Abstract
Background:
Brachymetatarsia is the presence of an abnormally short metatarsal, and distraction osteogenesis has been used for its treatment. The purpose of the present study was to compare the outcomes of patients who underwent distraction osteogenesis for the treatment of first and/or fourth brachymetatarsia.
Methods:
The data from forty-eight patients (sixty-four feet, seventy-four metatarsals) who underwent distraction osteogenesis for the treatment of brachymetatarsia were reviewed. The indications for surgery included a metatarsal that was at least 10 mm shorter than the adjacent metatarsal and that had an unacceptable cosmetic appearance. The study group comprised thirty-two first brachymetatarsia in nineteen patients (Group A) and forty-two fourth brachymetatarsia in twenty-nine patients (Group B). The average age was twenty years in Group A and eighteen years in Group B. The average duration of follow-up was 58.1 months in Group A and 56.1 in Group B. Metatarsal length, lengthening gain, healing time and index, and the American Orthopaedic Foot and Ankle Society hallux metatarsophalangeal-interphalangeal and lesser metatarsophalangeal-interphalangeal scores were evaluated.
Results:
All patients were satisfied with the final length of the metatarsal and all had achieved bone union at the time of the last follow-up. The mean lengthening gain was 17.2 mm (42.9%) in Group A and 16.3 mm (37.3%) in Group B. The mean healing index was 71.0 days/cm in Group A and 67.3 days/cm in Group B. The mean American Orthopaedic Foot and Ankle Society score was 91.2 points in Group A and 92.8 points in Group B at the last follow-up (twenty-two excellent, nine good, and one fair results in Group A, and twenty-nine excellent, eleven good, and two fair results in Group B). The most common complication was metatarsophalangeal joint stiffness, which occurred in thirteen rays in Group A and in twelve rays in Group B; malalignment of the lengthened metatarsal was observed six times in each group. No significant intergroup differences in the outcomes were found.
Conclusions:
Distraction osteogenesis for first and/or fourth brachymetatarsia provided successful lengthening of a metatarsal with eventual osseous union and was associated with similar outcomes in terms of healing index, function score, and the prevalence of complications between the two groups, although frequent complications were encountered and no improvement in foot function was found.
Level of Evidence:
Therapeutic Level IV. See Instructions to Authors for a complete description of levels of evidence.
Brachymetatarsia is defined as an abnormally short metatarsal and is usually either congenital or caused by premature closure of the physis resulting from posttraumatic, postsurgical, or pathologic conditions1. The deformity can develop following injury, surgical trauma, infection, or tumor or it can be linked to specific disease processes, such as Down syndrome, Apert syndrome, Albright osteodystrophy, sickle-cell anemia, diastrophic dwarfism, and poliomyelitis2,3. The fourth ray is most commonly involved, followed by the first and fifth rays4. The condition is often associated with concurrent involvement of other metatarsals or occurs symmetrically bilaterally5.
The deformity rarely causes pain at adjacent joints or functional impairment, although some patients cannot wear high-heeled shoes. The primary complaint relates to the appearance of the foot1,3,5. Surgery should be planned to restore weight-bearing alignment, which coincides with improved cosmesis.
Several surgical options have been described for the treatment of brachymetatarsia, such as one-stage lengthening with use of an interpositional bone graft, gradual lengthening by distraction osteogenesis with or without shortening of the adjacent metatarsals and phalanges, and shortening of adjacent metatarsals and phalanges1,6-10. One-stage lengthening procedures are preferred for metatarsals that only need to be lengthened up to 15 mm, whereas gradual lengthening allows for a greater lengthening gain and concomitant lengthening of the soft tissues11. Shortening of adjacent metatarsals may be used in conjunction with lengthening procedures to restore the parabolic arc of the metatarsal heads. Management should be tailored to the patient's individual deformity and expectations8,11,12.
Distraction osteogenesis has been reported to have several advantages: it does not require bone-grafting, tendons are stretched more easily, there are fewer neurovascular complications, weight-bearing is achieved sooner, and more length can be achieved1,8-10,13,14. Many reports have described the usefulness, complications, and techniques concerning first and fourth metatarsal lengthening by distraction osteogenesis1,3,5,8,11,15-17. However, few reports have studied the outcomes of patients who have undergone distraction osteogenesis for the treatment of first and fourth brachymetatarsia13. In addition, reports on objective outcomes of and problems associated with brachymetatarsia of each of these rays are limited and available reports include relatively small numbers of patients. Thus, the purpose of the present study was to compare the outcomes of patients who underwent distraction osteogenesis as treatment for first and/or fourth brachymetatarsia.
This study was approved by our institutional review board, and informed consent was obtained from all patients. We retrospectively reviewed forty-eight patients with brachymetatarsia (sixty-four feet, seventy-four metatarsals; sixteen patients underwent bilateral procedures) who received treatment with distraction osteogenesis within the period between January 1998 and October 2006. Metatarsal shortening was congenital in all patients and there were no other congenital foot deformities such as tarsal coalition, polydactyly, or other deficiencies. Patients were divided into two groups: Group A included those with first brachymetatarsia (nineteen patients, thirty-two metatarsals) and Group B included those with fourth brachymetatarsia (twenty-nine patients, forty-two metatarsals). Group A was composed of eighteen women and one man with an average age of twenty years (range, twelve to thirty-four years), and Group B was composed of twenty-eight women and one man with an average age of eighteen years (range, eleven to thirty-four years). Ten feet had combined lesions of the first and fourth metatarsals. The deformity was cosmetically unacceptable to most patients. It rarely caused pain at adjacent joints or functional impairment, although some patients complained of metatarsalgia, a callosity, shoe irritation caused by a cock-up toe deformity, or difficulty in wearing high-heeled shoes. Preoperatively, the first metatarsal length was shorter than the second by an average (and standard deviation) of 17.6 ± 2.5 mm (range, 12.2 to 20.6 mm), and the fourth metatarsal was shorter than the third by an average of 18.4 ± 2.3 mm (range, 15.6 to 23.7 mm). Average follow-up durations were 58.1 months (range, twenty-five to 129 months) in Group A and 56.1 months (range, twenty-five to 129 months) in Group B.
The indications for surgical treatment, which were based on the goals of functional recovery and anatomical restoration, included a metatarsal that was at least 10 mm shorter than the adjacent metatarsal (i.e., the second metatarsal for patients with first brachymetatarsia and the third metatarsal for patients with fourth brachymetatarsia) and full patient understanding of potential complications. All procedures in both groups were performed with use of the same method and by a single surgeon. Follow-up evaluations were performed by independent blinded evaluators not directly involved in the surgical procedures. We compared the clinical and radiographic outcomes of patients with first and/or fourth brachymetatarsia after treatment with distraction osteogenesis, and we additionally compared the results obtained in children with those obtained in adults, and the results of patients who had involvement of more than one metatarsal with those who had only one metatarsal involved in a single foot.
Surgical Technique
Under fluoroscopic guidance, two mini-Schanz half-pins (3 mm in diameter) were inserted into the proximal metaphysis and two more into the distal metaphysis of the metatarsal and directed from medial to lateral in cases of first brachymetatarsia. When the first metatarsal bone was too short to accept all four half-pins, one proximal pin was inserted in the medial cuneiform (three metatarsals). A unilateral external fixator (Dyna-EXTOR; BK Meditech, Seoul, South Korea) was applied to the medial aspect of the metatarsal, as parallel to the plantar surface of the foot as possible in the sagittal plane, and as parallel to the axis of the second metatarsal as possible in the transverse plane to prevent malalignment during metatarsal lengthening (Figs. 1-A and 1-B). A medial longitudinal incision of 1.5 cm was made at the metatarsal shaft, and the periosteum was longitudinally incised and carefully elevated. A transverse corticotomy was performed perpendicular to the plantar surface of the foot, with use of a motorized saw with continuous cooling. In cases of fourth brachymetatarsia, two mini-Schanz half-pins (2 mm in diameter) were inserted into the proximal metaphysis and two more into the distal metaphysis on the dorsolateral aspect of the metatarsal. During pin insertion, care was taken to ensure that the pins would not entrap the extensor digitorum longus tendon to the fifth toe. The proximal two pins were positioned lateral to the fifth extensor digitorum longus tendon, and the distal two pins were positioned medial to it. A unilateral external fixator was applied as parallel to the plantar surface of the foot as possible in the sagittal plane and parallel to the intersection of the axes of the third and fifth metatarsals in the transverse plane. The proper direction of metatarsal lengthening was confirmed by acutely distracting the osteotomy to a distance of approximately 5 mm and assessing the direction of the distraction with use of intraoperative fluoroscopy. The bone surfaces were then restored to apposition until the actual distraction process began.
Postoperatively, 0.25 mm of distraction was applied three times daily after a seven day latency period. We controlled the distraction rate and rhythm according to the radiographic findings and the patient's clinical condition. Radiographs were checked every other week to assess the degree of osteogenesis and the condition of the adjacent joint. Distraction was continued until a satisfactory parabola incorporating all five metatarsal heads had been achieved on radiographs (Figs. 2-A, 2-B, and 2-C).
All patients were permitted full weight-bearing, heel-touch ambulation with use of postoperative shoes, starting on the second postoperative day. The postoperative shoes have hard soles that relieve the weight on the forefoot and pressure on the metatarsal heads and help maintain a normal gait. The external fixator was removed when the callus surrounding the osteotomy had matured.
Radiographic Assessment
Radiographs were obtained with the foot plantigrade on the floor, and the x-ray beam was directed at the center of the forefoot and inclined 15° from vertical at a distance of 100 cm above the film to obtain the anteroposterior radiograph18. To obtain the oblique radiograph of the foot, the patient was positioned supine on the table with the knee flexed and the lateral border of the foot elevated approximately 40° or 50° so that the medial border of the foot was forced against the film cassette19. The central beam was directed vertically to the center of the forefoot. Lateral radiographs were obtained with the beam directed perpendicular to a point just above the base of the fifth metatarsal19.
We evaluated metatarsal lengths, lengthening gains and percentages, the healing time, and a healing index. First and fourth metatarsal lengths were measured on the anteroposterior and oblique radiographs of the foot, respectively, with use of the method described by Tanaka et al.18. Briefly, the axis of both the first and fourth metatarsals was represented by a line that connected the midpoints of the proximal and distal ends of the diaphysis. Metatarsal length was defined as the distance between the points of intersection of the axes of the metatarsals with the distal and proximal ends of the metatarsals. The amount of distraction length was determined when a satisfactory metatarsal head parabola had been achieved11. Cortical continuity of at least three cortices at the distraction site as seen on the anteroposterior and oblique radiographs was considered to represent radiographic consolidation or maturation. Healing time was calculated from the date of surgery to the date of radiographic consolidation. The healing index was calculated by dividing the days of healing time by the centimeters of length gained (days/cm).
Clinical Assessment
Patients were assessed clinically according to the American Orthopaedic Foot and Ankle Society (AOFAS) hallux metatarsophalangeal-interphalangeal and lesser metatarsophalangeal-interphalangeal scoring systems (100 points total)20, in which >85 points indicated a score of "excellent," 71 to 85 points indicated a score of "good," 56 to 70 points indicated a score of "fair," and <56 points indicated a score of "poor." All preoperative and postoperative AOFAS scores were obtained by independent blinded evaluators.
Patients were allocated to four groups with respect to metatarsophalangeal joint motion (dorsiflexion plus plantar flexion): normal (no loss of motion), mild restriction (>75° of motion), moderate restriction (30° to 75° of motion), and marked restriction (<30° of motion)20. The metatarsophalangeal joint motion was measured clinically, by passive dorsiflexion and plantar flexion of the toe, with use of a goniometer placed along the medial border of the foot for the first metatarsophalangeal joint and placed along the lateral border of the foot for the fourth metatarsophalangeal joint. We defined stiffness as moderate or marked restriction of metatarsophalangeal joint motion. All preoperative and postoperative metatarsophalangeal joint motion was measured by independent blinded evaluators.
We also evaluated for postoperative complications, including metatarsophalangeal stiffness, metatarsal malalignment, pin-track infection, pin breakage, callus fracture, delayed union or nonunion, and metatarsophalangeal arthritis.
Statistical Methods
Descriptive statistics (arithmetic means, standard deviation, and ranges) were calculated with use of standard formulas. To test for significant differences between groups, independent t tests were conducted. To test for intra-group differences in AOFAS scores and range of motion of the metatarsophalangeal joints before and after surgery, paired Student t tests were conducted. To test for differences between children and adults, Mann-Whitney U-tests were conducted. For all comparisons, a p value of <0.05 was considered to be significant. All statistical analyses were reviewed independently by a statistician.
Source of Funding
There was no external source of funding for any of the work in this study. No support in form of grants, equipment, or other items has occurred for this project. The authors have received nothing of value. This manuscript does not contain information about medical devices.
All patients were satisfied with the final length of the metatarsal(s), and all had achieved osseous union at the time of the last follow-up. As shown in Table I, the mean lengthening gains were 17.2 mm (range, 13.3 to 20.7 mm) in Group A and 16.3 mm (range, 9.7 to 20.7 mm) in Group B or 42.9% (range, 34.0% to 54.3%) and 37.3% (range, 17.8% to 51.7%), respectively. The mean healing index (healing time divided by centimeters of length gained [days/cm]) was 71.0 days/cm (range, 42.7 to 103 days/cm) in Group A and 67.3 days/cm (range, 53.9 to 83.7 days/cm) in Group B (Figs. 3-A, 3-B, and 3-C). The length gain and the healing index were not significantly different between the two groups (p > 0.05).
The mean AOFAS score was 86.4 points (range, 60 to 90 points) in Group A and 88.2 points (range, 65 to 90 points) in Group B preoperatively; it had improved to 91.2 points (range, 80 to 100 points) and 92.8 points (range, 75 to 100 points), respectively, at the last follow-up. In Group A, twenty-two metatarsals received a score of excellent; nine, good; and one, fair. In Group B, twenty-nine metatarsals received a score of excellent; eleven, good; and two, fair.
Preoperative mean metatarsophalangeal joint motion was 57.5° of dorsiflexion (range, 50° to 70°), 32.3° of plantar flexion (range, 20° to 50°) in Group A and 59.1° of dorsiflexion (range, 50° to 70°) and 31.4° of plantar flexion (range, 20° to 50°) in Group B. At the last follow-up, mean range of motion was decreased to 45.3° of dorsiflexion (range, 15° to 70°), and 27.2° of plantar flexion (range, 10° to 50°) in Group A and 55.2° of dorsiflexion (range, 20° to 70°), and 30.6° of plantar flexion (range, 15° to 50°) in Group B. Postoperative metatarsophalangeal joint dorsiflexion was significantly decreased in both groups compared with preoperatively (p < 0.05), and Group A had significantly more metatarsophalangeal dorsiflexion than Group B had (p < 0.05).
Preoperative mean metatarsophalangeal joint motion (dorsiflexion plus plantar flexion) was 89.8° (range, 80° to 100°) in Group A and 90.5° (range, 80° to 100°) in Group B, and it was decreased to 72.5° (range, 50° to 95°) and 85.8° (range, 50° to 95°) at the last follow-up, respectively. In Group A, metatarsophalangeal joint motion was normal in three rays; there was mild restriction (>75°) in sixteen rays, and there was moderate restriction (30° to 74°) in thirteen rays. In Group B, metatarsophalangeal joint motion was normal in ten rays; there was mild restriction in twenty rays, and moderate restriction in twelve rays. Total metatarsophalangeal joint motion was significantly decreased in both groups postoperatively (p < 0.05), and Group A had significantly less mean metatarsophalangeal motion than Group B had (p < 0.05).
A total of fifty-two complications (twenty-six complications in each group) occurred in thirty-five patients (forty feet). The minor complications usually had no residual consequences but were often annoying or resulted in delayed treatment or delayed rehabilitation. The major complications are the more serious problems that occurred during treatment or after treatment. Minor complications developed in thirty-three metatarsals (seventeen in Group A and sixteen in Group B) and major complications developed in nineteen metatarsals (nine in Group A and ten in Group B) (Table I). The most common complication encountered was metatarsophalangeal joint stiffness, which was seen in thirteen rays in Group A and in twelve in Group B. Malalignment of the lengthened metatarsal, including angular deformity (>5°), cavus deformity (>5°), or hallux valgus deformity (>15°), was present in six metatarsals in both Group A and Group B (three hallux valgus and three cavus deformities in Group A, and four varus angulations and two cavus deformities in Group B). One hallux valgus deformity with a painful bunion was later corrected with use of a distal chevron osteotomy. Four cavus deformities (two in Group A and two in Group B) underwent a dorsal closing wedge osteotomy at the metatarsal base, with correction achieved in all four. The other seven patients accepted the deformity functionally and cosmetically. A fracture through the callus occurred in three metatarsals (two patients) in Group A and in three metatarsals (three patients) in Group B. All six were treated with open reduction and internal fixation, and osseous consolidation was achieved. Three pin breakages (two in Group A and one in Group B) occurred during the lengthening period and were treated by placement of a new pin, and five pin-track infections (two in Group A and three in Group B) resolved uneventfully by administering oral antibiotics and performing dressing changes. Metatarsophalangeal arthritis was observed in one fourth toe at the time of the first follow-up.
We compared the results in children with those in adults (see Appendix). The children had better results compared with the adults for both first and fourth brachymetatarsia in terms of a lower healing index, a higher AOFAS score, and a lower incidence of complications. However, a significant difference was observed only in the healing index between the two groups.
When comparing the involvement of more than one metatarsal (a total of ten feet from groups A and B) with involvement of just one bone (twenty-two feet in Group A and thirty-two feet in Group B) in a single foot, those with multiple involvement presented at a younger age (mean, seventeen years) than those with involvement of just one bone (mean, twenty-five years in patients with first-ray involvement and twenty-two years in patients with fourth-ray involvement) and, not surprisingly, had a significantly lower healing index (p < 0.05). Otherwise, those with multiple-ray involvement had similar range of motion, AOFAS scores, and complication rates than did the patients with involvement of just one bone, even though the patients with multiple-ray involvement underwent distraction osteogenesis for first and fourth brachymetatarsia simultaneously.
Whether a patient's sex contributes to his or her willingness to undergo surgical correction of brachymetatarsia is not known. In comparison with men, women may be more concerned about the appearance of their feet, particularly because they tend to wear open-toed shoes more often than men do, so it is possible that women may be more willing to undergo the procedure13. The AOFAS position statement on cosmetic foot surgery states that when deciding to proceed with surgery, the patient and the surgeon must consider all of the risks and benefits of a procedure20. When the potential benefits outweigh the potential risks, then surgical intervention may be warranted. Nonsurgical treatment of this condition consists of padding and the use of accommodative devices in the shoe, but these will not relieve the underlying deformity or the cosmetic problem. The main goal of surgical correction of brachymetatarsia is the restoration of a functional metatarsal parabola21 through the correction of deformity.
Several investigators have reported varying mean healing indices for distraction osteogenesis in the treatment of brachymetatarsia, ranging from 60.0 to 98.0 days/cm for patients with first brachymetatarsia and 43.4 to 82.0 days/cm for patients with fourth brachymetatarsia (see Appendix)1,3,5,8,10,11,13,15,17. Choi et al.8 reported a higher mean healing index in the first than that in the fourth metatarsal. Oh et al.1,16 and Kim et al.11 reported a similar mean healing index in both. On the other hand, Shim and Park13 and Wada et al.3 reported a lower mean healing index in the first than in the fourth. In the present study, there was a higher healing index in the first (71.0) than in the fourth (67.3), but this difference was not statistically significant. This result may be explained by a longer fixation period and healing time, the need for greater lengthening gain, and higher tension in the surrounding soft-tissue structures in patients with first brachymetatarsia.
In terms of clinical outcomes, most of the previous studies reported only subjective outcomes and a few studies reported objective clinical results based on the AOFAS score1,5,11,16. Based on the AOFAS score, Kim et al.11 and Oh et al.1,16 reported, respectively, 80% and 100% good or excellent results for first brachymetatarsia, and 80% and 89% good or excellent results for fourth brachymetatarsia. We had good or excellent clinical outcomes in 97% of patients with first brachymetatarsia and in 95% of patients with fourth brachymetatarsia, demonstrating that distraction osteogenesis is an excellent treatment option for both first and fourth brachymetatarsia.
The most common complications were metatarsophalangeal joint stiffness and malalignment of lengthened metatarsals in both groups, both in our study and according to all previous reports (Appendix). Metatarsophalangeal joint stiffness is considered to be due to the tension created, in the tendons and the adjacent soft knee connections such as the transverse metatarsal ligament and the flexor sheath, with lengthening. In addition, some studies have demonstrated larger decreases in motion in patients who have achieved a length gain of more than 40%, and these studies have emphasized that the amount of lengthening should not exceed 40%10,11,13,15,22.
In the present study, metatarsophalangeal stiffness was the most common complication and moderate stiffness was observed in thirteen of thirty-two rays in Group A and twelve of forty-two rays in Group B. Shim and Park13, who used a technique that was similar to ours, reported that seven of thirty-nine rays developed joint stiffness or metatarsophalangeal subluxation that required a plantar capsulotomy. On the other hand, we had no cases of severe stiffness in our study. In our experience, metatarsophalangeal joint stiffness results from complex factors, such as distraction length, distraction rate and rhythm, fixation time, and the rehabilitation method. Therefore, to prevent metatarsophalangeal joint stiffness, we made an attempt to avoid excessive lengthening, a too-rapid distraction rate and rhythm, or a long external fixation time. Postoperatively, we also focused on early postoperative active and passive metatarsophalangeal stretching exercises to prevent joint stiffness.
Malalignment after distraction osteogenesis, such as cavus or hallux valgus deformity or varus angulation, is also an important complication. In the present study, the most common malalignments observed were cavus deformity and hallux valgus in Group A and varus angulation and cavus deformity in Group B. To avoid malalignment, we agree with other authors who recommend that, during the surgical correction of first brachymetatarsia, the external fixator axis must be as parallel as possible to the plantar surface along the medial aspect of the first metatarsal and parallel to the anatomical axis of the second metatarsal in the transverse plane. In fourth brachymetatarsia, the external fixator axis must be as parallel to the plantar surface as possible and parallel to the intersection of the axes of the third and fifth metatarsals in the transverse plane.
Few reports have compared the outcomes of children and adults with brachymetatarsia13,16. Oh et al.16 concluded that children had better results than adults because of a lower prevalence of major complications and higher AOFAS scores. Shim and Park13 noted that the healing index increased with age because the younger patients have a higher healing capacity. Similarly, in the present study, children had better results compared with adults: a lower healing index, a higher AOFAS score, and a lower complication rate. However, a significant difference was only observed in the healing index between the two age groups.
In conclusion, distraction osteogenesis for first and fourth brachymetatarsia can provide successful lengthening of the metatarsal with eventual osseous union. Both rays have similar overall outcomes based on the healing index, AOFAS scores, and the prevalence of complications. However, following the procedure, complications are frequent and improvement in foot function should not be expected.
Tables showing a comparison between adults and children and listing the results of other reports in the literature are available with the electronic version of this article on our web site at jbjs.org (go to the article citation and click on "Supporting Data").
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