Abstract
The reconstruction of mandibular defects with vascularized fibula flaps remains challenging in the paediatric population. The ability of the reconstructed mandible to grow remains controversial, and associated factors are unclear. A systematic search of the English-language and Chinese literature was conducted for the period January 1989 to April 2014 using selected key words associated with the topic. Individual patients aged <18 years who underwent mandibular reconstruction with the vascularized fibula flap and had known outcomes related to growth potential were included. Data on postoperative growth and associated factors, including condylar management, age at reconstruction, malignancy, and postoperative radiotherapy or chemotherapy, were reviewed systematically. In total, 51 patients reported in 15 articles were included. The proportion of patients with a preserved growth potential (58.8%) was higher than that of patients with no growth potential. Condylar preservation and reconstruction during the rapid growth period showed a trend towards an influence on the growth potential. Reconstruction after benign lesion resection seemed to facilitate postoperative growth, while postoperative radiotherapy inhibited growth. Reconstruction after benign lesion resection, reconstruction between 8 and 12 years of age, and condylar preservation facilitate postoperative mandibular growth, while postoperative radiotherapy inhibits the same.
The vascularized fibula flap was first introduced by Hidalgo in 1989, and since then it has become a popular graft for use in mandibular reconstruction. Compared with non-vascularized flaps, vascularized flaps offer the advantages of a minimal infection risk, a low absorption rate, the possibility of simultaneous implant insertion, and the ability to reconstruct soft tissue defects. Furthermore, vascularized fibula flaps can adapt to bone defects of various shapes and provide large vessel length and calibre for microvascular anastomosis and a double cortex layer for implant stability. Therefore, this flap is considered feasible for adult mandibular reconstruction.
However, mandibular reconstruction in the paediatric population remains a challenge, although it is gradually evolving with the development of appropriate anaesthesia techniques. Paediatric mandibular reconstruction with the vascularized fibula flap was first reported in 1993. Although the above-mentioned advantages, along with the high survival rate and minimal donor site morbidity, have lent immense popularity to this flap, the postoperative growth potential of the neomandible remains controversial. This uncertainty stems from several factors. Because the condyle is considered to be the growth centre for the entire mandible, its involvement by the lesion and preservation during surgery may affect the outcomes to a large extent. Patient age, which is related to the growth rate, is another potential contributing factor, along with factors such as malignancy and postoperative radiotherapy and chemotherapy. Impaired growth after surgery may affect functional and cosmetic outcomes, possibly leading to malocclusion and facial asymmetry. Such undesired outcomes, in turn, may affect nutrient ingestion, general health, and even the self-esteem of the child throughout life. However, there appears to be no systematic review on this topic available in the literature.
Therefore, this systematic review was conducted to evaluate the mandibular growth potential after reconstruction using the vascularized fibula flap in children. Details relevant to postoperative growth and associated influencing factors were collected, and the general outcomes and effects of those factors were assessed.
Patients and methods
Study identification
An initial online search of the PubMed, Ovid, and Embase databases for English-language articles and of the China National Knowledge Infrastructure (CNKI) and Chinese Scientific and Technological Journal (VIP) databases for Chinese publications was conducted for the period January 1989 to April 2014. A broad search strategy was implemented, utilizing the following key words: ‘mandibular reconstruction’, ‘paediatric’, ‘mandibular defects in children’, ‘vascularized fibula flap’, and ‘free fibula flap’. Two reviewers screened the titles and abstracts of the articles retrieved. Full-text articles were obtained for relevant studies and studies where the title and abstract information was insufficient to allow a consensus. The reference lists were also screened, and relevant citations were included in the next round.
Study selection
Two reviewers obtained and evaluated the full-text of articles retrieved during the first search round and from the reference list search. The following inclusion criteria were applied: individual age at reconstruction of <18 years, mandibulectomy and reconstruction with the vascularized fibula flap as the primary clinical procedure, and available follow-up data on the postoperative mandibular growth potential. The exclusion criteria were as follows: age >18 years, preoperative radiotherapy or chemotherapy, and no data on the growth ability of the grafted fibula segment. Articles in languages other than English and Chinese were also excluded.
During the retrieval procedure, the first reviewer reviewed the abstract and selected studies according to the inclusion and exclusion criteria. The second reviewer then reviewed and checked all the abstracts again and made his selection using the same criteria. The entire text of all selected studies was reviewed again by both reviewers in detail. Any differences were then discussed in consultation with a third party, until consensus was reached.
Study design
Considering the dearth of studies on the selected topic, those included in this review were limited to retrospective studies and case reports. There was no restriction on the publication date or status.
Outcome measures
Data on specific variables, including patient age, sex, pathological characteristics (benign or malignant), condylar management, postoperative radiotherapy or chemotherapy, follow-up period, and outcomes related to neomandibular growth or facial symmetry, were collected from each article. Cases reported from the same medical centre were contrasted to match and identify duplicate patient cases. The outcomes reported in the most recent study were adopted for such cases.
Data extraction and analysis
The first reviewer collected the data and imported it into an electronic database (Microsoft Excel). The second reviewer checked the extracted data for omissions or inaccuracies. The two reviewers discussed any differences and arrived at a consensus after consultation with a third party.
For the selected studies, preserved growth potential or postoperative growth potential was identified as an obvious actual subsequent growth after surgery. When a study included patients with both condylar resection and preservation, patients with preservation were classified into a ‘condyle preserved’ group, while those with resection were classified into a ‘condyle resected’ group.
A systematic review was conducted for patients with available postoperative growth potential data. The following factors were evaluated: age, condylar management (preserved or resected), pathological characteristics (benign or malignant), and postoperative radiotherapy or chemotherapy. According to the results of an anthropology study, children aged from 8 to 12 years show a high-level mandible growth rate, thus this period was termed the rapid growth stage. Based on this theory, the patients were divided into three age groups: 0–8, 8–12, and 12–18 years. The research and review strategy adopted in this study is presented in Fig. 1 .
Results
In total, 548 articles (167 in English and 381 in Chinese) were reviewed. Among these, 15 articles reporting the cases of 51 patients met the study inclusion criteria and were retrieved. Details of these 51 patients are presented in Tables 1 and 2 .
| First author | No. | Age, y | Sex | Diagnosis | Malignancy | Extension of the lesion | Condyle involved | Postop. XRT | Postop. Chemo | Follow-up | Outcome |
|---|---|---|---|---|---|---|---|---|---|---|---|
| Posnick, Iconomou, Phillips | 1 | 6 | F | Intraosseous arteriovenous malformation | Benign | Left parasymphysis to left ramus; 5 cm | No | No | No | 15 y | Growth |
| Posnick, Iconomou, Phillips | 2 | 6 | M | Primitive neuroectodermal tumour | Malignant | Left parasymphysis to right angle; 10 cm | No | No | Yes | 14+ y | Growth arrested |
| Iconomou, Phillips | 3 | 7 | M | Recurrent aggressive fibromatosis | Benign | Right parasymphysis to right condyle;12 cm | Yes | No | No | 15 y | Growth arrested |
| Iconomou, Phillips | 4 | 17 | F | Ewing sarcoma | Malignant | Left parasymphysis to left subcondyle; 10 cm | No | NA | NA | 6+ y | Growth arrested |
| Iconomou, Phillips | 5 | 17 | F | Hemifacial microsomia and frontal dysplasia | Benign | Left angle to left condyle; 8 cm | Yes | No | No | 6+ y | Growth arrested |
| Iconomou, Phillips | 6 | 3 | M | Fibrous dysplasia/ossifying fibroma | Benign | Right parasymphysis to left angle; 9 cm | No | No | No | 6+ y | Growth arrested |
| Phillips, Fenton | 7 | 5 | M | Metastatic retinoblastoma | Malignant | Left body to right body | No | NA | Yes | 4 y | Growth arrested |
| Olvera-Caballero | 8 | 10 | M | Giant dental cyst | Benign | Left condyle to midline | Yes | NA | NA | 1 y | Growth |
| Olvera-Caballero | 9 | 10 | M | Osteoid osteoma | Benign | Chin to the right ascending ramus | No | NA | NA | 1 y | Growth |
| Olvera-Caballero | 10 | 15 | F | Ameloblastoma | Benign | Chin to the right mandibular angle | No | NA | NA | 1 y | Growth |
| Olvera-Caballero | 11 | 10 | M | Ossifying fibroma | Benign | Left mandible body to left condyle | Yes | NA | NA | 1 y | Growth |
| Olvera-Caballero | 12 | 11 | M | Osteomyelitis | Benign | Right mandible body to right condyle | Yes | NA | NA | 1 y | Growth |
| Olvera-Caballero | 13 | 9 | M | Loss of bone after wound infection | Benign | Right mandible body | No | NA | NA | 1 y | Growth |
| Genden | 14 | 8 | M | Aggressive juvenile fibromatosis | Benign | Left ramus, body, and hemisymphyseal portion of mandible | No | No | No | 4 y 2 mo | Growth |
| Nahabedian | 15 | 10 | M | Ameloblastoma of the right mandible | Benign | Right angle to the condylar neck | No | NA | NA | 18 mo | Growth |
| Warren | 16 | 6 | M | Fibrous dysplasia | Benign | Right mandibular angle to left parasymphyseal area | No | NA | NA | 9–14 y | Growth |
| Bilkay | 17 | 14 | M | Osteosarcoma | Malignant | Mandible body | No | NA | NA | 2 y | Growth |
| Crosby | 18 | 12 | F | Rhabdomyosarcoma | Malignant | Left condyle, 10 cm | Yes | No | No | 10 y | Growth |
| Crosby | 19 | 5 | M | Desmoid fibromatosis | Benign | Left neck to parasymphysis, 6 cm | No | No | Yes | 6 y 5 mo | Growth |
| Crosby | 20 | 7 | M | Giant cell granuloma | Benign | Right angle to left mid-body, 10 cm | No | No | No | 4 y 9 mo | Growth |
| Crosby | 21 | 5 | F | Ewing sarcoma | Malignant | Right mandible, 7 cm | Yes | No | No | 4 y 9 mo | Growth arrested |
| Crosby | 22 | 14 | M | Desmoid fibromatosis | Benign | Parasymphysis to body, 10 cm | No | No | No | 2 y 8 mo | Growth arrested |
| Crosby | 23 | 14 | M | Osteosarcoma | Malignant | Left mandible, 8 cm | Yes | Yes | Yes | 2 y 4 mo | Growth |
| Crosby | 24 | 12 | F | Rhabdomyosarcoma | Malignant | Left mandible, 7 cm | Yes | No | No | 2 y | Growth arrested |
| Crosby | 25 | 6 | M | Desmoid fibromatosis | Benign | Right parasymphysis to left angle, 9 cm | No | No | No | 1 y 7 mo | Growth |
| Crosby | 26 | 13 | M | Ewing sarcoma | Malignant | Left mandible, 10 cm | Yes | No | No | 1 y 6 mo | Growth |
| Crosby | 27 | 13 | F | Ameloblastoma | Benign | Parasymphysis to parasymphysis, 6 cm | No | No | No | 11 mo | Growth |
| Crosby | 28 | 9 | F | Ameloblastoma | Benign | Right mandible, 16 cm | No | No | No | 9 mo | Growth |
| Guo | 29 | 18 | NA | Hemifacial microsomia, type 3 | Benign | NA | NA | No | No | 2+ y | Growth arrested |
| Guo | 30 | 16 | NA | Lymphoepithelioma | Benign | NA | NA | Yes | Yes | 2+ y | Growth arrested |
| Guo | 31 | 15 | F | Hemifacial microsomia, type 3 | Benign | Left ramus and condyle | Yes | No | No | 2+ y | Growth arrested |
| Guo | 32 | 8 | NA | Fibrosarcoma | Malignant | NA | NA | Yes | No | 2+ y | Growth arrested |
| Guo | 33 | 4 | NA | Gunshot wound to neck, chin | Benign | NA | NA | No | No | 2+ y | Growth arrested |
| Guo | 34 | 13 | NA | Ewing sarcoma | Malignant | NA | NA | No | No | 2+ y | Growth arrested |
| Guo | 35 | 11 | F | Ewing sarcoma | Malignant | Whole mandible | Yes | No | Yes | 2+ y | Growth arrested |
| Guo | 36 | 17 | NA | Rhabdomyosarcoma | Malignant | NA | NA | Yes | Yes | 2+ y | Growth arrested |
| Guo | 37 | 8 | M | Rhabdomyoma | Benign | Hemimandible | Yes | No | No | 2+ y | Growth arrested |
| Guo | 38 | 5 | NA | Neuroectodermal tumour | Benign | NA | NA | Yes | No | 2+ y | Growth arrested |
| Guo | 39 | 3 | NA | Rhabdomyosarcoma | Malignant | NA | NA | Yes | Yes | 2+ y | Growth arrested |
| Guo | 40 | 10 mo | NA | Germ cell tumour | Malignant | Right hemimandible | Yes | No | Yes | 2+ y | Growth arrested |
| Sinno | 41 | 1.7 | M | Fibromatosis (desmoid tumour) | Benign | Left mandible, 37 cm × 23 cm × 23 cm | No | NA | NA | 6 y | Growth |
| Li | 42 | 12 | F | Ameloblastoma | Benign | Right angle to left body, approximately 10 cm | No | NA | NA | 23 mo | Growth |
| Li | 43 | 13 | F | Ossifying fibroma | Benign | Lateral segment without condyle | No | NA | NA | 18 mo | Growth |
| Li | 44 | 14 | M | Ameloblastoma | Benign | Lateral segment without condyle | No | NA | NA | 16 mo | Growth |
| Li | 45 | 14 | M | Ameloblastoma | Benign | Hemimandible segment with condyle | Yes | NA | NA | 24 mo | Growth |
| Li | 46 | 15 | F | Fibrous dysplasia | Benign | Central segment | No | NA | NA | 28 mo | Growth |
| Li | 47 | 12 | F | Ameloblastoma | Benign | Lateral segment without condyle | No | NA | NA | 36 mo | Growth |
| Bianchi | 48 | 16 | M | Ewing sarcoma | Malignant | Right second premolar to right condyle | Yes | No | Yes | 6 y | Growth |
| Bianchi | 49 | 14 | M | Chronic sclerosing osteomyelitis | Benign | Left canine to left condylar neck | No | No | No | 6 y | Growth |
| Bianchi | 50 | 8 | M | Embryonal rhabdomyosarcoma | Malignant | Right second premolar to right condyle neck | No | No | Yes | 5 y | Growth |
| Pierse | 51 | 15 | M | Ganglioneuroma | Benign | Right condylar neck to right first premolar | No | No | No | 1 y | Growth |
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