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Int J Surg Case Rep. 2018;43:21-24. doi: 10.1016/j.ijscr.2018.01.007. Epub 2018 Feb 4.

Intraorbital hemorrhage following a secondary intervention at integrated zygomatic implants: A case report. Van Camp P1, Vrielinck L2, Gemels B3, Politis C4.

Author information 1 Department of OMFS St-John's Hospital, Genk, Belgium. Electronic address: [email protected]. 2 Department of OMFS St-John's Hospital, Genk, Belgium. Electronic address: [email protected]. 3 Department of OMFS University Hospitals, Leuven, Belgium. Electronic address: [email protected]. 4 Department of OMFS University Hospitals, Leuven, Belgium. Electronic address: [email protected].

Abstract INTRODUCTION: Zygomatic implant placement can be the best option for restoring masticatory function of an extremely atrophic upper jaw, but the procedure is more invasive than conventional implant placement and can be associated with complications. PRESENTATION OF CASE: We report a complication that occurred during a secondary corrective surgical procedure four years after zygomatic implant placement. The patient was a 54-year-old female who had been edentulous for 25 years. Four zygomatic implants were placed. Subsequent prosthetic rehabilitation was successful. Four years later, the patient complained of discomfort. It was found that the tips of the implants on the right side were subcutaneously palpable and surrounded by granulomatous tissue. Intraoral surgery was performed to remove the protruding tips of the two implants. Post-operatively, the patient developed severe orbital pain on the right side with proptosis and diffuse swelling of the eyelids. Emergency surgery was performed to drain the intraorbital hemorrhage. The patient healed uneventfully without loss of visual acuity.

DISCUSSION: Scarce prior reports describe trauma to the orbit during zygomatic implant surgery, mostly involving orbital penetration during zygoma implant placement. To our knowledge, the present case report is the first to describe an intraorbital hemorrhage that led to an orbital compression syndrome necessitating emergency surgery. CONCLUSION: In our case, corrective surgery in a patient with zygomatic implants resulted in an intraorbital hemorrhage, followed by an orbital compression syndrome. Emergency surgery was immediately performed, allowing hematoma drainage and eliminating compression of the intraorbital content. Symptoms quickly resolved and eyesight was not compromised. Copyright © 2018 The Author(s). Published by Elsevier Ltd.. All rights reserved. KEYWORDS: Complication; Intraorbital hemorrhage; Zygomatic implant PMID: 29414502 DOI: 10.1016/j.ijscr.2018.01.007

Intraorbital hemorrhage following a secondary intervention at integrated zygomatic implants Authors Van Camp Philippe (1) , Vrielinck Luc (2) , Gemels Bert (3), Agbaje Jimoh (4), Politis Constantinus (5) (1) MD, DDS, Resident OMFS St-John’s Hospital Genk (Belgium), [email protected] (2) MD, DDS, Head of the department of OMFS St-John’s Hospital Genk (Belgium), [email protected] (3) MD, DDS, Resident OMFS University Hospitals Leuven (Belgium), [email protected] (4) BDS, DMD, FMCDS, MMI, PhD [email protected] (5) MD, DDS, MHA, MM, PhD, Head of the department of OMFS University Hospitals Leuven (Belgium), [email protected]

Correspondence to: Philippe Van Camp Nieuwelaan 52 2520 Oelegem BELGIUM Tel: +32479353595 [email protected]

Intraorbital hemorrhage following a secondary intervention at integrated zygomatic implants ABSTRACT This case report describes a complication occurring during a secondary corrective surgical procedure, 4 years after placement of zygomatic implants. Four zygomatic implants were placed in a 54 year old female, who was edentulous for 25 years. Prosthetic rehabilitation was successful. 4 years later, she developed complaints: the tips of the implants on the right side, surrounded by granulomatous tissue, were palpable subcutaneously. Intraoral surgery was performed to remove the protruding tips of the two implants. Post-operatively, the patient developed severe orbital pain on the right side with proptosis and diffuse swelling of the eyelids. Emergency surgery ensued to drain the intraorbital hemorrhage. The patient healed uneventfully without loss of visual acquity.

KEYWORDS: Complication, zygomatic implant, intraorbital hemorrhage

INTRODUCTION The use of zygomatic implants may offer an additional treatment option to restore masticatory function of the extremly atrophic upper jaw and may also improve the facial appearance. Zygomatic implants may serve as a valid alternative to sinus augmentations or onlay bone grafts, followed by dental implant placement. Although the use of zygomatic implants is a more invasive procedure than the placement of conventional dental implants, this treatment option may be patient’s last resort or even be the better option when bone reconstructive options are contra-indicated or unwanted by the patient (1) (2). The success rate of zygomatic implants is high with a reported cumulative survival rate of 96.3% - 100% (1) (3) (2) (4) (5). Several complications have been described in the literature on the use of this specific type of implant such as: persistent infection of the maxillary sinus (up to 21.4% (3) (2) (4) (5)), buccosinusal fistula, infection around the implants, chronic gingivitis and damage to the infraorbital nerve (2) (6) (5). Penetration of the implant in the nasal cavity or even intracerebral penetration have also been noted (7) (1) (3). This case report describes an intraorbital hemorrhage following surgery to correct the protruding tips of zygomatic implants, 4 years after initial placement.

CASE REPORT A 54 year old Caucasian female presented at the Department of Oral and Maxillofacial Surgery at St-John’s Hospital (Genk, Belgium). The patient was healthy, did not smoke nor consume large amounts of alcohol. She only suffered from hypothyroidism; the only medication she took was levothyroxine 125µg once daily. The patient had been edentulous in the upper jaw for over 25 years. Over the years, several removable prostheses had been fabricated, but due to severe atrophy of the jaw, there was a severe lack of retention. To improve stability and function, it was decided to place 4 zygomatic implants. Simultaneously, teeth in the lower jaw were removed due to advanced periodontitis and a bone graft procedure was performed in the mandible using corticospongious bone graft from the anterior iliac crest. (Figure 1) Surgery was uneventful and 4 zygomatic fixtures were placed into the zygomatic bone. The patient was discharged from the hospital the following day. Healing went well and prosthetic rehabilitation in the upper jaw was carried out using a removable overdenture. In the lower jaw a temporary removable prosthesis was made. The overall result of the treatment was a net improvement in masticatory function and improved esthetics. Four years later, the patient returned to our department, complaining about a protrusion in the region of the zygomatic bone on the right side. This caused chronic irritation and pain in the overlying skin. On examination, it was observed that the symptoms resulted from the protruding tips of the apical part of the two zygomatic implants on the right side, surrounded by chronic granulomatous tissues. (Figure 2) It was decided to perform an exploration of the area and shorten these zygomatic implants under general anesthesia on an outpatient basis. Although a difficult procedure owing to the poor visualization of the region, surgery went as expected. We observed that by now (4 years later) the implants protruded a few millimeters (3-5 mm) past the outer border of the zygoma. (Figure 3) Most likely, a very low grade infection at the tips caused resorption of the zygomatic bone. Using a round drill of 3 mm diameter, the overhang of both implants was mechanically removed, until no more irregularities could be observed. After copious rinsing of the operating field, the wound was closed using resorbable sutures. Patient recovered uneventfully from the anesthesia and was transported to recovery room. About half an hour later, the patient developed severe pain at the right eye and diffuse swelling of the upper and lower eyelid. (Figures 4 and 5) Clinical inspection of the eye and extensive evaluation of eye sight was not possible due to the severity of swelling, but a proptosis of the eyeball could be observed. Differentiation between light and dark however was still present.

The most probable clinical explanation was a bleed in the orbit, although at that moment the precise cause was unknown. The patient was prepped for emergency surgery to decompress the orbital content. No CT-scan was issued at that time. No ophtalmologist was readily available for an emergency consult. General anesthesia was performed. A lower eyelid incision was used to gain entry to the right orbit. After identifying the lower orbital rim, the orbital floor was explored. During this procedure, it was observed that there was active drainage of blood from within the orbit. (Figure 6) When spontaneous draining was completed, 2 Penrose drains were inserted and left in place: one on the orbital floor and one on the lateral side of the zygoma. (Figure 7) Corticosteroids (methylprednisolone 40mg 4 times a day) were administered. After an ophtalmological consult several hours later, acetazolamide 500mg was added. Subsequently, the patient was admitted to the ward. After the procedure, eye sight was closely monitored. The next day, swelling had diminished. The patient could open her eyes spontaneously and see colours. She did complain of blurry vision and a mild degree of diplopia. A CT was performed. No obvious bony defects were noted in the protocol. However, on close examination of the CT images, a small bony defect can be visualized, precisely in the region where the implants tips were removed. (Figures 8 and 9) The next day, in absence of any sign of bleeding, the penrose drains were removed and patient was discharged from hospital. Also at follow-up consultation, no complaints of dimished eye sight were recorded; skin healing was uneventful. (Figure 10)

DISCUSSION This case report describes a very unusual complication following corrective surgery after zygomatic implants placement. Four years following routine zygomatic implant placement, the zygomatic bone on the right side had developed resorption around the tips of the implants in such a manner that they protruded subcutaneously causing complaints. Corrective surgery (shortening of the implants) caused minimal trauma to the orbit which led to the manifestation of an intraorbital hemorrhage. Subsequently an acute orbital compartment syndrome developed. Shortly after the procedure, the patient reported excruciating pain, with extensive swelling and protrusion of the eyeball. Emergency surgery was immediately performed: through an infra-orbital incision, entry was gained to the orbit, allowing drainage of the hematoma and eliminating the compression of the intraorbital content. Symptoms resolved quickly and eye sight was not compromised. However scarce, there has been some mention of trauma to the orbit during zygomatic implant surgery in the literature, mostly based on orbital penetration during the phase of

zygoma implant placement (4) (8). This case report is the first one to our knowledge, to describe an intraorbital hemorrhage leading to an orbital compression syndrome necessitating emergency surgery. The course of action taken upon the discovery of the complication was in accordance with the article of Brucoli et al (9) . One may remark that no radiographic evaluation was made between diagnosis of the complication and the time of emergency surgery. This is due to the clear link between the original surgery and the complication. Also, CT imaging would have caused time delay and would not have changed the course of action that was undertaken to resolve the issue at hand. In this particular case, the surgeon opted for an infra-orbital approach (instead of a lateral canthothomy) because he wanted to explore the entire anterior region of the orbital floor and lateral orbit since the exact source of the bleed was not known. CONCLUSION To our knowledge, this is the first description of an intraorbital hemorrhage leading to an orbital compression syndrome, within hours after corrective zygomatic implant surgery. Emergency surgery was performed to relief intraorbital pressure leading to a full recovery of patient’s eye sight.

ACKNOWLEDGMENTS None

CONFLICT OF INTEREST The authors declare no conflict of interest.

REFERENCES 1. Chrcanovic B, Abreu M. Survival and complications of zygomatic implants: a systematic review. Oral Maxillofac Surg. 2013; 17: p. 81-93. 2. Candel-Marti E, Carrillo-Garcia C, Penarrocha-Oltra D, Penarrocha-Diago M. Rehabilitation of atrophic posterior maxilla with zygomatic implants: review. J Oral Implantol. 2012; 5: p. 653-657. 3. Araujo R, Sverzut A, Tivellato A, Sverzut C. Retrospective analysis of 129 consecutive zygomatic implants used to rehabilitate severely resorbed maxillae in a two-stage protocol. Int J Oral Maxillofac Implants. 2017; 32: p. 377-384. 4. Davo R, Pons O, Rojas J, Carpio E. Immediate function of four zygomatic implants: a 1-year report of a prospective study. Eur J Oral Implantol. 2010; 3: p. 323-334. 5. Kahnberg KE, Henry P, Hirsch JM, Öhrnell LO, Andreasson L, Branemark PI, et al. Clinical evaluation of the zygoma implant: 3-year follow-up at 16 clinics. J Oral Maxillofac Surg. 2007; 65: p. 2033-2038. 6. Fernandez H, Gomez-Delgado A, Trujillo-Saldarriaga S, Varon-Cardona D, Castro-Nunez J. Zygomatic implants for the management of the severely atrophied maxilla: a retrospective analysis of 244 implants. J Oral Maxillofac Surg. 2014; 72: p. 887-891. 7. Reychler H, Olszewski R. Intracerebral penetration of a zygomatic dental implant and consequent therapeutic dilemmas: case report. Int J Oral Maxillofac Implants. 2010; 25: p. 416-418. 8. Duarte L, Filho H, Francischone C, Peredo L, Branemark PI. The establishment of a protocal for the total rehabilitation of atrophic maxillae employing four zygomatic fixtures in an immediate loading system - a 30-month clinical and radiographical follow-up. Clin Implant Dent Relat Res. 2007; 9: p. 186-196. 9. Brucoli M, Arcur F, Giarda M, Benech R , Benech A. Surgical management of posttraumatic intraorbital hematoma. J Craniofac Surg. 2012; 23: p. e58-e61. 10. Rodriguez-Chessa J, Netto H, Shibli J, de Moraes M, Mazzonetto R. Treatment of atrophic maxilla with zygomatic implants in 29 consecutive patients. Int J Clin Exp Med. 2014; 7: p. 426-430.

FIGURES CAPTIONS 1. Panoramic radiograph demonstrating placement of four zygomatic implants and onlay graft. 2. Clinical photograph showing the protrusion at the zygoma on the right side. 3. Clinical photograph taken per-operatively, showing the protrusion of the tips of the zygomatic implants. 4. Extensive swelling of the eyelid as well as proptosis, lateral view. 5. Extensive swelling of the eyelid as well as proptosis frontal view. 6. Access to the orbit using an infraorbital approach. 7. Status after emergency surgery with 2 drains in place. 8. CT 1 day post-operatively showing very close relation of the tip of the zygomatic implant with the lateral orbit, coronal view. 9. CT 1 day post-operatively showing very close relation of the tip of the zygomatic implant with the lateral orbit, sagittal view. 10. Clinical photograph, 2 years after surgery.

Complication after zygomatic implants placement: Intraorbital hemorrhage Authors

Van Camp P, MD, DDS, Resident OMFS Vrielinck Luc, MD, DDS, Head of the department of OMFS St-John’s Hospital Genk (Belgium) Gemels B, MD, DDS, Resident OMFS Jimoh Agbaje, BDS, DMD, FMCDS, MMI, PhD

Constantinus Politis, MD, DDS, MHA, MM, PhD

Correspondence to: Philippe Van Camp Nieuwelaan 52, 2520 Oelegem BELGIUM Tel: +32479353595 [email protected]

Acknowledments: none

Funding: none

ABSTRACT

In this case report, we elaborate on a complication after corrective surgery 4 years after placement of zygomatic implants. Four zygomatic implants were placed in a 54 year old female, who was edentulous for 25 years. Prosthetic rehabilitation was successful. 4 years later, she developed complaint from a protrusion at the zygomatic bone on the right side. The tips of the implants were palpable. Surgery was done to remove the tips of the two implants. Post-operatively, the patient developed severe orbital pain on the right side with proptosis and diffuse swelling of the eyelids. Emergency surgery ensued to drain the intraorbital hemorrhage. The patient healed uneventfully without vision disturbance.

KEY WORDS: Complication, zygomatic implant, Intraorbital hemorrhage

Introduction When a patient suffers from extensive atrophy in the upper jaw, the use of zygomatic implants may offer an additional treatment option to restore masticatory function of the patient and may also improve the facial appearance. . Although the use of zygomatic implants is a more invasive procedure than the placement of conventional dental implants, this treatment option may be patient’s last resort. (1) (2)The success rate of zygomatic implants is high with a reported cumulative survival rate of 96.3% - 100% (1) (3) (2) (4) (5). Zygomatic implants may serve as a valid alternative to sinus augmentations or onlay bone grafts. Several complications have been described in the literature on the use of zygomatic implants such as: persistent infection of the maxillary sinus (up to 21.4% (3) (2) (4) (5)), buccosinusal fistula, infection around the implants, chronic gingivitis and damage to the infraorbital nerve. (2) (6) (5)Penetration of the implant in the nasal cavity or even intracerebral penetration have also been noted. (7) (1) (3)This case report describes an intraorbital hemorrhage following surgery to correct the protruding tips of zygomatic implants, 4 years after initial placement.

Case report A 54 year old Caucasian female presented at the Department of Oral and Maxillofacial Surgery at StJohn’s Hospital (Genk, Belgium). The patient was healthy, did not smoke nor consume large amounts of alcohol. She only suffered from hypothyroidism; the only medication she took was levothyroxine 125µg once daily. The patient had been edentulous in the upper jaw for over 25 years. Over the years, several removable prostheses had been fabricated, but due to severe atrophy of the jaw, there was a severe lack of retention. To improve stability and function, it was decided to place 4 zygomatic implants. Simultaneously, teeth in the lower jaw were removed due to advanced periodontitis and a bone graft procedure was performed in the mandible using corticospongious bone graft from the iliac crest. The surgery went according to plan, 4 zygomatic fixtures were placed into the zygomatic bone. After one night in hospital, the patient was discharged. Healing went well and prosthetic rehabilitation in the upper jaw was carried out using a fixed removable overdenture. In the lower jaw a temporary removable prosthesis was made. The overall result of the treatment was a net improvement in masticatory function and improved esthetics. Four years later, the patient returned to our the department and complained about a protrusion in the region of the zygomatic bone on the right side. This caused chronic irritation and pain in the overlying skin. On examination, it was observed that the symptoms resulted from the protruding tips of the two zygomatic implants on the right side. It was decided to perform an exploration of the area and shorten these zygomatic implants under general anesthesia on an outpatient basis. Although a difficult procedure owing to the poor visualization of the region, surgery went as expected. We observed that by now (4 years later) the posterior implant protruded a few millimeters (3-5 mm). Using a round drill of 3 mm diameter, the overhang of both implants was mechanically removed, until no more irregularities could be

observed. After copious rinsing of the operating field, the wound was closed using resorbable sutures. Patient recovered uneventfully from the anesthesia and was transported to recovery room. About half an hour later, the patient developed severe pain at the right eye and diffuse swelling of the upper and lower eyelid. Clinical inspection of the eye and extensive evaluation of eye sight was not possible due to the severity of swelling, but a proptosis of the eyeball could be observed. Differentiation between light and dark however was still present. . Most probable because was a bleeding in the orbit causing compression: intraorbital hemorrhage. The most probable clinical explanation was a bleeding introrbitally although at that moment the precise cause of it was unknown.The patient was prepped for emergency surgery to decompress the orbital content. General anesthesia was performed. A lower eyelid incision was used to gain entry to the right orbit. After identifying the lower orbital rim, the orbital floor was explored. During this procedure, it was observed that there was active drainage of blood from within the orbit. When spontaneous draining was completed, 2 Penrose drains were inserted and left in place: one on the orbital floor and one on the lateral side of the zygoma. Corticosteroids (methylprednisolone 40mg 4 times a day) were administered. After an ophtalmological consult, acetazolamide 500mg was prescribed. Subsequently, the patient was admitted to the ward. After the procedure, eye sight was closely monitored. The next day, swelling had diminished. The patient could open her eyes spontaneously and see colours. She did complain of blurry vision and a mild degree of diplopia. A CT was performed which demonstrated a small remaining hematoma just above the orbital floor on the lateral side. No obvious bony defects were noted in the protocol. However, on close examination of the CT images, a small bone defect can be visualized, precisely in the region where the implants tips were removed. The next day, in absence of any sign of bleeding, the penrose drains were removed and patient was discharged from hospital. Also at follow-up consultation, no complaints of dimished eye sight were recorded. Discussion This case report describes a very unusual complication following corrective surgery afterzygomatic implants placement. Four years following routine zygomatic implant placement, the zygomatic bone on the right side had developed resorption around the tips of the implants in such a manner that they protruded subcutaneously causing complaints. Corrective surgery (shortening of the implants) caused minimal trauma to the orbit which led to the manifestation of an intraorbital hemorrhage. Subsequently an acute orbital compartment syndrome developed. Shortly after the procedure, the patient reported excruciating pain, with extensive swelling and protrusion of the eyeball. Swift and deliberate action is necessary. Emergency surgery was immediately performed: through an infra-orbital incision, entry was gained to the orbit, allowing drainage of the hematoma and eliminating the compression of the intraorbital content. Symptoms resolved quickly and eye sight was not compromised.. However scarce, there has been some mention of trauma to the orbit during zygomatic implant surgery in the literature, mostly based on surgical errors during the phase of zygoma implant

placement. (4) (8)This case report on the other hand, is the first one to our knowledge,to describe an intraorbital hemorrhage leading to an orbital compression syndrome necessitating emergency surgery. The course of action taken upon the discovery of the complication was in accordance with the article of Brucoli et al. (9) One may remark that no radiographic evaluation was made between diagnosis of the complication and the time of emergency surgery. This is of course is due to the clear link between the original surgery and the complication. Also, CT imaging would have caused time delay and would not have changed the course of action that was undertaken to resolve the issue at hand. In this particular case, the surgeon opted for an infra-orbital approach (instead of a lateral canthothomy) because he wanted to explore the entire anterior region of the orbital floor and lateral orbit. Conclusion To our knowledge, this is the first description of an intraorbital hemorrhage leading to an orbital compression syndrome, hours after corrective zygomatic implant surgery. Emergency surgery was necessary to relief intraorbital pressure threatening the patient’s eye sight. This case report clearly demonstrates the importance of thorough knowledge of the anatomy of the region during zygomatic implant surgery. It should be reserved for clinicians with extensive training. When a complication like this one should occur, swift and deliberate action must be taken.

1. Chrcanovic B, Abreu M. Survival and complications of zygomatic implants: a systematic review. Oral Maxillofac Surg. 2013; 17: p. 81-93. 2. Candel-Marti E, Carrillo-Garcia C, Penarrocha-Oltra D, Penarrocha-Diago M. Rehabilitation of atrophic posterior maxilla with zygomatic implants: review. J Oral Implantol. 2012; 5: p. 653-657. 3. Araujo R, Sverzut A, Tivellato A, Sverzut C. Retrospective analysis of 129 consecutive zygomatic implants used to rehabilitate severely resorbed maxillae in a two-stage protocol. Int J Oral Maxillofac Implants. 2017; 32: p. 377-384. 4. Davo R, Pons O, Rojas J, Carpio E. Immediate function of four zygomatic implants: a 1-year report of a prospective study. Eur J Oral Implantol. 2010; 3: p. 323-334. 5. Kahnberg KE, Henry P, Hirsch JM, Öhrnell LO, Andreasson L, Branemark PI, et al. Clinical evaluation of the zygoma implant: 3-year follow-up at 16 clinics. J Oral Maxillofac Surg. 2007; 65: p. 2033-2038. 6. Fernandez H, Gomez-Delgado A, Trujillo-Saldarriaga S, Varon-Cardona D, Castro-Nunez J. Zygomatic implants for the management of the severely atrophied maxilla: a retrospective analysis of 244 implants. J Oral Maxillofac Surg. 2014; 72: p. 887-891. 7. Reychler H, Olszewski R. Intracerebral penetration of a zygomatic dental implant and consequent therapeutic dilemmas: case report. Int J Oral Maxillofac Implants. 2010; 25: p. 416-418. 8. Duarte L, Filho H, Francischone C, Peredo L, Branemark PI. The establishment of a protocal for the total rehabilitation of atrophic maxillae employing four zygomatic fixtures in an immediate loading system - a 30-month clinical and radiographical follow-up. Clin Implant Dent Relat Res. 2007; 9: p. 186-196. 9. Brucoli M, Arcur F, Giarda M, Benech R , Benech A. Surgical management of posttraumatic intraorbital hematoma. J Craniofac Surg. 2012; 23: p. e58-e61. 10. Rodriguez-Chessa J, Netto H, Shibli J, de Moraes M, Mazzonetto R. Treatment of atrophic maxilla with zygomatic implants in 29 consecutive patients. Int J Clin Exp Med. 2014; 7: p. 426-430.