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Alteration of facial growth after radiotherapy: orthodontic, surgical and prosthetic rehabilitation
J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
Available online at
ScienceDirect www.sciencedirect.com
Case Report
Alteration of facial growth after radiotherapy: orthodontic, surgical and prosthetic rehabilitation P. Martin a, E. Muller b,*, C. Paulus c a
1, rue Saint Pierre de Vaise 69009 Lyon, France 132, avenue Franklin Roosevelt 69500 Bron, France c 5, rue Chambovet 69003 Lyon, France b
A R T I C L E I N F O
A B S T R A C T
Historique de l’article : Received 27 February 2019 Accepted 3 April 2019 Available online 10 April 2019
Most patients treated in childhood with radiotherapy for head and neck tumors develop craniofacial alterations related to impaired growth of the irradiated regions. These deformities have a significant functional and aesthetic impact, generating physiological and social troubles. The therapeutic solutions are mostly surgical and multidisciplinary. In this article, we describe the orthodontic and surgical care of a teenager treated with radiotherapy for a bilateral retinoblastoma at the age of 8 month and a half. The patient presented growth defects of the middle third of the face. These growth deficiencies are at the origin of significant aesthetic and physical disorders. Moreover, the three-dimensional skeletal abnormalities and the histological changes of these irradiated tissues make surgical management of the cases complex and specific.
C 2019 Published by Elsevier Masson SAS.
Keywords: Cranial irradiation Adverse effects Craniofacial abnormalities Dental implantation Maxillofacial development Radiation effects Orthognathic surgical procedures Bone graft
1. Introduction With an incidence of 1 on 15,000 births, the most common intraocular malignancy in childhood is retinoblastoma. The treatment of this tumor by ionizing radiation is reserved for the most extensive cases because of the risk of secondary sarcomas and the consequences of irradiation on developing tissues [1]. Amongst the known repercussions of face irradiation, there are immediate side effects impacting the child care during curative treatment. And delayed side effects, related to changes in the tissue environment (such as increased risk of osteoradionecrosis (ORN), the appearance of secondary tumor. . .). They have implications for future interventions that these patients will require [2]. The treatment of patients with radiation-induced facial dysmorphism after head growth is complex in several aspects. On the one hand, these malformations have a strong physiological and physical impact, sources of poor social integration, loss of selfconfidence and anxiety [2]. On the other hand, the histological changes of the irradiated tissues (with the appearance of ORN risk, poor regeneration capacity) require adapted surgical treatments.
* Corresponding author at: 132, avenue Franklin Tel Roosevelt, 69500 Bron, France. E-mail address: [email protected] (E. Muller). https://doi.org/10.1016/j.jormas.2019.04.004 C 2019 Published by Elsevier Masson SAS. 2468-7855/
Moreover, due to the importance of aesthetic and functional deficits induced by the deterioration of growth, the proposed surgical treatments are often included in a multidisciplinary therapy. In this article we describe the orthodontic-surgical care of a teenager treated with radiotherapy for a bilateral retinoblastoma at the age of 8 months and a half. 2. Observation Quentin, 8 months and a half, was brought in consultation for a strabismus detected by his parents. Using an examination of the ocular fundus, magnetic resonance imaging (MRI) and a computed tomography (CT), a bilateral retinoblastoma diagnosis was established. After evaluating the tumor extension, a conservative treatment was set up. It consisted of external radiotherapy of the main tumor, followed by cryoapplication of the residual lesions. The child received a total dose of 40 Gy (2.5 Gy per session, alternating right and left side), 20 Gy per side, the rest of the face and neck was protected by a lead mask. It should be noted that there was a side effect during treatment: a sleep disorder that disappeared after stopping the therapy. Short-term follow-up (one year after radiation treatment) of the child showed normal height and weight measurements. Subsequently, a growth abnormality
370
P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
appeared on the irradiated regions and was accentuated during the development of the patient. At the age of 18, Quentin had a bilateral tridimensional growth alteration associated with dental disorders (severe hypodontia). Clinical and complementary examinations allowed to determine the skeletal alteration: in transversal: o hypotelorism, o a decrease in inter-zygomatic distance; in sagittal and vertical: o hypoplasia of the maxilla, o at the dental level, microdontia and hypodontia; and at the occlusal level: a class III mesiocclusion. The 18-year-old patient and his parents were keenly seeking a functional and aesthetic rehabilitation (Fig. 1). The patient, who attended school at the time, presented a strong psycho-social disorder related to this malformation. A multi-stage orthodontic-surgical treatment was proposed: a pre-surgical orthodontic phase. three surgical procedures (intermaxillary disjunction, LeFort I osteotomy and bone graft, followed by implantology). occluso-prosthetic rehabilitation. In order to prepare for the upper jaw surgical modifications, an orthodontic treatment was used on the mandibular teeth (not affected by microdontia). Then, to remove the transversal deficit, a surgically assisted maxillary expansion by transpalatal distraction was performed and a bone borne transpalatal distraction devices (TPD) was placed to improve maxillary extension (Fig. 2). The second intervention, this time to reduce the vertical and sagittal deficit, was scheduled 8 months after the first. A LeFort I osteotomy was performed and the TPD removed. The forward movement of the maxilla enabled to reduce the skeletal class III and the vertical deficit. At the same time, a bone graft apposition (with autologous bone harvested from the iliac crest) was performed. The aim was to improve the transverse gain of the bone disjunction, and to allow implant placement in a favorable axis (Fig. 3a and b). After 10 months of consolidation, the third two-stage intervention was performed in the maxillary. First, the residual teeth were extracted, then ten endosseous implants were placed. The initial bone graft was reinforced with a synthetic bone graft substitutes (Bioss8 Gestlitch) and covered with a platelet-rich fibrin (PRF) membrane (Fig. 4). A temporary removable prosthesis was put in place during the waiting time prior to implant loading. After a healing period of 8 months, necessary for implant osseointegration, an implant-supported fixed bridge was set up to
Fig. 1. Photo showing maxillary hypodontia.
Fig. 2. Lateral cephalometric radiography showing skeletal growth loss and the TPD.
rehabilitate the occlusion and smile of the patient (Fig. 5a and b). Class I report and functional occlusion were obtained. From the aesthetic point of view, we were able to obtain better superior lip support (Fig. 6). The surgical treatments and prosthodontic restoration of a proper vertical dimension of occlusion enabled the patient to obtain more acceptable facial proportions (Fig. 7a and b).
Fig. 3. a: Panoramic radiograph showing patient hypodontia, at 18 y.-o (before treatment); b: Panoramic radiograph after the second surgery (bone graft and Le Fort I osteotomy).
P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
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Fig. 6. Dental rehabilitation with an incisor Class I occlusion. Fig. 4. Photos showing dental implant placement during the third intervention.
3. Discussion In the care of irradiated patients during childhood, the occurrence of complications depends on the location of the irradiation, the dose delivered and the age of the patient during anti-tumoral treatment. On the anatomical level, ionizing radiation causes a modification of the histological properties of the affected tissues (osteocyte destruction, microvascular damage, fibrosis in
Fig. 5. a and b: Placement of the implant-supported fixed bridge.
the medullary space) [2]. In surgical approaches, risks of ORN complications ensue from these tissue alterations. The latter causing a lasting decrease in the cellular defense and healing potential [3]. In the case presented in this article, the surgical treatment of these regions required complex procedures adapted to the tissue weakening following radiotherapy. Regarding the dose and age of administration of radiotherapy, patients treated with craniofacial irradiation before 5 years and receiving a dose over 30 Gy, develop skeletal abnormalities in 90% of cases [2,4]. Facial asymmetries affecting irradiated children have substantial psychosocial implications. The structural defect worsens with the development of the child to a maximum after pubertal growth. Facial growth is a complex, three-dimensional mechanism comprising many vasculonervous organs and pedicles. The growth depends on the development of other anatomical structures and surrounding tissues, and it responds to different hormonal and mechanical stimulations [2,5]. The complexity of this skeletal structure, its development and the histological consequences of irradiations on these same regions make it impossible to perform surgery with normal procedures [2]. In the treated case, Quentin accumulated 20 Gy per side, 40 Gy in total during radiotherapy. According to the literature, cases of osteoradionecrosis are generally found for irradiations above 60 Gy and most often located in the mandible [6]. Hypovascularization of irradiated tissue may be a risk factor for implant failure [7,8]. Although the patient has undergone irradiation below the mentioned thresholds, possible risks of developing complications remain. Correspondingly, the surgical therapy was adapted to minimize this risk. Surgical approaches were concentrated on the upper maxillary, more vascularized, and less predisposed to ORNs than the mandible. The approaches and detachments were carried out at a minimum. The use of vasoconstrictors during local anesthesia was avoided. Quentin and his parents were very keen for a therapeutic solution despite the risks incurred, clearly explained and exposed (risk of ORN, poor bone healing, graft loss, nonintegration of implants). The patient, who was at high school at the time and practiced theater, was extremely motivated by the treatment; he suffered a functional and aesthetic daily discomfort, which was more and more difficult to support. A fixed implant-supported prosthodontic rehabilitation was proposed, involving several surgical, orthodontic, prosthetic and orthophonic phases.
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P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
Fig. 7. a and b: Patient face (7a) and profile (7b) before and after rehabilitation.
On the functional level, the patient was able to recover a class I occlusion, a restoration of the manducatory function and a correction of the crossed articulate. On the aesthetic level, the vertical, sagittal and transversal skeletal deficit have been lifted. The upper lip became naturally supported. Accordingly, the patient gained a proper smile. Phonation defects were supported by follow-up with a speechlanguage pathologist. The treatment of Quentin had a favorable socio-psychological impact, with a gain of confidence from the patient, as well as from his entourage, thanks to the reduction of the skeletal facial anomaly, the disappearance of phonation disorders and the rehabilitation of a harmonious smile curve. Disclosure of interest The authors declare that they have no competing interest.
References [1] Aerts I, Lumbroso-Le Rouic L, Gauthier-Villars M, Brisse H, Doz F. Actualite´s du re´tinoblastome. Arch Pediatr 2016;23:112–6. [2] Gevorgyan A, La Scala GC, Neligan PC, Pang CY, Forrest CR. Radiation-induced craniofacial bone growth disturbances. J Craniofac Surg 2007;18:1001. [3] Vissink A, Jansma J, Spijkervet FKL, Burlage FR, Coppes RP. Oral sequelae of head and neck radiotherapy. Crit Rev Oral Biol Med Off Publ Am Assoc Oral Biol 2003;14:199–212. [4] Dahllo¨f G. Craniofacial growth in children treated for malignant diseases. Acta Odontol Scand 1998;56:378–82. [5] Mercier J-M, Perrin J-P, Longis J, Arzul L, Corre P. [Facial asymmetries and their skeletal component]. Rev Stomatol Chir Maxillo-Faciale Chir Orale 2014;115:219–28. [6] Dhanda J, Pasquier D, Newman L, Shaw R. Current concepts in osteoradionecrosis after head and neck radiotherapy. Clin Oncol 2016;28:459–66. [7] Colella G, Cannavale R, Pentenero M, Gandolfo S. Oral implants in radiated patients: a systematic review. Int J Oral Maxillofac Implants 2007;22:616–22. [8] Ihde S, Kopp S, Gundlach K, Konstantinovic´ VS. Effects of radiation therapy on craniofacial and dental implants: a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontology 2009;107:56–65.
Available online at
ScienceDirect www.sciencedirect.com
Case Report
Alteration of facial growth after radiotherapy: orthodontic, surgical and prosthetic rehabilitation P. Martin a, E. Muller b,*, C. Paulus c a
1, rue Saint Pierre de Vaise 69009 Lyon, France 132, avenue Franklin Roosevelt 69500 Bron, France c 5, rue Chambovet 69003 Lyon, France b
A R T I C L E I N F O
A B S T R A C T
Historique de l’article : Received 27 February 2019 Accepted 3 April 2019 Available online 10 April 2019
Most patients treated in childhood with radiotherapy for head and neck tumors develop craniofacial alterations related to impaired growth of the irradiated regions. These deformities have a significant functional and aesthetic impact, generating physiological and social troubles. The therapeutic solutions are mostly surgical and multidisciplinary. In this article, we describe the orthodontic and surgical care of a teenager treated with radiotherapy for a bilateral retinoblastoma at the age of 8 month and a half. The patient presented growth defects of the middle third of the face. These growth deficiencies are at the origin of significant aesthetic and physical disorders. Moreover, the three-dimensional skeletal abnormalities and the histological changes of these irradiated tissues make surgical management of the cases complex and specific.
C 2019 Published by Elsevier Masson SAS.
Keywords: Cranial irradiation Adverse effects Craniofacial abnormalities Dental implantation Maxillofacial development Radiation effects Orthognathic surgical procedures Bone graft
1. Introduction With an incidence of 1 on 15,000 births, the most common intraocular malignancy in childhood is retinoblastoma. The treatment of this tumor by ionizing radiation is reserved for the most extensive cases because of the risk of secondary sarcomas and the consequences of irradiation on developing tissues [1]. Amongst the known repercussions of face irradiation, there are immediate side effects impacting the child care during curative treatment. And delayed side effects, related to changes in the tissue environment (such as increased risk of osteoradionecrosis (ORN), the appearance of secondary tumor. . .). They have implications for future interventions that these patients will require [2]. The treatment of patients with radiation-induced facial dysmorphism after head growth is complex in several aspects. On the one hand, these malformations have a strong physiological and physical impact, sources of poor social integration, loss of selfconfidence and anxiety [2]. On the other hand, the histological changes of the irradiated tissues (with the appearance of ORN risk, poor regeneration capacity) require adapted surgical treatments.
* Corresponding author at: 132, avenue Franklin Tel Roosevelt, 69500 Bron, France. E-mail address: [email protected] (E. Muller). https://doi.org/10.1016/j.jormas.2019.04.004 C 2019 Published by Elsevier Masson SAS. 2468-7855/
Moreover, due to the importance of aesthetic and functional deficits induced by the deterioration of growth, the proposed surgical treatments are often included in a multidisciplinary therapy. In this article we describe the orthodontic-surgical care of a teenager treated with radiotherapy for a bilateral retinoblastoma at the age of 8 months and a half. 2. Observation Quentin, 8 months and a half, was brought in consultation for a strabismus detected by his parents. Using an examination of the ocular fundus, magnetic resonance imaging (MRI) and a computed tomography (CT), a bilateral retinoblastoma diagnosis was established. After evaluating the tumor extension, a conservative treatment was set up. It consisted of external radiotherapy of the main tumor, followed by cryoapplication of the residual lesions. The child received a total dose of 40 Gy (2.5 Gy per session, alternating right and left side), 20 Gy per side, the rest of the face and neck was protected by a lead mask. It should be noted that there was a side effect during treatment: a sleep disorder that disappeared after stopping the therapy. Short-term follow-up (one year after radiation treatment) of the child showed normal height and weight measurements. Subsequently, a growth abnormality
370
P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
appeared on the irradiated regions and was accentuated during the development of the patient. At the age of 18, Quentin had a bilateral tridimensional growth alteration associated with dental disorders (severe hypodontia). Clinical and complementary examinations allowed to determine the skeletal alteration: in transversal: o hypotelorism, o a decrease in inter-zygomatic distance; in sagittal and vertical: o hypoplasia of the maxilla, o at the dental level, microdontia and hypodontia; and at the occlusal level: a class III mesiocclusion. The 18-year-old patient and his parents were keenly seeking a functional and aesthetic rehabilitation (Fig. 1). The patient, who attended school at the time, presented a strong psycho-social disorder related to this malformation. A multi-stage orthodontic-surgical treatment was proposed: a pre-surgical orthodontic phase. three surgical procedures (intermaxillary disjunction, LeFort I osteotomy and bone graft, followed by implantology). occluso-prosthetic rehabilitation. In order to prepare for the upper jaw surgical modifications, an orthodontic treatment was used on the mandibular teeth (not affected by microdontia). Then, to remove the transversal deficit, a surgically assisted maxillary expansion by transpalatal distraction was performed and a bone borne transpalatal distraction devices (TPD) was placed to improve maxillary extension (Fig. 2). The second intervention, this time to reduce the vertical and sagittal deficit, was scheduled 8 months after the first. A LeFort I osteotomy was performed and the TPD removed. The forward movement of the maxilla enabled to reduce the skeletal class III and the vertical deficit. At the same time, a bone graft apposition (with autologous bone harvested from the iliac crest) was performed. The aim was to improve the transverse gain of the bone disjunction, and to allow implant placement in a favorable axis (Fig. 3a and b). After 10 months of consolidation, the third two-stage intervention was performed in the maxillary. First, the residual teeth were extracted, then ten endosseous implants were placed. The initial bone graft was reinforced with a synthetic bone graft substitutes (Bioss8 Gestlitch) and covered with a platelet-rich fibrin (PRF) membrane (Fig. 4). A temporary removable prosthesis was put in place during the waiting time prior to implant loading. After a healing period of 8 months, necessary for implant osseointegration, an implant-supported fixed bridge was set up to
Fig. 1. Photo showing maxillary hypodontia.
Fig. 2. Lateral cephalometric radiography showing skeletal growth loss and the TPD.
rehabilitate the occlusion and smile of the patient (Fig. 5a and b). Class I report and functional occlusion were obtained. From the aesthetic point of view, we were able to obtain better superior lip support (Fig. 6). The surgical treatments and prosthodontic restoration of a proper vertical dimension of occlusion enabled the patient to obtain more acceptable facial proportions (Fig. 7a and b).
Fig. 3. a: Panoramic radiograph showing patient hypodontia, at 18 y.-o (before treatment); b: Panoramic radiograph after the second surgery (bone graft and Le Fort I osteotomy).
P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
371
Fig. 6. Dental rehabilitation with an incisor Class I occlusion. Fig. 4. Photos showing dental implant placement during the third intervention.
3. Discussion In the care of irradiated patients during childhood, the occurrence of complications depends on the location of the irradiation, the dose delivered and the age of the patient during anti-tumoral treatment. On the anatomical level, ionizing radiation causes a modification of the histological properties of the affected tissues (osteocyte destruction, microvascular damage, fibrosis in
Fig. 5. a and b: Placement of the implant-supported fixed bridge.
the medullary space) [2]. In surgical approaches, risks of ORN complications ensue from these tissue alterations. The latter causing a lasting decrease in the cellular defense and healing potential [3]. In the case presented in this article, the surgical treatment of these regions required complex procedures adapted to the tissue weakening following radiotherapy. Regarding the dose and age of administration of radiotherapy, patients treated with craniofacial irradiation before 5 years and receiving a dose over 30 Gy, develop skeletal abnormalities in 90% of cases [2,4]. Facial asymmetries affecting irradiated children have substantial psychosocial implications. The structural defect worsens with the development of the child to a maximum after pubertal growth. Facial growth is a complex, three-dimensional mechanism comprising many vasculonervous organs and pedicles. The growth depends on the development of other anatomical structures and surrounding tissues, and it responds to different hormonal and mechanical stimulations [2,5]. The complexity of this skeletal structure, its development and the histological consequences of irradiations on these same regions make it impossible to perform surgery with normal procedures [2]. In the treated case, Quentin accumulated 20 Gy per side, 40 Gy in total during radiotherapy. According to the literature, cases of osteoradionecrosis are generally found for irradiations above 60 Gy and most often located in the mandible [6]. Hypovascularization of irradiated tissue may be a risk factor for implant failure [7,8]. Although the patient has undergone irradiation below the mentioned thresholds, possible risks of developing complications remain. Correspondingly, the surgical therapy was adapted to minimize this risk. Surgical approaches were concentrated on the upper maxillary, more vascularized, and less predisposed to ORNs than the mandible. The approaches and detachments were carried out at a minimum. The use of vasoconstrictors during local anesthesia was avoided. Quentin and his parents were very keen for a therapeutic solution despite the risks incurred, clearly explained and exposed (risk of ORN, poor bone healing, graft loss, nonintegration of implants). The patient, who was at high school at the time and practiced theater, was extremely motivated by the treatment; he suffered a functional and aesthetic daily discomfort, which was more and more difficult to support. A fixed implant-supported prosthodontic rehabilitation was proposed, involving several surgical, orthodontic, prosthetic and orthophonic phases.
372
P. Martin et al. / J Stomatol Oral Maxillofac Surg 120 (2019) 369–372
Fig. 7. a and b: Patient face (7a) and profile (7b) before and after rehabilitation.
On the functional level, the patient was able to recover a class I occlusion, a restoration of the manducatory function and a correction of the crossed articulate. On the aesthetic level, the vertical, sagittal and transversal skeletal deficit have been lifted. The upper lip became naturally supported. Accordingly, the patient gained a proper smile. Phonation defects were supported by follow-up with a speechlanguage pathologist. The treatment of Quentin had a favorable socio-psychological impact, with a gain of confidence from the patient, as well as from his entourage, thanks to the reduction of the skeletal facial anomaly, the disappearance of phonation disorders and the rehabilitation of a harmonious smile curve. Disclosure of interest The authors declare that they have no competing interest.
References [1] Aerts I, Lumbroso-Le Rouic L, Gauthier-Villars M, Brisse H, Doz F. Actualite´s du re´tinoblastome. Arch Pediatr 2016;23:112–6. [2] Gevorgyan A, La Scala GC, Neligan PC, Pang CY, Forrest CR. Radiation-induced craniofacial bone growth disturbances. J Craniofac Surg 2007;18:1001. [3] Vissink A, Jansma J, Spijkervet FKL, Burlage FR, Coppes RP. Oral sequelae of head and neck radiotherapy. Crit Rev Oral Biol Med Off Publ Am Assoc Oral Biol 2003;14:199–212. [4] Dahllo¨f G. Craniofacial growth in children treated for malignant diseases. Acta Odontol Scand 1998;56:378–82. [5] Mercier J-M, Perrin J-P, Longis J, Arzul L, Corre P. [Facial asymmetries and their skeletal component]. Rev Stomatol Chir Maxillo-Faciale Chir Orale 2014;115:219–28. [6] Dhanda J, Pasquier D, Newman L, Shaw R. Current concepts in osteoradionecrosis after head and neck radiotherapy. Clin Oncol 2016;28:459–66. [7] Colella G, Cannavale R, Pentenero M, Gandolfo S. Oral implants in radiated patients: a systematic review. Int J Oral Maxillofac Implants 2007;22:616–22. [8] Ihde S, Kopp S, Gundlach K, Konstantinovic´ VS. Effects of radiation therapy on craniofacial and dental implants: a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontology 2009;107:56–65.