Bone Lengthening in the Pediatric Upper Extremity

Farr, Sebastian MD; Mindler, Gabriel MD; Ganger, Rudolf MD; Girsch, Werner MD

Journal of Bone & Joint Surgery - American Volume:
doi: 10.2106/JBJS.16.00007
Current Concepts Review
Abstract

Bone lengthening has been used successfully for several congenital and acquired conditions in the pediatric clavicle, humerus, radius, ulna, and phalanges.

Common indications for bone lengthening include achondroplasia, radial longitudinal deficiency, multiple hereditary exostosis, brachymetacarpia, symbrachydactyly, and posttraumatic and postinfectious growth arrest.

Most authors prefer distraction rates of <1 mm/day for each bone in the upper extremity except the humerus, which can safely be lengthened by 1 mm/day.

Most authors define success by the amount of radiographic bone lengthening, joint motion after lengthening, and subjective patient satisfaction rather than validated patient-related outcome measures.

Bone lengthening of the upper extremity is associated with a high complication rate, with complications including pin-track infections, fixation device failure, nerve lesions, nonunion, fracture of regenerate bone, and joint dislocations.

Author Information

1Department of Pediatric Orthopaedics and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Vienna, Austria

E-mail address for S. Farr: Sebastian.farr@oss.at

Article Outline

Since the technique of distraction osteogenesis was first reported in the early twentieth century, it has gained wide popularity among orthopaedic surgeons1,2. Its use allows for gradual elongation of foreshortened extremities and/or correction of bone deformities, with the aims of restoring alignment and improving function and appearance. Although the majority of the literature has described applications in lower-limb deformities, the technique and its principles might well be applied to several pediatric upper-limb conditions that require functional improvements through an increase of bone and soft-tissue length. This review article focuses on the historical background, biological basics, technical issues, results, and possible complications associated with bone-lengthening procedures in the pediatric upper extremity.

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History of Bone Lengthening

The first techniques for bone lengthening in a prenarcotic era were somewhat aggressive and painful procedures2. In 1905, Codivilla reported a femoral lengthening technique with a femoral osteotomy and acute lengthening via traction using a transcalcaneal nail1. Other milestones of leg lengthening were established by Ombredanne who described the first gradual but fast distraction using an external fixator in 19133, by Putti who reported slow femoral distraction lengthening in 19214, and by Wittmoser who described the first use of a ring fixator in 19535. A safe way to correct even severe complex deformities was later developed by Ilizarov, using a modular ring fixator and tensioned wires6.

The use of bone-lengthening techniques in the upper extremity is a rather recent development with few reports and studies published until now. Since 1978, case reports and small series of humeral lengthening procedures in children using a monolateral external fixation device have been published7-13. The Wagner device, which consisted of a telescoping device connecting the Schanz screws proximal and distal to the osteotomy site, was mostly used for humeral lengthening, but forearm bone lengthening has also been attempted14. In 1986, Pritchett reported ulnar lengthening with either acute lengthening (and iliac bone graft) or gradual distraction in children and adolescents with multiple hereditary exostoses15. The use of the Ilizarov technique for pediatric humeral lengthening became popular for several pathologic conditions6,13,16-19. Early reports showed that in addition to humeral lengthening, the Ilizarov technique is capable of gradual lengthening of the forearm15,20-22, metacarpals, and phalanges23-27. Several years later, hexapod devices such as the Taylor spatial frame (TSF; Smith & Nephew) were used for the correction of upper-extremity deformities in children28. Recently, first reports of humeral lengthening using motorized intramedullary nails were published29. The growing knowledge about mechanical factors (e.g., fixator rigidity) and biological factors (e.g., effect of smoking on bone healing and callus stimulation through biological agents) has helped to improve clinical results achieved with bone lengthening.

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Biology of Distraction Osteogenesis

Ilizarov explained in detail the concept of “tension-stress effect” for successful lengthening, showing that the application of slow, steady traction can metabolically activate bone and thereby induce bone regeneration6. First, the periosteum, bone marrow, and medullary blood supply have to be preserved. Furthermore, a less invasive and periosteum-sparing osteotomy should be performed. The stability of external fixation and a lengthening of 1 mm/day in 4 increments after a 5-day latency period are other important aspects of the Ilizarov lengthening concept30,31. Moreover, different concepts of biomechanical and cellular involvements in the process of bone regeneration have recently been developed32-34.

In addition to inducing bone regeneration, assessment of the regenerate bone is a crucial part of the postoperative follow-up of patients undergoing bone lengthening. Different imaging methods35, such as ultrasonography36-38, magnetic resonance imaging39, computed tomography40, and scintigraphy41, have been used in clinical and experimental settings. The majority of studies used radiography-based measures such as densitometry42, dual x-ray absorptiometry43-45, or the pixel ratio value46,47 to assess new bone formation. Thus, the current gold standard for clinical follow-up after lengthening is radiographic analysis and interpretation of sufficient bone cortex formation with plain radiography. A few radiographic classifications have been developed48-50 to describe the status of the regenerate bone for lower-limb lengthening. Moreover, the healing index (the time from the operation until full load-bearing for each 1 cm of regenerate bone) is important to describe early consolidation, delayed union, and nonunion51. As with lower-limb lengthening, patient age has been shown to have a significant effect on healing time in metacarpal lengthening52,53. Time to consolidation seems to be longer in the forearm than in the femur or tibia54, shorter in the humerus than in the femur55,56, and generally very good in the humerus8.

Concerning the optimal speed of distraction, Ilizarov31 discovered that a lengthening rate of 1 mm/day was very effective in a canine model; he also found a distraction rate of 0.25 cm 4 times a day to be the most effective one. In addition to speed of distraction, the exact latency period, according to Aronson, until the definitive start of distraction is another crucial part of lengthening and was proposed by Ilizarov to begin 5 to 10 days after osteotomy6,51. However, not every long bone has the same biological and mechanical requirements51. Therefore, adjustments to the speed and start of distraction have to be considered. For the pediatric upper extremity, most reports have noted distraction rates of <1 mm/day, except for the humerus, which is lengthened by 1 mm/day in most reports12,20. The rate of distraction has to be chosen on the basis of the bone, the individual patient, and concomitant diseases and might have to be adjusted during radiographic follow-up.

Different biological agents such as platelet-rich plasma57, intravenously administered bisphosphonates58, bone morphogenetic proteins59,60, and low-intensity pulsed ultrasound61,62 can locally or systemically stimulate bone growth. However, the evidence for successful use of these agents in the pediatric population is very limited, and they have not been used specifically for upper-extremity lengthenings63-65. Furthermore, experimental studies in animals have shown that mechanical stimulation by a piezoelectric loader, in particular elbow loading, leads to an increase in bone length and weight, bone mineral density, and bone mineral content66.

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Techniques and Devices

Bone lengthening can be achieved by using unilateral fixators, circular frames or hexapod systems, or intramedullary devices. Moreover, combinations of a circular frame with intramedullary wires have also been reported67. Special devices68,69 and techniques (e.g., step-cut osteotomy)70 have been developed for lengthening of phalanges.

The application of a unilateral fixator is preferred when increasing the length of a single bone is the primary goal of the procedure and no or only minimal axial bone deviation is evident. Common devices for this purpose include the Limb Reconstruction System (Orthofix)71, MiniRail System (Orthofix)72, Multi-Axial Correction external fixation system (MAC frame; EBI/Biomet)73,74, and Pennig Fixator (Orthofix)75.

More complex deformities that include foreshortened and malaligned bones might require lengthening and realignment with the use of a circular frame. Systems such as the classic Ilizarov frame enable the surgeon to simultaneously lengthen 2 bones (e.g., ulna and radius)76, perform asymmetric forearm lengthening77, or achieve correction of a single-plane deformity using hinges78. The Ilizarov frame can also serve as a rotational apparatus, as described by Rubin et al.79. Small-sized circular frames (e.g., Ilizarov Small Bone Fixation System; Smith & Nephew) are routinely used to manage conditions such as radial longitudinal deficiency and deformities after physeal arrest in toddlers and infants80. Hexapod devices (e.g., TSF)81 became very popular for the correction of lower-limb deformities. However, they can also be used to successfully treat complex 3-dimensional upper-limb conditions28. Bone lengthening might furthermore be enhanced by the addition of intramedullary Kirschner wires82,83 or elastic stable intramedullary nails67,84, which “guide” the formation of adequate callus and provide additional stability after bone formation.

Recent developments in the sector of limb lengthening include motorized intramedullary lengthening devices, which are currently mainly used in femora and tibiae85,86. Nevertheless, the first reports mentioning their use in pediatric upper extremities, mostly in foreshortened humeri, have emerged29.

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Indications for Bone Lengthening in the Pediatric Upper Extremity

Numerous surgical indications have been reported for congenital and acquired or posttraumatic conditions (Tables I through IV). Humeral length deficiencies of >5 cm can be considered an indication for restoration of upper-limb length for the improvement of functionality and, especially, appearance21. Moreover, humeral lengthening can become necessary to maintain physiological body proportions whenever the lower limb is lengthened >6 cm21. Common indications for humeral lengthening include congenital humeral varus deformity87,88, postinfectious growth arrest12,21,73,74,89-91, growth disturbances after trauma28,67,73,90,91 or bone cyst formation12,89, achondroplasia21,74,89,90,92, and neoplasms such as enchondromatosis (Ollier disease)21,28,78,89.

Regarding distraction osteogenesis of the pediatric forearm, the most common indications are radial and/or ulnar longitudinal deficiency20,76,84,93-97 (Figs. 1-A, 1-B, and 1-C), multiple hereditary exostoses14,71,76,97-109 (Figs. 2-A and 2-B), and posttraumatic need for restoration of forearm length14,77,80,84,107-112. Rare cases of humeroradioulnar or radioulnar synostosis79,113, Madelung deformity112, and congenital pseudarthrosis of the forearm114 have also been addressed with external fixators. Children with congenital below-the-elbow amputations might also benefit from elongation of the short forearm stumps because prosthetic fitting might otherwise be difficult to achieve115,116.

The pediatric hand has been another frequent target for bone lengthening since the beginning of this technique. Metacarpal lengthening in cases of traumatic (thumb) bone loss24,69,83,117-122, brachymetacarpia53,123-127, symbrachydactyly82,126,128,129, and syndromes (e.g., Apert)130 have thus far been the most noteworthy indications reported in the literature. Phalangeal lengthening procedures for even rare disorders such as Kirner deformity have been successfully performed117,118,131. Recently, lengthening of the clavicle for congenital dysplasia of the clavicle has been described to improve appearance and function and to diminish pain132,133.

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Clinical Results

The current literature on this topic is almost exclusively characterized by retrospective case series (Level-IV evidence). Moreover, much heterogeneity is evident in the literature concerning good clinical outcome for the investigated population. Clinical outcome measures such as appropriate scores for the upper limb are rarely reported; the vast majority of authors have defined success by the amount of bone lengthening shown radiographically, joint motion after lengthening, and subjective patient satisfaction. However, such data, especially data on patient satisfaction, are rarely collected in a standardized fashion using validated psychological instruments106.

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Lengthening of the Humerus

The purpose of humeral lengthening is to diminish any functional deficits (which are often only subtle) and improve the appearance and thus the psychological well-being of the patient. Several reports on lengthening of pediatric humeri have shown the ability to achieve impressive length gains of up to 11 or even 13.5 cm (Table I)21,134. Pawar et al.74 and Ruette and Lammens89 reported their results after the lengthening of 15 limbs in 11 patients and 26 limbs in 17 patients, respectively. Average bone length gains of 7 cm and 8.8 cm, respectively, were observed in their retrospective series. Despite this relatively large amount of lengthening, the healing index was acceptably low at approximately 32 and 31 days/cm, respectively, emphasizing the good ossification potential of this tubular long bone. Despite a host of surgical complications encountered in the series presented by Ruette and Lammens after a minimum follow-up of 2 years, they stated that all patients returned to their previous level of sports and would therefore undergo surgery again89. Excellent mean Disabilities of the Arm, Shoulder and Hand (DASH) scores with no shoulder or elbow stiffness were observed at the 1-year follow-up74. The authors also emphasized that monolateral fixators are more convenient than circular frames for humeral lengthening, considering that the shoulder needs no abducted position during rest (Figs. 3-A and 3-B). Also, circular frames such as the TSF can be used with acceptable patient comfort whenever medially open, two-thirds rings are being used28. Even bilateral humeral lengthening procedures were successful in enabling patients to perform activities of daily living, such as the ability to perform perineal hygiene, wash one’s face, brush teeth, and handle a wheelchair92. Nevertheless, these lengthening procedures are accompanied by a substantial risk of complications.

Bernstein et al. reported that stumps of congenital and acquired above-the-elbow amputations can also be successfully elongated to improve both nonprosthetic function and prosthetic fitting135. Large amounts of increase in the humeral stump length of up to 120% of the original length were observed135. Humeral bone healing can furthermore be enhanced if flexible intramedullary nails are added67. Popkov et al. (in a Level-II study) observed a decrease in the humeral healing index from 21.3 to 19.4 days/cm (after monofocal osteotomy and use of a flexible intramedullary nail [FIN] for a discrepancy with a congenital etiology), 19.8 to 15.8 days/cm (after monofocal osteotomy and FIN for a discrepancy with an acquired etiology), and 15.2 to 11.9 days/cm (after bifocal osteotomy and FIN for a discrepancy with an acquired etiology)67. The authors speculated that bone formation during the lengthening process was likely stimulated by the sliding motion of the intramedullary nails, thus leading to the decreased healing index and eventual decreased time of external fixation.

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Lengthening of the Clavicle

Recently, a report on clavicular lengthening in 5 syndromic patients (7 clavicles) with congenital clavicular hypoplasia and abnormal scapular position showed promising results regarding improvement in function (100% of the patients were improved), appearance (80% were improved), shoulder motion (80% were improved), pain (60% were improved), breathing (20% were improved), and shoulder stability (20% were improved) for this indication (Table II)132,133.

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Lengthening of the Forearm

Symmetric or asymmetric lengthening of a short or deviated forearm may be indicated to realign the radius and ulna in order to improve function, improve motion, or reduce pain. Recent studies have shown good clinical results after forearm balancing procedures in patients with hereditary multiple exostosis disease71,98,104. Ettl et al. achieved a mean ulnar length gain of 26 mm in 9 patients and 10 extremities98. Postoperative forearm rotation improved in 4 extremities, was unchanged in 4, and deteriorated in 2 extremities. Similar results concerning forearm rotation (5 of 12 children had improvement, 4 were unchanged, and 3 worsened) and wrist range of motion (1 of 12 children had improvement, 10 were unchanged, and 1 worsened) were observed by Vogt et al. after a mean follow-up of 24.6 months71. However, mild recurrence of the deformity appeared at the mid-term period71. Concomitant procedures (osteochondroma excision and osteotomy of the radius) are commonly performed to achieve the desired clinical and radiographic outcomes71,98-100,102-104.

Distraction osteogenesis has also been used in patients with radial longitudinal deficiency to enhance activities of daily life (Figs. 1-A, 1-B, and 1-C). Raimondo et al. subjectively observed that children were able to play on monkey bars, lift weights, handle overhead objects, and perform hobbies and sports without adaptions postoperatively76. Large lengthening amounts of up to 15 cm have therefore been reported95. Although good aesthetic results can be expected, some deterioration of the hand-forearm angle and ulnar bowing at 1.0 to 8.5 years after deformity correction and ulnar lengthening were reported by Farr et al.93. If lengthening procedures are begun at a very young age, repetitive procedures might be necessary to achieve a balanced forearm length because of growth-related recurrence of deformity94. Despite high patient satisfaction after forearm lengthening in patients with radial longitudinal deficiency, the lengthened extremity is still often used only in an assistive manner76. Moreover, potential complications such as finger stiffness or neurapraxia may occur.

External fixation has been used for gradual correction of severe hyperpronation deformity in congenital radioulnar synostosis from a mean value of 100° to a mean of 15° of supination79. This method can also be used to gradually lengthen the forearm (50 to 82 mm) in preparation for a second-stage vascularized bone transfer96,114. Bone lengthening has therefore been successfully applied for indications such as congenital pseudarthrosis of the forearm, radial longitudinal deficiency, and neonatal compartment syndrome.

Postinfectious forearm deformities were addressed by Zhang et al., who reported marked improvement of clinical parameters (radial deviation, wrist flexion-extension, and grip strength) for 13 children after deformity correction with a monolateral fixator136. Despite delayed bone healing in 3 patients, all patients were satisfied with the intervention, with no growth disturbances observed after a mean follow-up of 4.5 years136. Whenever posttraumatic growth arrest of the distal radial physis is evident, gradual distraction osteogenesis can also be considered80,110. Two small case series revealed good clinical outcomes after gradual correction of the radius. Page and Szabo80 reported mean postoperative scores of 11 on the DASH questionnaire, 76 for the Mayo wrist score, 82 on the Short Form-12 questionnaire, and 1 on the visual analog scale for pain. Moreover, Gündeş et al.110 showed a mean postoperative DASH score of 2 and a mean Mayo wrist score of 89. Villa et al. reported that 11 of 12 patients had functional and cosmetic improvement after a length gain of up to 13 cm; 9 of the 12 patients were further noted to be in better psychological condition after achieving the final forearm length77. As reported by Jager et al., the healing index can be reduced by approximately 30% to 22.2 days/cm of regenerate bone if intramedullary wires are added84 (Table III).

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Lengthening in the Hand

Metacarpal bones and phalanges are lengthened mainly for the purposes of improved appearance or index-to-thumb pinch. Bozan et al. used monolateral and circular frames to elongate 18 metacarpals for a mean length gain of 16.5 mm in children with brachymetacarpia53 (Table IV). All patients younger than 18 years showed significantly accelerated bone healing (decreased healing index; p = 0.002)53. Erdem et al. stated that to avoid metacarpophalangeal joint dislocation, metacarpal lengthening should not exceed 40% of the original bone length123. In another study, all patients were satisfied after adequate digital length was eventually achieved using postdistraction iliac crest bone-grafting126. Miyawaki et al., in contrast, showed increased pinch power (superior to the contralateral side) after digital lengthening using a monofixator and intramedullary Kirschner wire82. Digital lengthening over an intramedullary Kirschner wire also provided a higher percentage of bone lengthening (70% of the original length versus 49%) at the cost of an increased complication rate82. Additional interventions such as web space deepening, bone grafting, or various flaps might be necessary120. Whenever digital lengthening is performed for congenital hand malformations, different growth patterns and different amounts of remaining growth have to be considered94. Although this procedure may often be accompanied with substantial problems such as nonunion, necessitating subsequent bone-grafting in up to 26%69, digital lengthening may lead to improved outcomes such as better key pinch122 and grasp69 and a high rate of patient satisfaction134.

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Complications

Complications are generally very common in association with bone-lengthening procedures and have been further subdivided by Paley into problems (manageable without further surgery), obstacles (further surgery necessary), and complications (intraoperative injuries)137. The most common complications are superficial pin-track infections, which are caused by a comparatively long time in a frame for many upper-limb conditions. Therefore, the prevalence has been reported to range from 0% to 100%90,91,96,103,104,107,108,117,123,136, with the majority of studies revealing pin-track infection rates of approximately 25% to 50% (Tables I through IV) (see Appendix). In contrast, deep soft-tissue or bone infections are rare events, with a prevalence ranging from 0% to 11%83,114.

Fractures of regenerate bone most commonly occur after device removal, especially if insufficient regenerate bone has been created. In some reports, the prevalence of fractures has been reported to be as high as 50%108. Therefore, despite the potential risk for deep infection, plating of large calluses after long distraction distances has been proposed (Fig. 1-C)93. Delayed bone consolidation and nonunion are further relevant complications, which are regularly reported in the current literature (Figs. 4-A, 4-B, and 4-C). Independent of the lengthened bone, rates have been reported to vary between 0% and 43%69,78,84,93,94,97,98,104,107,117,119,136. Seitz et al., who, to our knowledge, reported the largest clinical series on pediatric upper-limb lengthening, observed an overall nonunion rate of 6% in 141patients134. This type of complication usually requires further surgery with autologous bone-grafting and/or plating for restoration of stability (Fig. 4-C). Premature consolidation is a rather rare event that occurred in only a few series21,71,73,89,96,107,128; Seitz et al. observed this complication in 0.7% of their patients overall134. Device failure, such as pin and wire breakage and loosening, has also frequently been reported and may require subsequent repeat surgery24,68,69,71,83,87,89,94,96,100,116,120,130.

Although reports of vascular complications are uncommon in the literature, neurologic injuries have been observed in many studies12,20,21,74,76-79,89,91,92,95,96,98,101,107,126,129. Injury of the radial nerve specifically is common in association with humeral lengthening procedures12,21,67,74,78,89,91,92. However, most such injuries are temporary and resolve spontaneously. According to the literature, complex regional pain syndrome type II has been reported in 1 patient77.

Further potential problems during lengthening include joint subluxation and dislocation (Fig. 4-B). Ettl et al. and Ip et al. each reported 1 case of progressive radial head dislocation in exostosis-related forearm lengthening98,102. Moreover, dislocation of the thumb metacarpal base during phalangeal lengthening117, subluxation of the proximal interphalangeal joint during severe camptodactyly distraction129, and elbow subluxation in association with a very short below-the-elbow stump135 have been reported. Temporary wrist or finger contractures can occur during forearm lengthening20,76,107,138, whereas elbow contractures occur more often during humeral distraction than during forearm lengthening89,90, with a reported prevalence of up to 27%89. Axial deviation is another potential complication resulting from bone lengthening in the upper extremity68,83,84,89,97,139.

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Overview and Perspectives

Despite recent developments of fixation devices and web-based software tools, bone lengthening remains a highly complex procedure with a need for dedicated patient compliance and endurance. To avoid potential problems during the lengthy, arduous procedures, the importance of the patient’s social, psychological, and economic background and support must not be underestimated. Future therapeutic approaches with forceful axial loading after completed distraction would likely be beneficial to enhance bone regeneration and consolidation. Moreover, early recognition of regenerate bone at risk for mechanical failure is of paramount importance, as are technical variations such as lengthening over an intramedullary nail, which can accelerate consolidation of regenerate bone67,140,141. Practicable and reliable means for assessment of the regenerate bone are important in deciding when to remove the external fixation142. Favorable outcomes can be achieved if indications are carefully assessed. Patient satisfaction may be high overall, even in the event of complications106,134.

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Appendix Cited Here...

Tables showing complications and clinical remarks for pediatric humeral lengthening procedures, pediatric clavicular lengthening procedures, pediatric forearm lengthening procedures, and pediatric hand lengthening procedures are available with the online version of this article as a data supplement at jbjs.org.

Investigation performed at the Department of Pediatric Orthopaedics and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Vienna, Austria

Disclosure: The authors indicated that no external funding was received for any aspect of this work. The Disclosure of Potential Conflicts of Interest forms are provided with the online version of the article.

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