Received: 29 November 2022 Revised: 13 March 2023 Accepted: 5 May 2023 DOI: 10.1002/micr.31058 CASE REPORT Treatment of Freiberg's disease using a reverse pedicled metatarsal bone flap: Case report Francisco Soldado MD, PhD 1,2,3,4 | Felipe Moreira Borim MD 1,2,4 | 1,2 Jorge Knörr MD, PhD 1 Surgery and Morphological Sciences, noma de Barcelona (UAB), Universitat Auto Abstract Barcelona, Spain Despite no surgical procedures receiving unanimous support for treating Freiberg's 2 Orthopediatrics Surgery Department, Hospital Universitari Vall d'Hebron, Barcelona, disease, several surgical treatment options have been described. For the past few Spain years, bone flaps have been shown in children to present promising regenerative 3 International Pediatric Hand Surgery and properties. We report a novel technique using a reverse pedicled metatarsal bone Microsurgery Institute, Barcelona University Children's Hospital HM Nens, HM Hospitales, flap from the first metatarsal to treat one case of Freiberg's disease in a 13-year-old Barcelona, Spain female. The patient presented 100% involvement of the second metatarsal head, 4 Bioengineering, Cell Therapy and Surgery in with a 6  2 mm defect, unresponsive to 16 months of conservative measures. Congenital Malformations – VHIR, Hospital Universitari Vall d'Hebron, Barcelona, Spain A 7 mm  3 mm pedicled metatarsal bone flap (PMBF) was obtained from the lateral proximal first metatarsal metaphysis, mobilized and pedicled distally. It was inserted Correspondence Felipe Moreira Borim, Orthopedic Surgery at the dorsum of the distal metaphysis of the second metacarpal towards the center Department, Hospital Universitari Vall d'Hebron, Passeig Vall d'Hebron 119-129, of the metatarsal head, reaching the subchondral bone. Initial favorable clinical and Barcelona 08035, Spain. radiological results were maintained for over 36 months during the last follow-up. Email: dr.moreiraborim@gmail.com Based on the powerful vasculogenic and osteogenic properties of bone flaps, this novel technique could effectively induce bone revascularization and prevent further collapse of the metatarsal's head. 1 | I N T RO DU CT I O N for metatarsal head revascularization using a retrograde bone flap har- vested from the first metatarsal bone based on the first dorsal meta- Freiberg's disease (Freiberg, 1914) is the fourth most common form of tarsal vessels. We successfully used this technique in one case primary osteochondrosis, affecting the metatarsal head (MTH), most refractory to conservative measures. commonly the second, with significant predilection to the adolescent athletic female population (Carmont et al., 2009; Gillespie, 2010). Sev- eral surgical treatment options have been described to treat Freiberg's 2 | CASE REPORT disease, with no evidence of any of them being superior to the others (Schade, 2015; Seybold & Zide, 2018). Recently, vascularized bone A 13-year-old female semi-professional contemporary dancer pre- grafts and flaps have gained particular attention in reconstructive sur- sented to our clinic with right forefoot pain. On physical examination, gery because of how effective they promote healing when used to passive metatarsophalangeal joint extension and flexion were treat complex bone nonunion, avascular necrosis and other challeng- 20 degrees each. Radiographs showed a central subchondral cyst ing scenarios in pediatric patients, owing to their high angiogenic and extending 2/3 of the transversal width of the MTH and a mild flatten- osteogenic potentials (Fontecha et al., 2016; Payatakes & ing of the coronal curvature (Figure 1a). Oblique radiograph view Sotereanos, 2009; Urbaniak et al., 1995). We report a novel technique showed MTH flattening (Figure 1b). A Smillie stage III Freiberg's This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. © 2023 The Authors. Microsurgery published by Wiley Periodicals LLC. Microsurgery. 2023;1–5. wileyonlinelibrary.com/journal/micr 1 10982752, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/micr.31058 by Readcube (Labtiva Inc.), Wiley Online Library on [18/12/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 2 SOLDADO ET AL. F I G U R E 1 (a) A-P X-ray showing a central subchondral cyst at the metatarsal head (MTH) and a mild flattening of the coronal curvature. (b) Oblique X-ray showing MTH flattening. (c, d) T2-weighted-magnetic resonance shows osteonecrosis with abnormal intensity involving 100% of the axial area of the MTH. F I G U R E 2 (a) Red arrow: First dorsal metatarsal vessels are identified running adjacent to the first metatarsal and superficial to the first dorsal interosseous muscle. (b) Blue arrow: a 7 mm  3 mm pedicled metatarsal bone flap was obtained from the lateral proximal first metatarsal metaphysis with the aid of a chisel. (c) X-ray of the bone tunnel being made with a 3 mm cannulated drill entering at the dorsum of the distal metaphysis of the second metacarpal. disease was diagnosed. MRI showed osteonecrosis with abnormal The FDMV was ligated proximally at its origin from the dorsalis intensity involving 100% of the axial area of the metatarsal head pedis vessels, and the flap was mobilized and pedicled distally. Flap (Figure 1c,d). Unresponsive to 16 months of conservative measures, bleeding was verified after tourniquet release. A bone tunnel was per- including dancing cease, plantar foot orthosis and physical therapy, so formed with a 3 mm cannulated drill entering the dorsum of the distal surgical treatment was indicated. metaphysis of the second metacarpal towards the center of the meta- Under general anesthesia and tourniquet without exsanguination, tarsal head and reaching the subchondral bone (Figure 2). The bone a dorsal zig-zag incision over the first intermetacarpal space was per- graft was slightly reduced with the aid of a rongeur and introduced formed. The first dorsal metatarsal vessels (FDMV), of small caliber, into the bone tunnel (Figure 2c). The flap can readily reach the site of were identified running adjacent to the first metatarsal and superficial implant, without being kinked or stretched during insetting. No fixa- to the first dorsal interosseous muscle (Figure 2a). FDMV provided tion was used (Figure 3). Plantar pain resolved after surgery. No com- several periosteal branches to the proximal first metatarsal metaphy- plications occurred. sis. A 7 mm  3 mm pedicled metatarsal bone flap (PMBF) was A plaster splint was placed for 3 weeks, after which an orthosis obtained from the lateral proximal first metatarsal metaphysis with preventing forefoot weight-bearing was placed. Six weeks after the the aid of a chisel (Figure 2b). The PMBF was designed slightly larger intervention, the patient was allowed to bear full weight; X-ray imag- than the bone defect (6 mm  3 mm). ing showed no donor site morbidity from the sustracted bone flap that 10982752, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/micr.31058 by Readcube (Labtiva Inc.), Wiley Online Library on [18/12/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License SOLDADO ET AL. 3 could warrant against it (Figure 4a,b). No specific rehabilitation proto- col was performed, and 4 months later, the patient resumed dancing. Twelve months after the intervention, MRI showed signal normaliza- tion, suggesting bone revascularization (Figure 4c,d). Dorsal and plan- tar active and passive second MTTP joint ROM was 45 and 60, and she has resumed her dancing activities. She has remained asymptom- atic for the past 42 months (Figure 5). 3 | DI SCU SSION It is generally accepted that Freiberg's disease has a multifactorial cause, including trauma, foot mechanics, arterial insufficiency, sys- temic disorders and a genetic component (Blitz & Yu, 2005; Carmont et al., 2009; Wax & Leland, 2019). Conservative measures are the first line of treatment when managing symptomatic cases, and it is thought to be effective in pain relief in 60% of non-athletes (Mizel & F I G U R E 3 Schematic representation of the surgical technique. Yodlowski, 1995). Given the poor prognosis for conservative treat- Graft harvesting from the first dorsal metatarsal vessels and bone ment, various surgical alternatives divided into joint-preserving and tunnel with pedicled bone flap implantation. joint-reconstructing procedures have been developed. In contrast, no F I G U R E 4 (a, b) A-P X-ray showing MTH revascularization; (c, d) T2-weighted-magnetic resonance shows normal intensity around the axial area of the MTH and complete healing of the donor's site. F I G U R E 5 Magnetic resonance images at 42 months: (a, c) are T2-weighted and (b, d) are T1-weighted-magnetic resonance images that show normal intensity that resembles normal healthy bone at the second metatarsal's head and dorsum. 10982752, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/micr.31058 by Readcube (Labtiva Inc.), Wiley Online Library on [18/12/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 4 SOLDADO ET AL. single operation has received unanimous support (Schade, 2015; Although less invasive techniques like joint debridement, core Seybold & Zide, 2018). In a recent review, Schade found better out- decompression, autogenous bone grafting and metatarsal osteotomies comes in terms of complete resolution of pain and return to activity (Seybold & Zide, 2018) have also shown promising results regarding with joint-preserving procedures as opposed to joint-reconstructing forefoot function and pain relief, these techniques have not shown ones (Schade, 2015). any revascularization potential. On this matter, vascularized periosteal Revascularizing techniques have proven effective in osteone- and bone flaps have shown revascularization properties in many chal- croses, such as osteonecrosis of the femoral head (ONFH), Kienböck's, lenging scenarios (Barrera-Ochoa et al., 2019; Urbaniak et al., 1995) Preiser's, proximal pole scaphoid osteonecrosis, among others based on the cambium layer's excellent osteogenic and angiogenic (Payatakes & Sotereanos, 2009; Urbaniak et al., 1995). Our rationale properties at this age (Gilbert & Mathoulin, 2000; Lin et al., 2014). for applying this technique follows free vascularized fibular grafting Other properties of vascularized periosteal bone flaps in children (FVFG) in ONFH by providing an angiogenic source and mechanical include their ease of harvesting at a young age and the flap's elasticity support (Fontecha et al., 2016; Urbaniak et al., 1995). FVFG, following readily permits it to conform to the recipient bed configuration. debridement of the necrotic bone, by using an extra-articular tunnel Although we used a technically more demanding technique than other and spongious bone grafting, has been shown to modify the natural described treatments, it was not more invasive. history of this disease drastically (Eward et al., 2012; Korompilias Postoperative radiological studies suggested that the non- et al., 2009; Urbaniak et al., 1995). This has been attributed to the resected cancellous metatarsal bone was revascularized. In addition, spongious bone graft's revascularization and the fibula's cortices' the powerful osteogenic properties of the periosteum led to new mechanical support (Fontecha et al., 2016; Korompilias et al., 2009). bone formation, conferring mechanical support and early initiation of Our technique, except for the necrotic bone debridement, was similar weight bearing. in that it was limited to the width of the tunnel. Moreover, following The current study suffers from some limitations inherent to a ONFH concepts, this patient had extensive transversal involvement case report, such as the small sample and the absence of compari- without severe joint collapse. sons with other treatment techniques as controls. The follow-up The fact that the natural history of Freiberg's disease and the risk was short to assess some parameters, such as the development of factors for joint collapse are not well known makes it problematic to osteoarthritis of the revascularized metatarsal head. Furthermore, propose evidence-based indications for revascularization techniques. we are applying concepts used in ONFH while Freiberg's natural This scientific dilemma occurs in different locations where osteone- history remains unknown and might differ significantly. Despite crosis can occur (Payatakes & Sotereanos, 2009). that, this technique was used in long-term refractory cases to con- For the past years, the vascular anatomy of the dorsum of the servative measures. A potential donor morbidity might be a frac- foot has been described with a very constant pattern (Petersen ture. However, considering the proportionally small size of the et al., 2002). Since metatarsal heads are distally located, only grafts graft obtained from the proximal metaphysis of the first metacar- based on the dorsal metatarsal vessels can be transplanted. Bone flaps pal, it might be negligible. Furthermore, a recent study has shown based on the lateral tarsal or arcuate vessels would not reach the the value of indocyanine for detection and prediction of conges- metatarsal head. A dorsal metatarsal artery, originating from the arcu- tion of pedicled flaps (Yoshimatsu et al., 2023), we believe this ate vessels, is present in each intermetatarsal space. Thus, a vascular- technique could have given additional valuable information. Clini- ized bone flap can be harvested to revascularize any metatarsal head. cal resolution and radiological imaging indicated bone revasculari- Similarly to Hou et al. (2013), we have found some variability of zation. Thus, our novel technique of using a vascularized bone the first dorsal metatarsal artery (FDMA) regarding its origin, size and graft to treat metatarsal's head osteonecrosis merits consideration, relationship with the first dorsal interosseous muscle (FDIM). In our while future comparative studies might be of scientific value. case, we found a small FDMA arising from the dorsalis pedis artery PMBF might be a valid alternative to other conventional joint- providing proximal branches to the proximal first metacarpal and run- preserving procedures to treat Freiberg's infraction, given its excellent ning distally, slightly covered by the FDIM. Some variants of the clinical and radiological results. The flap provides a revascularized FDMA might not be amenable to harvesting this flap. In such cases, environment that is favorable for healing. harvesting from the second dorsal metatarsal vessels remains an option. Soldado et al. (2013) described a vascularized periosteal flap CONFLIC T OF INTER E ST STATEMENT obtained from the FDMA (with antegrade flow) that was also effective Francisco Soldado is an Editorial Board member of Microsurgery and in revascularizing and preventing a collapse in osteonecrosis of the a co-author of this article. To minimize bias, they were excluded from talus. all editorial decision-making related to the acceptance of this article Some vascularized periosteal grafts and flaps have been described for publication. to treat foot and ankle osteonecrosis and avascular necrosis (Haddock et al., 2013). We opted for a bone flap owing to the difficulty of intro- DATA AVAILABILITY STAT EMEN T ducing a periosteal flap in a very narrow tunnel and because structural The data that support the findings of this study are available on bone has proven to be mechanically beneficial in preventing further request from the corresponding author. The data are not publicly collapse (Fontecha et al., 2016). available due to privacy or ethical restrictions. 10982752, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/micr.31058 by Readcube (Labtiva Inc.), Wiley Online Library on [18/12/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License SOLDADO ET AL. 5 CONS EN T Korompilias, A. V., Lykissas, M. G., Beris, A. E., Urbaniak, J. R., & The patient and parents were informed that data concerning the case Soucacos, P. N. (2009). Vascularised fibular graft in the management of femoral head osteonecrosis: Twenty years later. Journal of Bone and would be submitted for publication (case report and any accompany- Joint Surgery, 91(3), 287–293. ing images) and provided consent. Lin, Z., Fateh, A., Salem, D. M., & Intini, G. (2014). Periosteum: Biology and applications in craniofacial bone regeneration. Journal of Dental ORCID Research, 93, 109–116. Mizel, M. S., & Yodlowski, M. L. (1995). Disorders of the lesser metatarso- Francisco Soldado https://orcid.org/0000-0003-1759-9868 phalangeal joints. The Journal of the American Academy of Orthopaedic Felipe Moreira Borim https://orcid.org/0000-0002-4888-7822 Surgeons, 3(3), 166–173. Payatakes, A., & Sotereanos, D. G. (2009). Pedicled vascularized bone RE FE R ENC E S grafts for scaphoid and lunate reconstruction. The Journal of the Ameri- can Academy of Orthopaedic Surgeons, 17(12), 744–755. Barrera-Ochoa, S., Mendez-Sanchez, G., Mir-Bullo, X., Knörr, J., Petersen, W. J., Lankes, J. M., Paulsen, F., & Hassenpflug, J. (2002). The Bertelli, J. A., & Soldado, F. (2019). Vascularized thumb metacarpal arterial supply of the lesser metatarsal heads: A vascular injection periosteal flap for scaphoid nonunion in adolescents: A prospective study in human cadavers. Foot & Ankle International, 23(6), 491–495. cohort study of 12 patients. The Journal of Hand Surgery, 44(6), 521. Schade, V. L. (2015). Surgical management of Freiberg's infraction: A sys- e1–521.e11. tematic review. Foot & Ankle Specialist, 8(6), 498–519. Blitz, N. M., & Yu, J. H. (2005). Freiberg's infraction in identical twins: A Seybold, J. D., & Zide, J. R. (2018). Treatment of Freiberg disease. Foot and case report. The Journal of Foot and Ankle Surgery, 44(3), 218–221. Ankle Clinics, 23(1), 157–169. Carmont, M. R., Rees, R. J., & Blundell, C. M. (2009). Current concepts Soldado, F., Barrera-Ochoa, S., Fontecha, C. G., Haddad, S., Barastegui, D., review: Freiberg's disease. Foot & Ankle International, 30(2), Barber, I., & Rego, P. (2013). Vascularized periosteal graft from the first 167–176. metatarsal bone: A new technique to prevent collapse of osteonecro- Eward, W. C., Rineer, C. A., Urbaniak, J. R., Richard, M. J., & Ruch, D. S. sis of the talus in children. A case report. Microsurgery, 33(1), 56–59. (2012). The vascularized fibular graft in precollapse osteonecrosis: Is Urbaniak, J. R., Coogan, P. G., Gunneson, E. B., & Nunley, J. A. (1995). long-term hip preservation possible? Clinical Orthopaedics and Related Treatment of osteonecrosis of the femoral head with free vascularized Research, 470(10), 2819–2826. fibular grafting. A long-term follow-up study of one hundred and three Fontecha, C. G., Roca, I., Barber, I., Menendez, M. E., Collado, D., hips. The Journal of Bone and Joint Surgery. American Volume, 77, Mascarenhas, V. V., Barrera-Ochoa, S., & Soldado, F. (2016). Femoral 681–694. head bone viability after free vascularized fibular grafting for osteone- Wax, A., & Leland, R. (2019). Freiberg disease and avascular necrosis of crosis: SPECT/CT study. Microsurgery, 36(7), 573–577. the metatarsal heads. Foot and Ankle Clinics, 24(1), 69–82. Freiberg, A. (1914). Infraction of second metatarsal bone, a typical injury. Yoshimatsu, H., Karakawa, R., Scaglioni, M. F., Fuse, Y., & Yano, T. (2023). Surgery, Gynecology and Obstetrics, 19, 191–193. Use of intraoperative indocyanine green angiography for detection Gilbert, A., & Mathoulin, C. (2000). Vascularized bone grafts in children. and prediction of congestion in pedicled Island flaps. Microsurgery, Specifics and indications. Annales de Chirurgie Plastique et Esthétique, 1–8. 45(3), 309–322. [in French]. Gillespie, H. (2010). Osteochondroses and apophyseal injuries of the foot in the young athlete. Current Sports Medicine Reports, 9(5), 265–268. Haddock, N. T., Wapner, K., & Levin, L. S. (2013). Vascular bone transfer How to cite this article: Soldado, F., Moreira Borim, F., & options in the foot and ankle: A retrospective review and update on Knörr, J. (2023). Treatment of Freiberg's disease using a strategies. Plastic and Reconstructive Surgery, 132(3), 685–693. reverse pedicled metatarsal bone flap: Case report. Hou, Z., Zou, J., Wang, Z., & Zhong, S. (2013). Anatomical classification of Microsurgery, 1–5. https://doi.org/10.1002/micr.31058 the first dorsal metatarsal artery and its clinical application. Plastic and Reconstructive Surgery, 132(6), 1028e–1039e.