The Journal of Craniofacial Surgery
Volume 26, Number 4, June 2015
9. Hagberg H, Lamberg K, Astro¨m G. Alpha-2b interferon and oral clodronate for Gorham’s disease. Lancet 1997;350:1822–1823 10. Handl-Zeller L, Hohenberg G. Radiotherapy of Morbus Gorham-Stout: the biological value of low irradiation dose. Br J Radiol 1990;63: 206–208
Emergency Dental Treatment of a Patient With Sturge-Weber Syndrome Maria Cecı´lia Querido de Oliveira, DDS, Vanessa Nobre Maia, DDS, MSc, PhD, Juliana Bertoldi Franco, DDS, and Maria Paula Siqueira de Melo Peres, DDS Abstract: The Sturge-Weber (SWS) syndrome is a rare condition with congenital capillary malformations. Hemorrhages may occur on dental treatment, which can have a dramatic effect on a patient’s vital sign. The aim of the present brief clinical study was to briefly report a case of a female patient with SWS who underwent an endodontic treatment. A 25-year-old patient (C.O.B.S.) with SWS and vascular malformation in cervicofacial right region was admitted to the dental clinic with pulsatile pain in tooth 16. An emergency dental treatment with cavity preparation and access to root canals was performed with intraligamentary and intrapulpal anesthesia with 3% prilocain 3% with felypressin 0.03 UI/mL because the tooth was located in the region of the vascular malformation, and any surgical treatment could lead to hemorrhages. After 7 days, the patient was recalled with the absence of painful symptoms. The endodontic treatment in patients with SWS is feasible, and safety should be an alternative to surgical treatments. Key Words: Endodontics, hydrocephalus, maxillofacial abnormalities
S
turge-Weber syndrome (SWS), also called encephalotrigeminal angiomatosis, is a congenital capillary malformation, which is usually unilateral and reaches 1 or more branches of the trigeminal nerve.1– 4 Also, SWS has cerebral involvement characterized by seizures and ophthalmologic abnormalities manifested by glaucoma and capillary malformations of the conjunctiva, episcleral, coronary, and retina.1 –4 Sturge-Weber syndrome is a rare condition with approximate prevalence of 1:50,000 born alive. The facial port-wine stains From the Center Institution of Hospital Clinics, Faculty of Medicine, University of Sao Paulo, Sao Paulo, Brazil. Received November 30, 2014. Accepted for publication December 26, 2014. Address correspondence and reprint requests to Maria Cecı´lia Querido de Oliveira, DDS, Center Institution of Hospital Clinics, Faculty of ´ guiar Medicine, University of Sao Paulo, Doutor Ene´as de Carvalho A St., 225, Cerqueira Ce´sar, Sao Paulo, SP 05403–000, Brazil; E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001684 #
2015 Mutaz B. Habal, MD
Brief Clinical Studies
FIGURE 1. Facial capillary malformation (port-wine stains).
(PWS) are vascular malformations characteristics of SWS and are manifested at birth with no tendency to regression and are most commonly located in the face and cervical region.5 Also, the commitment of ophthalmic branch of trigeminal nerve is pathognomonic of SWS syndrome.6 However, this syndrome is rare, and the hemostasis of patients with SWS may be a critical procedure if a dental surgery is required, thereby it should be conducted in hospitals because those hemorrhages may have a dramatic effect on a patient’s vital sign.6,7 Thus, the aim of this study was to (1) briefly report a case of a female patient with SWS who underwent an emergency dental treatment; and (2) to describe an operative approach of SWS patients for avoiding surgical treatment.
Brief Clinical Study A 25-year-old patient (C.O.B.S.) with SWS and vascular malformation in cervicofacial right region was admitted to the Dental Division, Center Institution of Hospital Clinics, Faculty of Medicine, University of Sao Paulo with pulsatile pain in tooth 16. The patient had facial capillary malformation (PWS) and ophthalmic commitment, manifested by vascular malformations in the conjunctiva of the right eye (Fig. 1). Also, the patient reported the presence of prosthetic valve in the brain because of a hydrocephalus episode (Fig. 2). The intraoral examination showed generalized gingivitis and extensive caries in tooth 16 with occlusal and distal faces affected. The radiographic examination showed the presence of a radiolucency image extending to the root canal and an important alveolar bone loss in the distal region. An emergency dental treatment with cavity preparation and access to root canals was performed with intraligamentary and intrapulpal anesthesia with 3% prilocain and 3% with felypressin 0.03 UI/mL because the tooth was located in the region of the vascular malformation, and any surgical treatment could lead to hemorrhages. Then, the roots were prepared, irrigated with sodium hypochlorite solution (Dakin’s fluid), dressed with formocresol, and the cavity was provisionally cemented with zinc oxide-eugenol without any posterior complications. After 7 days, the patient was recalled, and the absence of painful symptoms was observed. Afterward, the patient was forwarded to final endodontic treatment.
DISCUSSION Sturge-Weber syndrome is characterized by the presence of facial capillary malformation—facial PWS—and is generally unilateral and reaches 1 or more branches of the trigeminal nerve. The PWS is the most frequent clinical manifestation despite being mandatory for diagnoses of this syndrome.8 In the present case report, the patient showed classical characteristics of SWS which include facial PWS in the right side and the
FIGURE 2. Presence of prosthetic valve in the brain.
e305
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
Brief Clinical Studies
presence of a prosthetic valve in the brain because of a previous hydrocephalus episode (Figs. 1 and 2). Volumetric increase and vascular alterations in jugal mucosa, buccal vestibule, oral floor, and gingival hyperplasia with discreet vascular hyperplasia were also observed. Those clinical manifestations are in accordance with other studies.6,9 The dental treatment of those patients includes a complete anamnesis considering the principal complaint, the history of actual disease, medical history, systems reviewing, medications intake, and the investigation of previous bleeding in the region of the lesion(s).6 Additionally, intraoral and extraoral examinations evaluating the treatments required (restorative, surgical, periodontal, endodontic, and other treatments) associated with image examinations (computed tomography scan, periapical, and panoramic radiographies) are essential for SWS diagnosis and treatment planning.6 Also, the hemostasis of patients with SWS can be a significant problem when a surgery is planned. To the authors’ best knowledge, there are few studies reporting the risk of bleeding in patients with required endodontic treatments. Some authors reported a case report of a patient with PWS and an endodontically affected central incisor (tooth 21).6 These authors conducted a conventional endodontic treatment with satisfactory clinical outcomes, showing that the endodontic treatment is a feasible alternative in comparison to dental exodontias, because the surgery in these patients requires hospitalization and a multidisciplinary team prepared for their treatment.10
CONCLUSIONS Within the limitations of the present case report, it might be concluded that clinicians must be attentive to the presence of vascular malformations in the head and neck region and also have the expertise to perform emergency treatments in patients with those conditions. The endodontic treatment in patients with SWS is feasible and safe, and it might be an alternative to surgical treatments because patients may experience hemorrhages or undergo treatments that require multidisciplinary care and hospitalization.
REFERENCES 1. Comi AM. Presentation, diagnosis, pathophysiology, and treatment of the neurological features of Sturge-Weber syndrome. Neurologist 2011;17:179 2. Neville BW, Damm DD, Allen CM, et al. Tumors of soft tissues: Oral and maxillofacial pathology. Rio de Janeiro: Guanabara Koogan 2004 3. Alkonyi B, Chugani HT, Karia S, et al. Clinical outcomes in bilateral Sturge-Weber syndrome. Pediatr Neurol 2011;44:443 4. Piram M, Lorette G, Sirinelli D, et al. Sturge-Weber syndrome in patients with facial port-wine stain. Pediatr Dermatol 2012;29:32 5. Gontijo B, Pereira LB, Silva CMR. Malformac¸o˜es vasculares. An Bras Dermatol 2004;79:7 6. Suprabha BS, Baliga M. Total oral rehabilitation in patient with portwine stains. J Indian Pedod Prev Dent 2005;23:99 7. Caiazzo A, Mehra P, Papageorge MB. The use of preoperative percutaneous transcatheter vascular occlusive therapy in the management of Sturge-Weber syndrome: report of a case. J Oral Maxillofac Surg 1998;56:775 8. Zhou Q, Yang XJ, Zheng JW, et al. Hemangioma concurrent with arteriovenous malformation in oral and maxillofacial region: report of a case and review of the literature. J Oral Maxillofac Surg 2011;69:1100 9. De Benedittis M, Petruzzi M, Pastore L, et al. YAG laser for gingivectomy in Sturge-Weber syndrome. J Oral Maxillofac Surg 2007;65:314 10. Montegomery S. Endodontic treatment involving intraoral nevus flammeus. J Endod 1979;5:63
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Volume 26, Number 4, June 2015
Simple Repair of a Blow-Out Fracture by the Modified Caldwell–Luc Approach Min Woo Park, DDS, MSD, Soung Min Kim, DDS, MSD, Emmanuel Kofi Amponsah, DDS, PhD,y and Suk Keun Lee, DDS, MSDz Abstract: Here we report a patient with a blow-out fracture of the orbital floor that was treated by an intraoral transmaxillary approach. This 38-year-old man suffered a sudden blow to the periorbital area, which caused prolapse of the orbital contents into the maxillary sinus. The modified Caldwell–Luc approach was used to repair the orbital blow-out fracture and the maxillary sinus during was packed with Frazin gauze for 7 days to prevent recurrence of the prolapse. This was an easy and minimally invasive technique for the management of a blow-out fracture of the orbital floor. Key Words: Blow-out fracture, modified Caldwell–Luc approach
INTRODUCTION
O
rbital floor blow-out fractures often result in prolapse of the orbital contents into the maxillary sinus.1 Although the first report of a blow-out fracture was in 1957,2 controversy remains regarding methods for surgical treatment, including the best approach to the orbit. Traditionally, a transorbital approach to the orbital floor via various lower eyelid incisions has been used in the field of plastic surgery.3 However, subciliary and transconjunctival approaches have high incidences of cosmetic complications.4 A transmaxillary approach to the orbital floor was first reported in the 1972 by Walter5 and became well-established, thanks to advances in endoscopic sinus surgery.6 However, controversy remains concerning the method for support of the orbital floor after elevation of the orbital contents. At the time of this report we use Frazin gauze packing to support the orbital floor for 7 days; this technique is simple and minimally invasive.
CASE REPORT A 38-year-old man fell and hit his face on the floor and visited the department of oral and maxillofacial surgery with ecchymosis of the From the Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Korea; yDepartment of Oral and Maxillofacial Surgery, Brong Ahafo Regional Hospital, Sunyani, Ghana; and zDepartment of Oral Pathology, College of Dentistry, Gangneung-Wonju National University, Gangneung, Korea. Received December 18, 2014. Accepted for publication January 29, 2015. Address correspondence and reprint requests to Dr Soung Min Kim, DDS, MSD, Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, 101 Daehak-ro, Jongno-gu, Seoul 110-768, Korea; E-mail: [email protected] This research was supported by the International Research & Development Program of the National Research Foundation of Korea (NRF) funded by the Ministry of Science, ICT & Future Plannig (Grant number: 2014K1A3A9A01033785) The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001698 #
2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Volume 26, Number 4, June 2015
9. Hagberg H, Lamberg K, Astro¨m G. Alpha-2b interferon and oral clodronate for Gorham’s disease. Lancet 1997;350:1822–1823 10. Handl-Zeller L, Hohenberg G. Radiotherapy of Morbus Gorham-Stout: the biological value of low irradiation dose. Br J Radiol 1990;63: 206–208
Emergency Dental Treatment of a Patient With Sturge-Weber Syndrome Maria Cecı´lia Querido de Oliveira, DDS, Vanessa Nobre Maia, DDS, MSc, PhD, Juliana Bertoldi Franco, DDS, and Maria Paula Siqueira de Melo Peres, DDS Abstract: The Sturge-Weber (SWS) syndrome is a rare condition with congenital capillary malformations. Hemorrhages may occur on dental treatment, which can have a dramatic effect on a patient’s vital sign. The aim of the present brief clinical study was to briefly report a case of a female patient with SWS who underwent an endodontic treatment. A 25-year-old patient (C.O.B.S.) with SWS and vascular malformation in cervicofacial right region was admitted to the dental clinic with pulsatile pain in tooth 16. An emergency dental treatment with cavity preparation and access to root canals was performed with intraligamentary and intrapulpal anesthesia with 3% prilocain 3% with felypressin 0.03 UI/mL because the tooth was located in the region of the vascular malformation, and any surgical treatment could lead to hemorrhages. After 7 days, the patient was recalled with the absence of painful symptoms. The endodontic treatment in patients with SWS is feasible, and safety should be an alternative to surgical treatments. Key Words: Endodontics, hydrocephalus, maxillofacial abnormalities
S
turge-Weber syndrome (SWS), also called encephalotrigeminal angiomatosis, is a congenital capillary malformation, which is usually unilateral and reaches 1 or more branches of the trigeminal nerve.1– 4 Also, SWS has cerebral involvement characterized by seizures and ophthalmologic abnormalities manifested by glaucoma and capillary malformations of the conjunctiva, episcleral, coronary, and retina.1 –4 Sturge-Weber syndrome is a rare condition with approximate prevalence of 1:50,000 born alive. The facial port-wine stains From the Center Institution of Hospital Clinics, Faculty of Medicine, University of Sao Paulo, Sao Paulo, Brazil. Received November 30, 2014. Accepted for publication December 26, 2014. Address correspondence and reprint requests to Maria Cecı´lia Querido de Oliveira, DDS, Center Institution of Hospital Clinics, Faculty of ´ guiar Medicine, University of Sao Paulo, Doutor Ene´as de Carvalho A St., 225, Cerqueira Ce´sar, Sao Paulo, SP 05403–000, Brazil; E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001684 #
2015 Mutaz B. Habal, MD
Brief Clinical Studies
FIGURE 1. Facial capillary malformation (port-wine stains).
(PWS) are vascular malformations characteristics of SWS and are manifested at birth with no tendency to regression and are most commonly located in the face and cervical region.5 Also, the commitment of ophthalmic branch of trigeminal nerve is pathognomonic of SWS syndrome.6 However, this syndrome is rare, and the hemostasis of patients with SWS may be a critical procedure if a dental surgery is required, thereby it should be conducted in hospitals because those hemorrhages may have a dramatic effect on a patient’s vital sign.6,7 Thus, the aim of this study was to (1) briefly report a case of a female patient with SWS who underwent an emergency dental treatment; and (2) to describe an operative approach of SWS patients for avoiding surgical treatment.
Brief Clinical Study A 25-year-old patient (C.O.B.S.) with SWS and vascular malformation in cervicofacial right region was admitted to the Dental Division, Center Institution of Hospital Clinics, Faculty of Medicine, University of Sao Paulo with pulsatile pain in tooth 16. The patient had facial capillary malformation (PWS) and ophthalmic commitment, manifested by vascular malformations in the conjunctiva of the right eye (Fig. 1). Also, the patient reported the presence of prosthetic valve in the brain because of a hydrocephalus episode (Fig. 2). The intraoral examination showed generalized gingivitis and extensive caries in tooth 16 with occlusal and distal faces affected. The radiographic examination showed the presence of a radiolucency image extending to the root canal and an important alveolar bone loss in the distal region. An emergency dental treatment with cavity preparation and access to root canals was performed with intraligamentary and intrapulpal anesthesia with 3% prilocain and 3% with felypressin 0.03 UI/mL because the tooth was located in the region of the vascular malformation, and any surgical treatment could lead to hemorrhages. Then, the roots were prepared, irrigated with sodium hypochlorite solution (Dakin’s fluid), dressed with formocresol, and the cavity was provisionally cemented with zinc oxide-eugenol without any posterior complications. After 7 days, the patient was recalled, and the absence of painful symptoms was observed. Afterward, the patient was forwarded to final endodontic treatment.
DISCUSSION Sturge-Weber syndrome is characterized by the presence of facial capillary malformation—facial PWS—and is generally unilateral and reaches 1 or more branches of the trigeminal nerve. The PWS is the most frequent clinical manifestation despite being mandatory for diagnoses of this syndrome.8 In the present case report, the patient showed classical characteristics of SWS which include facial PWS in the right side and the
FIGURE 2. Presence of prosthetic valve in the brain.
e305
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
Brief Clinical Studies
presence of a prosthetic valve in the brain because of a previous hydrocephalus episode (Figs. 1 and 2). Volumetric increase and vascular alterations in jugal mucosa, buccal vestibule, oral floor, and gingival hyperplasia with discreet vascular hyperplasia were also observed. Those clinical manifestations are in accordance with other studies.6,9 The dental treatment of those patients includes a complete anamnesis considering the principal complaint, the history of actual disease, medical history, systems reviewing, medications intake, and the investigation of previous bleeding in the region of the lesion(s).6 Additionally, intraoral and extraoral examinations evaluating the treatments required (restorative, surgical, periodontal, endodontic, and other treatments) associated with image examinations (computed tomography scan, periapical, and panoramic radiographies) are essential for SWS diagnosis and treatment planning.6 Also, the hemostasis of patients with SWS can be a significant problem when a surgery is planned. To the authors’ best knowledge, there are few studies reporting the risk of bleeding in patients with required endodontic treatments. Some authors reported a case report of a patient with PWS and an endodontically affected central incisor (tooth 21).6 These authors conducted a conventional endodontic treatment with satisfactory clinical outcomes, showing that the endodontic treatment is a feasible alternative in comparison to dental exodontias, because the surgery in these patients requires hospitalization and a multidisciplinary team prepared for their treatment.10
CONCLUSIONS Within the limitations of the present case report, it might be concluded that clinicians must be attentive to the presence of vascular malformations in the head and neck region and also have the expertise to perform emergency treatments in patients with those conditions. The endodontic treatment in patients with SWS is feasible and safe, and it might be an alternative to surgical treatments because patients may experience hemorrhages or undergo treatments that require multidisciplinary care and hospitalization.
REFERENCES 1. Comi AM. Presentation, diagnosis, pathophysiology, and treatment of the neurological features of Sturge-Weber syndrome. Neurologist 2011;17:179 2. Neville BW, Damm DD, Allen CM, et al. Tumors of soft tissues: Oral and maxillofacial pathology. Rio de Janeiro: Guanabara Koogan 2004 3. Alkonyi B, Chugani HT, Karia S, et al. Clinical outcomes in bilateral Sturge-Weber syndrome. Pediatr Neurol 2011;44:443 4. Piram M, Lorette G, Sirinelli D, et al. Sturge-Weber syndrome in patients with facial port-wine stain. Pediatr Dermatol 2012;29:32 5. Gontijo B, Pereira LB, Silva CMR. Malformac¸o˜es vasculares. An Bras Dermatol 2004;79:7 6. Suprabha BS, Baliga M. Total oral rehabilitation in patient with portwine stains. J Indian Pedod Prev Dent 2005;23:99 7. Caiazzo A, Mehra P, Papageorge MB. The use of preoperative percutaneous transcatheter vascular occlusive therapy in the management of Sturge-Weber syndrome: report of a case. J Oral Maxillofac Surg 1998;56:775 8. Zhou Q, Yang XJ, Zheng JW, et al. Hemangioma concurrent with arteriovenous malformation in oral and maxillofacial region: report of a case and review of the literature. J Oral Maxillofac Surg 2011;69:1100 9. De Benedittis M, Petruzzi M, Pastore L, et al. YAG laser for gingivectomy in Sturge-Weber syndrome. J Oral Maxillofac Surg 2007;65:314 10. Montegomery S. Endodontic treatment involving intraoral nevus flammeus. J Endod 1979;5:63
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Volume 26, Number 4, June 2015
Simple Repair of a Blow-Out Fracture by the Modified Caldwell–Luc Approach Min Woo Park, DDS, MSD, Soung Min Kim, DDS, MSD, Emmanuel Kofi Amponsah, DDS, PhD,y and Suk Keun Lee, DDS, MSDz Abstract: Here we report a patient with a blow-out fracture of the orbital floor that was treated by an intraoral transmaxillary approach. This 38-year-old man suffered a sudden blow to the periorbital area, which caused prolapse of the orbital contents into the maxillary sinus. The modified Caldwell–Luc approach was used to repair the orbital blow-out fracture and the maxillary sinus during was packed with Frazin gauze for 7 days to prevent recurrence of the prolapse. This was an easy and minimally invasive technique for the management of a blow-out fracture of the orbital floor. Key Words: Blow-out fracture, modified Caldwell–Luc approach
INTRODUCTION
O
rbital floor blow-out fractures often result in prolapse of the orbital contents into the maxillary sinus.1 Although the first report of a blow-out fracture was in 1957,2 controversy remains regarding methods for surgical treatment, including the best approach to the orbit. Traditionally, a transorbital approach to the orbital floor via various lower eyelid incisions has been used in the field of plastic surgery.3 However, subciliary and transconjunctival approaches have high incidences of cosmetic complications.4 A transmaxillary approach to the orbital floor was first reported in the 1972 by Walter5 and became well-established, thanks to advances in endoscopic sinus surgery.6 However, controversy remains concerning the method for support of the orbital floor after elevation of the orbital contents. At the time of this report we use Frazin gauze packing to support the orbital floor for 7 days; this technique is simple and minimally invasive.
CASE REPORT A 38-year-old man fell and hit his face on the floor and visited the department of oral and maxillofacial surgery with ecchymosis of the From the Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Korea; yDepartment of Oral and Maxillofacial Surgery, Brong Ahafo Regional Hospital, Sunyani, Ghana; and zDepartment of Oral Pathology, College of Dentistry, Gangneung-Wonju National University, Gangneung, Korea. Received December 18, 2014. Accepted for publication January 29, 2015. Address correspondence and reprint requests to Dr Soung Min Kim, DDS, MSD, Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, 101 Daehak-ro, Jongno-gu, Seoul 110-768, Korea; E-mail: [email protected] This research was supported by the International Research & Development Program of the National Research Foundation of Korea (NRF) funded by the Ministry of Science, ICT & Future Plannig (Grant number: 2014K1A3A9A01033785) The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001698 #
2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
