abnormalities in the cases known to be related to thalidomide and in those in which thalidomide was not a factor were similar except that the incidence of other skeletal deficiencies was higher in the thalidomide group. Thirty-one of the 117 radial club-hand deformities (in sixty-eight patients) under my personal supervision were treated by centralization of the carpus on the ulna with satisfactory improvement of the deformity. In three cases wrist deformity recurred mainly in a volar direction, apparently the result of muscle imbalance. No significant impairment of ulnar growth occurred and straightening of the wrist did not affect function adversely. Pollicization of the index finger was done on twenty-eight occasions. Although problems developed in the early cases, these can be avoided using the methods described and the operation can improve both function and appearance. A scheme of management is recommended.
An appreciation of the clinicopathologic presentation of children with radial dysplasia (radial club hand) is important. Management decisions depend upon the degree of radial ray defect present; in most cases these are surgical. Current surgical options favor centralization without resection of carpal bones, stabilized by tendon transfers. When the thumb ray is deficient, pollicization is a rewarding procedure.
Sixty-four patients with 101 radial deficiencies form the basis of this review of anatomy, treatment, and long-term follow-up. Average follow-up was 8.6 years, with a range of 1 to 27 years. A radiologic classification of radial deficiencies is presented. The preferred method of treatment is centralization. The goal is to create a centralized, cosmetically and functionally improved hand, yet maintain wrist motion. There were 21 good, 20 satisfactory, and 10 unsatisfactory results. Good and satisfactory results all had adequate preoperative stretching, proper surgical technique, and postoperative brace compliance. Failure to adhere to outlined principles of soft tissue release and adequate centralization resulted in the unsatisfactory results.
"Radialization," a new technique for operative treatment of the radial club hand, is presented. It has been successfully used in 30 hands (23 patients) since 1979. It is named "radialization" because after all fibrotic tissues are excised, the hand and radial carpal bones are placed over the distal end of the ulna; the hand is fixed with a Kirschner wire in a position of moderate ulnar deviation. Usually, no carpal bones need to be removed. The improved mechanical forces are further stabilized by transposition of the radial wrist extensor and flexor to the ulnar side; this favors a better muscle balance. The optimal age for surgery is between 6 and 12 months.
1.1. A series of forty-one children with radial club hand deformity is described. In twenty-two this was bilateral.2.2. The importance of early splintage to prevent soft tissue contracture is stressed. A simple ratchet type of splint, which has proved effective, is described. Splintage is only required at night.3.3. Operation by centralisation of the carpus over the ulna has proved satisfactory in correcting the deformity and producing wrist stability. A limited range of wrist movement is still possible and so far there does not appear to have been any detrimental effect on the growth of the lower ulnar epiphysis. A satisfactory correction can be maintained by a soft tissue periosteal flap.4.4. While the expected improvement in appearance has been achieved the loss of function which had been anticipated has not materialised.5.5. Surgical correction of the wrist deformity should not be contemplated unless there is active flexion of the elbow to about ninety degrees. In many cases where the elbow is stiff in extension at birth there is a tendency to gradual loosening of the elbow during the first year or two. Where this does not occur a posterior release of the elbow capsule (as carried out in arthrogryphosis) can be successful.6.6. Pollicisation of the index finger should be seriously considered. The result will depend to a considerable extent on the structure and function of the index finger.7.7. In order to get the best possible functional result early operation should be considered.
A modified centralization procedure for radial club hand caused by partial or total absence of the radius is presented. This technique differs from the methods presently used in that no carpal bones are resected. Adequate release of the wrist from the fibrotic radial anlage is provided through two Z-plasty incisions to allow the hand to move easily to the new position. Stabilization is then obtained with a Kirschner wire placed via the lunate and capitate into the long finger metacarpal and then directed in a retrograde fashion into the ulna. A total of 12 centralization procedures with the above technique have shown very good results. Long-term follow-up results demonstrate remodeling of the ulna, which becomes broad in its distal end, resembling a radius.
In 1970, the results of the treatment of fourteen patients with radial club hand were reported. Ten years later, we re-examined ten of these patients. There was little change in the measurements of ulnar bowing or ulnar length over the intervening decade, and the treated patients retained the cosmetic and functional improvement previously described. They also adapted satisfactorily to performing activities of daily living.
Centralization arthroplasty via a transverse ulnar approach was used to correct radial clubhand deformity in 21 children. The patients were evaluated quantitatively by measurements of hand-forearm angle and hand-forearm position.
A new incision is described for the correction of radial club hand. By the use of a bilobed flap, redundant skin is transferred from the ulnar side of the wrist to the radial side, where there is tension as the wrist becomes straight. The flap has been shown to be safe and effective in six consecutive cases, and access to the wrist and surrounding soft tissue structures is excellent.
This paper presents a different technique of treatment for Bayne type IV radial club hand using a microvascular joint transfer in order to reconstruct the absent half of the wrist joint, aiming for better movement and stability at the wrist joint with preservation of longitudinal growth. The method uses preoperative soft tissue distraction to obtain proper alignment of the hand on the ulna before the second metatarsophalangeal joint with the whole metatarsal bone is transplanted. The treatment takes about 4 months and the optimum period for surgery is during the second year of life. Pollicization is added later in the normal manner.
The new technique has been used in 12 cases by the author since 1987 and the results of the first nine cases are reported with a mean follow-up of 6 years. This technique appears to be promising but is demanding because of the microvascular joint transplantation at an early age.
The experience with congenital radius deficiency, or radial hemimelia, at the Shriners' Hospital for Children, Los Angeles Unit, was reviewed. A cohort of 29 limbs in 23 patients was identified with an average follow-up period of 50 months. Radiographic parameters were assessed using the hand-forearm angle, hand-forearm position, and ulnar bow. We compared radialization to modified centralization, assessed the efficacy of ulnar osteotomy, and assessed the effect of age, preoperative deformity, ulnar osteotomy, and Bayne's type on the final result. Revisions were noted and a survivorship analysis performed. The cohort had statistically significant correction of hand-forearm angle and hand-forearm position. Radialization was similar to modified centralization in the final outcome. Ulnar osteotomy was an efficacious way to correct ulnar deformity. Age, preoperative deformity, performance of an ulnar osteotomy, and Bayne's type did not affect the final wrist position. Survivorship analysis was performed using revision as the end point, with a survivorship rate at 5 years of 67%. Significant risk factors for revision included radial or positive hand-forearm angle and young age at the time of the index procedure. There was a suggestion that small postoperative hand-forearm position, or radial translation, increased the risk of revision. Preoperative deformity, performance of an ulnar osteotomy, and Bayne's type did not affect the risk of revision. These data offer support for the hypothesis that a more ulnar translation and an ulnar angulation of the wrist is a means of reducing the radial lever arm and thus the incidence of deformity recurrence and need for revision.
Fourteen children representing 19 cases of radial clubhand had centralization of the carpus on the distal ulna during an 18-year period. Age at the time of the initial surgery averaged 3.2 years (range, 0.7-8.1 years) and the follow-up periods averaged 6.5 years (range, 1.5-22.2 years). There were 16 type IV radial and 3 type III clubhands. Preoperative, postoperative, and follow-up x-rays were used to determine the initial deformity, amount of surgical correction, and degree of recurrence. The total angulation (the combination of the radial deviation of the hand and the ulna bow) was measured. The average preoperative angulation measured 83 degrees (range, 55 degrees to 110 degrees ). Centralization corrected the angulation an average of 58 degrees (range, 15 degrees to 95 degrees ) to an average immediate postoperative total angulation of 25 degrees (range, 5 degrees to 60 degrees ). At the final follow-up examination there was a loss of 38 degrees (range, 5 degrees to 105 degrees ) and the total angulation increased to an average of 63 degrees (range, 20 degrees to 120 degrees ). The difference between the preoperative, postoperative, and follow-up angles was statistically significant. There was a significant correlation between the preoperative angle and the final angle, the preoperative angle and the amount of correction, the amount of correction obtained at surgery and the recurrence of the deformity, and the age at time of initial surgery and the amount of recurrence.
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