Abstract
Purpose
There is no consensus on the type of surgical treatment of congenital pseudarthrosis of the clavicle due to its rarity. The purpose of this study is to provide evidence in favor of a surgical technique and to explore factors correlated with bone consolidation.
Methods
Systematic review of the literature and an analysis of the data for each subject, including all available subjects from the published series and clinical cases since 1990. Fisher’s exact tests or T-tests were used to evaluate the effect of independent variables (age at surgery and type of treatment) on bone healing.
Results
The literature search provides 305 articles; 30 were selected, reporting 191 patients and 194 clavicles. One hundred and fifty-one clavicles were operated on at a mean age of nine years and four months (from 8 months to 21 years). Thirteen clavicles (8, 6%) had not consolidated at the last follow-up. Concerning the type of fixation, the rate of healing was similar for plates and pins (p = 0.27). The rate of consolidation was higher with autograft than with allograft, xenograft, or no graft (p = 0.00001), and was 100% for vascularized graft. The mean age at surgery was higher for patients who healed at the last follow-up (118 vs. 61 months, p = 0.001).
Conclusion
In the event of surgical indication for congenital pseudarthrosis of the clavicle, it is recommended to perform autograft and stable fixation (level 4) after seven years old (level 4).
Similar content being viewed by others
Data availability
No availability of data and materials.
References
Persiani P, Molayem I, Villani C et al (2008) Surgical treatment of congenital pseudarthrosis of the clavicle : a report on 17 cases. Acta Orthop Belg 74:161–166
Cadilhac C, Odent T, Finidori G, et al (2021) Pseudarthrose congénitale de la clavicule [Internet]. In: Orthopédie pédiatrique. Elsevier; [cité 2021 févr 20]. page 57‑60.Available from: https://linkinghub.elsevier.com/retrieve/pii/B9782294701764500074
Alldred AJ (1963) Congenital pseudarthrosis of the clavicle. J Bone Joint Surg Br 45-B:312‑9.
Solla F, Bertoncelli CM, Rampal V (2020) Does the PROSPERO registration prevent double review on the same topic? BMJ Evid-Based Med bmjebm-2020–111361.
Solla F, Lemoine J, Musoff C et al (2019) Surgical treatment of congenital pseudarthrosis of the forearm: review and quantitative analysis of individual patient data. Hand Surg Rehabil 38:233–241
Ullot Font R, Cepero Campà S, Gargantilla Vázquez A et al (2006) Seudoartrosis congénita de clavícula. An Pediatría 2006(64):273–276
Kohler R, Chappuis JP, Daudet M (1980) Congenital pseudarthrosis of the clavicle. Apropos of 7 cases. Chir Pediatr 21:201–7
Galanopoulos I, Ashwood N, Garlapati AK, et al (2012) Congenital pseudarthrosis of clavicle: illustrated operative technique and histological findings. Case Rep 2012;2012:bcr2012006908‑bcr2012006908.
Kołodziej L, Bohatyrewicz A, Kotrych D (2008) Congenital pseudoarthrosis of the clavicle: is operative treatment necessary? A report of four cases and literature review. Chir Narzadow Ruchu Ortop Pol 73:277‑80, 252‑6.
Faraud A, Bonnevialle N, Allavena C et al (2014) Outcomes from surgical treatment of middle-third clavicle fractures nonunion in adults: a series of 21 cases. Orthop Traumatol Surg Res 100:175–180
Lazarides S, Zafiropoulos G (2006) Conservative treatment of fractures at the middle third of the clavicle: the relevance of shortening and clinical outcome. J Shoulder Elbow Surg 15:191–194
Hollo D, Kolling C, Audigé L et al (2020) Plating and cortical bone grafting of clavicular nonunions: clinical outcome and its relation to clavicular length restoration. JSES Int 4:508–514
Ettl V, Wild A, Krauspe R et al (2005) Surgical treatment of congenital pseudarthrosis of the clavicle: a report of three cases and review of the literature. Eur J Pediatr Surg 15:56–60
Studer K, Baker MP, Krieg AH (2017) Operative treatment of congenital pseudarthrosis of the clavicle: a single-centre experience. J Pediatr Orthop B 26:245–249
Kohler R, Solla F, Pinson S et al (2005) Pseudarthrose congénitale de l’avant-bras associée à la neurofibromatose. Rev Chir Orthopédique Réparatrice Appar Mot 91:773–781
Stephen B. Schnall, John D. King et al (1988) Congenital pseudarthrosis of clavicle: a review of litterature and surgical result of six cases. J Pediatr Orthop 316‑21.
LeGaye J, Noel H, Lokietek W (1991) Congenital pseudarthrosis of the clavicle. Apropos of a case report and literature review. Acta Orthop Belg 57:209–12
Schoenecker PL, Johnson GE, Howard B et al (1992) Congenital pseudarthrosis. Orthop Rev 1992(21):855–860
Köster G, Kunze E, von Knoch M (2008) (2008) Die operative Behandlung der kongenitalen Klavikulapseudarthrose. Z Für Orthop Ihre Grenzgeb 137:414–418
Malawski S, Zasacki W (1999) (1999) Congenital pseudoarthrosis of the clavicle: review of literature and a case report. Chir Narzadow Ruchu Ortop Pol 64:341–345
Cadilhac C, Fenoll B, Peretti A (2020) Etude de 25 cas chez l’enfant 2020;6.
Lorente Molto FJ, Bonete Lluch DJ, Garrido IM (2001) (2001) Congenital pseudarthrosis of the clavicle: a proposal for early surgical treatment. J Pediatr Orthop 21:689–693
Dzupa V, Bartonicek J, Zidka M (2007) Fracture of the clavicle after surgical treatment for congenital pseudarthrosis. Med Sci Monit 4.
Güneş T, Erdem M (2005) Sen C (2005) A case of congenital pseudarthrosis of the clavicle. Acta Orthop Traumatol Turc 39:266–269
Lelei L (2005) Congenital pseudoarthrosis of the clavicle: case report. East Afr. Med. J. [Internet] 2005 [cité 2021 févr 21]; 82. Available from: http://www.ajol.info/index.php/eamj/article/view/9319
Jeon I-H, Wilson I, Angus WW (2006) Reconstruction of congenital pseudarthrosis of the clavicle in an adult. J Orthop Sci 11:229–231
Beslikas T, Dadoukis D, Gigis I et al (2007) Congenital pseudarthrosis of the clavicle: a case report. J Orthop Surg 15:87–90
Sponer P, Neumann D, Karpas K (2008) Congenital pseudoarthrosis of the clavicle in a boy with Prader-Willi’s syndrome. Acta Chir Orthop Traumatol Cech 75:134–136
Glotzbecker MP, Shin EK, Chen NC et al (2009) Salvage reconstruction of congenital pseudarthrosis of the clavicle with vascularized fibular graft after failed operative treatment: a case report. J Pediatr Orthop 29:411–415
Currarino G, Herring JA (2009) Congenital pseudarthrosis of the clavicle. Pediatr Radiol 39:1343–1349
Chandran P, George H, James LA (2011) Congenital clavicular pseudarthrosis: comparison of two treatment methods. J Child Orthop 5:1–4
Elliot RR, Richards RH (2011) Failed operative treatment in two cases of pseudarthrosis of the clavicle using internal fixation and bovine cancellous xenograft (Tutobone). J Pediatr Orthop B 20:349–353
Singla R, Devgan A, Gogna P et al (2014) Congenital pseudoarthrosis of the clavicle with bifurcation. Indian J Orthop 48:435
Salman Ghaffari, Masoud Shayesteh Azar, Mohammad Hossein (2015) Congenital pseudoarthrosis of the clavicle; a rare case. Int. Arch. Med. [Internet] 2015 [cité 2021 févr 21]; Available from: http://imed.pub/ojs/index.php/iam/article/view/967
Abdellaoui H, Atarraf K, Chater L, et al (2017) Congenital pseudarthrosis of the clavicle treated by Masquelet technique. BMJ Case Rep bcr-2017–221557.
Di Gennaro GL, Cravino M, Martinelli A et al (2017) Congenital pseudarthrosis of the clavicle: a report on 27 cases. J Shoulder Elbow Surg 26:e65-70
Dung T, Le K, Van B, Son T et al (2017) Congenital pseudarthrosis of the clavicle in a 7-year-old girl: a case report treated with smooth elastic intramedullary pinning without bone grafting and literature review. Open J Orthop Rheumatol 2:023–024
Giwnewer U, Rubin G, Dohovni E et al (2018) Congenital pseudoarthrosis of the clavicle: should we reconsider timing and fixation method? J Orthop Surg 26:230949901879953
Haddad B, Zribi S, Haraux E et al (2019) Induced membrane technique for clavicle reconstruction in paediatric patients: report of four cases. Orthop Traumatol Surg Res 105:733–737
Kim AE, Vuillermin CB, Bae DS et al (2020) Congenital pseudarthrosis of the clavicle: surgical decision making and outcomes. J Shoulder Elbow Surg 29:302–307
Author information
Authors and Affiliations
Contributions
All the authors contributed to the study conception and design. Material preparation, data collection, and analysis were performed by Federico Solla and Carlo Bertoncelli. The first draft of the manuscript was written by Cédric Assouto and Federico Solla, and all the authors commented on previous versions of the manuscript. All the authors read and approved the final manuscript.
Cédric Assouto performed the study and wrote the article.
Marc-Olivier Gauci supervised the study, wrote, and revised the article.
Sikha Bagui supervised the study, wrote, and revised the article.
Marco Monticone supervised the study, wrote, and revised the article.
Carlo Mario Bertoncelli supervised the study and revised the text.
Virginie Rampal supervised the study and revised the text.
Federico Solla conceived and supervised the study, wrote, and revised the text.
Corresponding author
Ethics declarations
Ethics approval and consent to participate
The present study did not require ethics approval.
Informed consent was obtained from all individual participants included in the study.
The authors affirm that human research participants provided informed consent for publication of the images in Fig. 1, 2, 3 and 4.
Conflict of interest
The authors declare no competing interests.
Additional information
Publisher's note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Assouto, C., Bertoncelli, C.M., Gauci, MO. et al. Congenital pseudarthrosis of the clavicle: a systematic review. International Orthopaedics (SICOT) 46, 2577–2583 (2022). https://doi.org/10.1007/s00264-022-05470-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00264-022-05470-6