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OBJECTIVE: Surgical technique in distraction osteogenesis for the treatment of brachymetatarsia can influence the final prognosis. However, there are currently no standardized guidelines for surgical procedures and complication management. The aim of this study is to investigate the effect of bone lengthening with external fixation by minimally invasive osteotomy based on Ilizarov technique in the treatment of congenital brachymetatarsia. METHODS: A retrospective study was conducted on patients with congenital brachymetatarsia treated by metatarsal lengthening, from June 2017 to December 2020. There were 11 patients with 17 shorted fourth metatarsals, including 10 females and 1 male, with age of 24.6 ± 4.5 years (16-31 years). Six patients were bilaterally involved. Orthofix external fixator mini track was installed through dorsal approach and the fourth MTP joints were temporarily fixed by Kirschner wire. Bone lengthening was performed after a minimally invasive osteotomy at the proximal metatarsals. American Orthopedic Foot and Ankle Society (AOFAS) lesser metatarsophalangeal-interphalangeal (MTP-IP) scores, metatarsal length, complications were recorded. Statistical comparison was performed using the paired t-student test for pre- and postoperative AOFAS MTP-IP scores. RESULTS: Patients were followed up for 55 ± 10.8 months. The mean length of the fourth metatarsal bone was 49.9 ± 2.9 mm preoperatively. The mean metatarsal shortage was 18.8 ± 3.1 mm. The mean lengthening achieved was 19.8 ± 3.3 mm, with a lengthening ratio of 39.7% ± 6.6%. The lengthened callus ossified completely at 3-4 months after operation. All patients were satisfied with the results of lengthening. The AOFAS scores were improved significantly from 83.7 ± 4.2 preoperatively to 93.2 ± 2.7 postoperatively (t = -10.27, p < 0.001). One patient with traumatic metatarsophalangeal joint subluxation was treated by joint reduction and Kirschner wire fixation. One patient had metatarsophalangeal joint release and Kirschner wire fixation due to flexion contracture. Pin tract infections were controlled by wound care and antibiotics in 6 patients. All patients had no nonunion, necrosis of toes, and sensory disturbance of toes. CONCLUSION: Metatarsal lengthening by minimally invasive osteotomy with external fixator had satisfactory results in the treatment of congenital brachymetatarsia.

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Brachydactyly is a genetic condition leading to shortened or absent digits in hands or feet. It can occur independently or as part of syndromes. This case focuses on Brachydactyly type B, the rarest form. An 8-month-old from the Philippines was referred due to a missing third toe. Examination revealed a hypoplastic left third toe. X-rays confirmed the finding. Treatment options were discussed, including conservative therapy and follow up. Diagnosis involved history, examination, and imaging. Prenatal diagnosis is limited for isolated cases but useful for syndromic forms if a family mutation is known. Prognosis varies depending on the severity and associated syndromes. Currently there is no definitive treatment; management involves genetic counseling and therapy. Due to limited cases, Type B is underreported, highlighting the need for more research into its genetics.

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PURPOSE: The primary aim is to assess the efficacy of the surgical callus distraction technique of the metatarsus in paediatric patients. Secondary objectives are to assess complications and treatment duration. We have also described the details of our surgical technique. MATERIALS AND METHODS: A case series review of paediatric patients who had metatarsal lengthening at our unit between 2014 and 2022. Patient demographics, duration of time in frame, complications and metatarsal length achieved were recorded. The AOFAS Midfoot and the MOXFQ were taken pre-operatively and at final follow-up. RESULTS: Sixteen metatarsals in 8 patients (14 feet) underwent lengthening between 2014 and 2022 using the MiniRail OrthoFix 100 (Orthofix Medical Inc, Lewisville, TX, USA). The mean age was 13.3 (12-17) years. The average duration between surgery and implant removal was 5.2 months. According to Paley's classification, there was one obstacle encountered in a patient who required a revision of their osteotomy and one problem in another patient who had an infected metatarsophalangeal joint stabilising k-wire treated with oral antibiotics. The Mean AOFAS Midfoot score improved from 53.10 to 86.40 (p < 0.0001) and the Mean MOXFQ improved from 32.5000 to 12.1250 (p < 0.05); these were statistically significant. CONCLUSION: Gradual metatarsal lengthening using the MiniRail external fixator is a safe and effective method to treat brachymetatarsia in paediatric patients. This preliminary report describes and supports metatarsal lengthening in appropriate patients. Holistic care in terms of a pre-operative assessment, psychological support and preparation for the extended rehabilitation period are vital.

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Purpose: Brachymetatarsia is marked by the shortened length of one or more metatarsal bones. Several operative options have been suggested without demonstrating the superiority of one treatment over another. This study aims to assess the main available treatment, bone lengthening achieved, clinical outcomes, and complications pertaining to congenital brachymetatarsia interventions. Methods: A literature search of PubMed, Embase, Scopus, and Cochrane databases was performed according to PRISMA guidelines. Result: After the screening process, 13 articles were included in the review. Brachymetatarsia mainly occurs in females at a ratio of 14.8:1, and bilateral involvement in 25.8 % of cases. The fourth metatarsal is most affected, followed by the first metatarsal. The main surgical procedures are one-stage approach which results in limited length gain but fewer complications, and gradual lengthening which achieves greater length but has a higher complication rate, including metatarsophalangeal or interphalangeal subluxation, fractures, and infections. The first metatarsal reported better values concerning lengthening rate and healing index compared to fourth metatarsal, while no differences were observed in distraction rates or clinical outcomes. More complications, such as stiffness, fractures, and cavus foot were associated with first metatarsal lengthening. Conclusions: It is not possible to identify a gold standard technique for addressing this condition. The two predominant surgical techniques bear advantages and drawbacks. A judicious case-by-case assessment is mandatory to determine the optimal surgical approach. Exploring the combination of techniques could be promise in mitigating the limitations associated with the main two methods and achieving better forefoot balance. Level of evidence: Level II, systematic review.

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BACKGROUND: This study examined the functional and clinical outcomes of subacute two stage metatarsal lengthening with gradual distraction for brachymetatarsia. This technique was developed to overcome the disadvantages of one-stage metatarsal lengthening and gradual distraction. METHODS: Four feet of three patients with congenital brachymetatarsia underwent subacute two stage metatarsal lengthening with gradual distraction. Pain, function, and alignment were assessed preoperatively and at follow-ups using the American Orthopaedic Foot and Ankle Society (AOFAS) lesser metatarsophalangeal-interphalangeal scale, and any complications were recorded. RESULTS: The patients were followed up for a mean of 18.1 ± 6.9 (range, 12.6-28.1) months. The mean metatarsal length gain was 15.2 ± 3.2 (range, 12.1-18.5) mm, and the corresponding percent increase was 32.5 % ± 7.0 % (range, 25.7-41.1 %). The mean AOFAS score (0-100) was 97.5 ± 5.0 at the final follow-up. The external fixator index was 10.2 ± 1.5 (range, 8.1-11.6) days/cm. None of the patients experienced metatarsophalangeal stiffness, subluxation or dislocation of the metatarsophalangeal joint, loss of correction, pin tract infection, delayed union, nonunion, or angular deformities. CONCLUSION: Subacute two stage metatarsal lengthening with gradual distraction is a reliable alternative treatment for brachymetatarsia.

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Braquimetatarsia es el acortamiento anormal de uno o más metatarsianos. Tiene una incidencia de 0.02%-0.05% predominando en mujeres de 25 a 1. En un 72% puede ser bilateral. La etiología no es clara pero se plantea el cierre prematuro de la fisis dando un retardo en el crecimiento. La mayoría de las veces presenta síntomas causados por alteración en la mecánica de la parábola metatarsal como dolor e hiperqueratosis. Puede existir metatarsalgia y agregar en la evolución desviaciones digitales adicionales. El principal motivo de consulta es estético. El objetivo del reporte es mostrar dos técnicas quirúrgicas y los resultados clínicos correspondientes usando el score AOFAS, además de los resultados imágenológicos con hasta 5 años de seguimiento. Se describen dos pacientes y se analizan dos técnicas quirúrgicas diferentes para alargar los metatarsianos. Se trata de alargamientos agudos con interposición de injerto intercalar, en un caso mediante el procedimiento de Sandro Gianini con injerto de cresta ilíaca y en el otro con injerto de metatarsiano adyacente con modificación de la longitud del segundo y tercer metatarsiano restituyendo en ambos casos la parábola metatarsal. Obtuvimos buenos resultados al igual que series numerosas pudiendo recomendarse en casos similares.

Brachymetarsal is the abnormal shortening of one or more metatarsals. It has an incidence of 0.02%-0.05%, predominantly in women from 25 to 1. In 72% it can be bilateral. The etiology is not clear, but premature closure of the physis is suggested, giving growth retardation. Most of the time it presents symptoms caused by an alteration in the mechanics of the metatarsal parabola, such as pain and hyperkeratosis. There may be metatarsalgia and add additional digital deviations in the evolution. The main reason for consultation is aesthetic. The objective of the work is to show the surgical technique and the clinical results using the AOFAS score, in addition to the imaging results with up to 5 years of follow-up. Two patients are described and two different surgical techniques to lengthen the metatarsals are analyzed. These are acute lengthenings with intercalary graft interposition, in one case using the S.Gianini procedure with an iliac crest graft and in the other with an adjacent metatarsal graft with modification of the length of the second and third metatarsals, restoring in both cases the metatarsal parabola. We obtained good results. results as well as numerous series and can be recommended in similar cases.

Braquimetatarsal é o encurtamento anormal de um ou mais metatarsos. Tem incidência de 0,02%-0,05%, predominantemente em mulheres de 25 a 1 ano. Em 72% pode ser bilateral. A etiologia não é clara, mas sugere-se o fechamento prematuro da fise, causando retardo de crescimento. Na maioria das vezes apresenta sintomas decorrentes de uma alteração na mecânica da parábola metatarsal, como dor e hiperqueratose. Pode haver metatarsalgia e adicionar desvios digitais adicionais na evolução. O principal motivo da consulta é a estética. O objetivo do trabalho é mostrar a técnica cirúrgica e os resultados clínicos utilizando o escore AOFAS, além dos resultados de imagem com até 5 anos de seguimento. Materiais e métodos: São incluídos dois pacientes e analisadas duas técnicas cirúrgicas diferentes para alongar os metatarsos. São alongamentos agudos com interposição de enxerto intercalar, em um caso utilizando a técnica de S. Gianini com enxerto de crista ilíaca e no outro com enxerto de metatarso adjacente com modificação do comprimento do segundo e terceiro metatarsos, restaurando em ambos os casos o metatarso parábola. Obtivemos bons resultados, bem como inúmeras séries, podendo ser recomendados em casos semelhantes.

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BACKGROUND: Symptomatic patients with severe brachymetatarsia are commonly treated with callus distraction using external mini-fixator (EF) or internal device (ID). This study points out advantages and limitations of both methods comparing clinical and radiographical parameters. METHODS: Retrospective analysis of 21 metatarsal bones in twelve patients. Twelve metatarsals were treated with ID (Genos Mini), nine with EF (MiniRail). RESULTS: Mean lengthening distance was 17.3 mm using EF and 11.7 mm using ID (p = 0.016). Adverse results were observed in 89% of metatarsals treated with EF and in 33% treated with ID (p = 0.011). Postoperative surgical intervention was required in 33% using EF compared to 0% using ID (p = 0.031). Mean total German Foot Function Index (FFI-T) improved from 49 to 33 using EF and from 47 to 22 using ID (p < 0.001). CONCLUSION: CD is a reliable surgical treatment for BMT. Surgeons should be aware of implant-related advantages and complications when counseling patients about treatment options.

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Aim: To describe the surgical technique for gradual lengthening of the metatarsal using the Orthofix MiniRail System (Orthofix Medical Inc., Lewisville, TX, USA). Background: Brachymetatarsia refers to the shortening of the metatarsal bone. When indicated, metatarsal lengthening is performed to treat this deformity. Technique: A dorsal approach to the short metatarsal is performed, protecting the neurovascular structures and the extensor tendons. The most proximal wire or screw is inserted first, followed by the most distal wire or screw. The distal wire or screw should not be inserted in the metatarsal head. The middle wires or screws are inserted thereafter. All wires or screws should be placed perpendicular to the anatomic axis of the bone. Once the MiniRail lengthener is assembled, a 1.6 mm K-wire is inserted from the tip of the toe into the metatarsal head, blocking the motion of the metatarsophalangeal joint and avoiding joint subluxation during lengthening. The osteotomy is then performed perpendicular to the metatarsal shaft, in between the middle two wires and screws. Conclusion: The surgical technique for gradual lengthening of the metatarsal using the Orthofix MiniRail System was described in detail with accompanying step-by-step intraoperative clinical and fluoroscopic images. Clinical significance: This surgical technique of metatarsal lengthening is simple and effective. An understanding of the surgical technique of gradual lengthening of the metatarsal bone is important when treating shorting deformities of more than 1 cm. How to cite this article: Marwan Y, Abu Dalu K, Bernstein M, et al. Metatarsal Gradual Lengthening for Brachymetatarsia Using a Mini-rail External Fixator System. Strategies Trauma Limb Reconstr 2022;17(3):184-188.

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Level of Evidence: Level V.

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BACKGROUND: Mutations in NARS2 (MIM: 612803) are associated with combined oxidative phosphorylation deficiency 24 (COXPD24; MIM: 616239) that is a rare mitochondrial and a multisystem autosomal recessive disorder. AIMS: We aimed to detect the underlying genetic factors in two siblings with progressive ataxia, epilepsy, and severe-to-profound hearing impairment. METHODS: After doing medical assessments and pertinent tests (i.e., auditory brainstem responses, pure tone otoacoustic emission test, cardiac examinations, computed tomography, and electroencephalogram), because of the clinical and probable genetic heterogeneity, whole-exome sequencing was performed, and co-segregation analysis was confirmed by Sanger sequencing. Biological impacts of the novel variant were evaluated using sequence-to-function bioinformatics tools. RESULTS: A novel homozygous missense variant, NM_024678.6:c.545 T > A; p.(Ile182Lys), in exon 5 of NARS2 was identified in both patients and verified by Sanger sequencing. In silico analyses introduced this variant as pathogenic. Mitral valve prolapses with mild regurgitation, brachymetatarsia, severe hallux valgus, and clubbed fingers were reported as novel manifestations in association with NARS2 gene. By doing a literature review, we also underscored the high heterogeneity of disease phenotype. CONCLUSIONS: Herein, we report some novel phenotype and genotype features of two female patients in an Iranian consanguineous family with COXPD24, caused by a variant in NARS2-NM_024678.6: c.545 T > A; p.(Ile182Lys). Moreover, our data expanded the phenotype and genotype spectrum of NARS2-related disorder and confirmed an unpredictable nature of genotype-phenotype correlation in COXPD24.

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Brachymetatarsia (BM), or hypoplastic metatarsal, is an abnormal shortening of one or more metatarsal bones with a female-to-male ratio of 10.53:1. Different causes are described in the literature, such as congenital, acquired, or iatrogenic, associated with different conditions and syndromes. Its presence may develop deformity and pain; however, often feet are pain free and the major worries of patients are cosmetics. Non-operative treatments aim to improve the comfort of metatarsal heads and the possible dorsal conflict through comfortable shoes or the use of specific orthotics. The surgical treatment is anything but straightforward, with "one-stage" or "two stage" techniques, the latter better called "by gradual distraction". One-stage procedures are more rapid techniques but have limited ability to restore the desired length due to neurovascular compromise caused by acute lengthening. Insufficient correction is also possible. On the contrary, by gradual distraction procedures allow gradual distraction lengthening of more than 1.5 cm, but require the use of an external fixator, with a higher risk of complications in more than about 50% of surgeries. The adjacent metatarsal shortening should be considered in combination with other techniques, to diminish the excessive lengthening. In each case, surgeries should be always decided on each patient's concerns, deformities, and clinical needs.

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This article describes the 3 main surgical options for correction of congenital brachymetatarsia in childhood. The one-stage lengthening by lengthening osteotomy and lengthening with graft interposition are suitable for defects less than 10 mm. For the greater defects from 10 mm to more than 20 mm, gradual lengthening by callus distraction with an external or internal fixator is appropriate. Over the last years, callus distraction with an internal minifixator became commonly established because of the significantly improved aftercare with early full weight-bearing and high postoperative comfort for the child. All 3 surgical procedures are presented with comprehensive image material.

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Gradual lengthening by distraction osteogenesis is widely used for congenital brachymetatarsia. The usual presenting complaint is the patient's cosmetic appearance. Osteotomy is an integral element. A nonincisional surgical approach for osteotomy can reduce dorsal longitudinal scarring and help preserve the periosteal blood supply to the bone during surgical dissection. Between June 2003 and January 2019, we performed gradual lengthening by callus distraction with nonincisional osteotomy for congenital brachymetatarsia on 13 digits in 5 patients. All 5 patients were female, and their average age was 18 years old. The lengthened bones involved 3 first metatarsals, 2 third metatarsal, and 8 fourth metatarsal bones. The mean gain in length was 15.2 mm (10-21 mm). The mean duration of distraction was 36.2 days (30-48 days). The mean duration of consolidation was 62.8 days (28-103 days). The lengthened segment consolidated in all cases. One patient had early consolidation and did not wish to undergo further surgery. There were no cases of trouble due to drilling for osteotomy. All patients had a normal gait and were satisfied with the cosmetic results.

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OBJECTIVE: Here we present the first known examples of brachydactyly from an Andean archaeological context by describing the affected bones, presenting a differential diagnosis, and discussing the cultural implications of there being shortened metapodials in multiple tombs. MATERIALS: 3232 well-preserved tubular bones representing an MNI of 250 human adults. METHODS: Each bone was visually inspected. Measurements were taken with an osteometric board, sliding calipers, and a flexible tape measure. RESULTS: Of 1210 metapodials excavated from eight burial contexts, ten were atypically short. CONCLUSIONS: The ten shortened elements represent an MNI of three individuals with brachydactyly from two tombs. The presence of at least two individuals with brachydactyly in one tomb adds support to a previous suggestion that tombs were used for familial interment. It is plausible that the third individual from a different tomb was related to the other two, and the different burial contexts may reflect postmarital practices. SIGNIFICANCE: These cases offer insight into tomb use and underscore the importance of identifying rare developmental anomalies in the archaeological record as their presence may indicate genetic relationships within or among archaeological cemeteries. LIMITATIONS: With commingled contexts and incomplete recovery of skeletal remains, individualizing the brachydactylous elements was not possible. There is also a lack of comparative data from other Andean sites. SUGGESTIONS FOR FURTHER RESEARCH: Identify more Andean cases of brachydactyly to learn if the relatively frequent involvement of the first digit is more common among Andean skeletal samples than North American, or if it is unique to Marcajirca.

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Hallux valgus deformity is one of the most common foot and ankle diseases, while brachymetatarsia is a rare foot anomaly with pathological shortening of a metatarsal bone. We present a case of hallux valgus deformity possibly due to second brachymetatarsia. As the hallux valgus was associated with dorsal dislocation of the second toe that made it difficult to evaluate the length of the second toe, the patient was unaware of the second metatarsal shortening until the lengths of the toes compared by manual reposition of the second MTP joint. In this case, proximal osteotomy of the first metatarsal on the hallux valgus and single-stage bone lengthening of the second metatarsal with iliac bone grafting on the second brachymetatarsia were performed. One year after the operation, the callosity of the third toe resolved and the clinical scores were improved. In the case of a hallux valgus deformity with second dorsal dislocation of the toes, surgeons should consider that there are rare cases with second metatarsal shortening. When hallux valgus associated with second brachymetatarsia is diagnosed, second metatarsal lengthening should be considered in addition to hallux valgus surgery.

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Brachymetacarpia and brachymetatarsia are rare congenital presentations denoted by shortening of metacarpals and metatarsals respectively, in which the deformity usually presents around childhood/early adolescence.The aetiology is usually congenital besides several other syndromic or endocrinologic associations.Cosmetic issues such as absence of a normal-looking knuckle while making a fist or disruption of finger-tip curvature besides functional issues are the main indications for surgical intervention in brachymetacarpia.In the foot, apart from cosmetic issues, pain due to transfer metatarsalgia as well as callosities along with toe deformities which lead to difficulty of using footwear are the main indications for intervention.Lengthening of the affected bone, either acute with grafting or gradual, is the mainstay of treatment. Gradual lengthening can be either single-stage as in callotasis, or two-stage where the primary procedure is followed by bone grafting after the length has been achieved.Adolescence, specifically between 12 and 15 years, is the preferred period for surgical intervention in these cases. Cite this article: EFORT Open Rev 2021;6:15-23. DOI: 10.1302/2058-5241.6.200087.

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BACKGROUND: Distraction osteogenesis is frequently used for brachymetatarsia. METHODS: Brachymetatarsia of the fourth ray was treated with the Ilizarov method in 10 females. Distraction rate was 1.00-0.75mm a day. Radiography and ultrasonography were used to study the regeneration course throughout the distraction and fixation phases. RESULTS: Mean lengthening achieved was 2.28cm in the average distraction period of 30.2 days and average fixation phase of 50.2 days. Ultrasonography detected the regeneration signs and zonal structure of the regenerate earlier than radiography. It confirmed slow regeneration in two patients and their distraction rate was corrected. One patient had premature consolidation on distraction day 10 and needed re-osteotomy. CONCLUSIONS: Combination of radiography and ultrasonography to study bone regeneration in brachymetatarsisa provides a better monitoring of distraction callus progression. Ultrasonography is of great value to detect regeneration problems during early stages of lengthening.

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BACKGROUND: Brachymetatarsia is a rare abnormality of the foot which occurs most frequently in the first and fourth metatarsals. The aim of this study was to evaluate the efficacy of gradual metatarsal lengthening by external fixator for treatment of brachymetatarsia of the fourth ray. The hypothesis was that with external fixation it would be possible to achieve the desired length of the metatarsal with a low rate of complications. Secondarily, in cases requiring a greater amount of correction, it was hypothesized that an opportune rate of bone consolidation would be achievable using a traditional oscillating saw without predrilling or use of a cold osteotome. METHODS: Between 2013 and 2016, 12 eligible patients and 13 feet underwent gradual metatarsal lengthening by an external fixator (MiniRail System M103, Orthofix) due to brachymetatarsia of the fourth ray. Mean age at surgery was 24.5±5.3 years (range 19-36), with mean follow-up of 22.3±8.3 months. Clinical evaluation was performed with the AOFAS lesser metatarsophalangeal-interphalangeal (MTP-IP) score. Radiographic assessment was performed on follow-up using non-weightbearing dorsoplantar foot radiographs. RESULTS: The mean AOFAS lesser MTP-IP score improved from a preoperative score of 76.6±7.1 points (range 62-85 points) to a postoperative score of 90.3±3.0 points (range 86-95 points). The average amount of lengthening was 16.8±3.9mm (range 8-22mm). Mean shortening, final lengthening, Healing Index, period of treatment, and complications are also reported. The operative technique is described. CONCLUSIONS: Gradual metatarsal lengthening with external fixator is an effective treatment for brachymetatarsia and can restore forefoot anatomy with good clinical outcomes, a low rate of morbidity and complications in selected cases. Particular attention should be given when treating patients with shortening >20mm.

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Brachymetatarsia is a rare deformity with controversial clinical presentation. Multiple acute and gradual lengthening surgical techniques have been described for correction of this type of foot deformity. All techniques try to create a better appearance, facilitate shoeing, or solve possible transfer metatarsalgia. Either acute lengthening (1-stage procedure) or gradual lengthening (2 stages) is selected based on the patient's deformities, concerns, and clinical needs.

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INTRODUCTION: Brachymetatarsia can be treated by lengthening of the affected metatarsals or shortening of the normal neighboring metatarsals. Combination of the two methods may be more effective rather than using one skill due to less complications. CASE REPORT: A 56-year-old female patient with the bilateral first, fourth-ray brachymetatarsia underwent Weil osteotomy of the second, third, fifth metatarsals, and callotasis of the fourth metatarsal. Follow-up examination had been carried out since the surgery was done about 10 years ago. CONCLUSIONS: In the decade after observation, the patient was very satisfied with the shape and function of the feet, and no complications occurred.