Q U I N T E S S E N C E I N T E R N AT I O N A L
IMPLANTOLOGY
Luis Naval
Dental implants and osteomyelitis in a patient with osteopetrosis Luis Naval, DDS, PhD, DMD1/Manuel Sanchez Molini2/Gonzalo Herrera 2/Beatriz Naval, DDS, MSc 3 Osteopetrosis (OP) is a rare genetic metabolic bone disorder characterized by osteoclast dysfunction impairing bone resorption. Together with normal osteoblastic activity this dysfunction promotes intense bone sclerosis with reduction of marrow spaces as well as bone turnover. Maxillofacial complications are common after tooth extraction. Osteomyelitis (OM) of the mandible occurs in around 10% of the cases with OP. The pur-
pose of this paper is to report the first case of OP treated with dental implants, and to describe the protocol used to treat OM that developed after failure of one implant. The treatment of OM included long-term use of antibiotics and marginal bone resection with a piezoelectric device. Similar cases to confirm the efficacy of this treatment are required. (Quintessence Int 2014;45:765–768; doi: 10.3290/j.qi.a32443)
Key words: bone resorption, dental implants, osteomyelitis, osteopetrosis
Osteopetrosis (OP) is also known as Albers-Schönberg disease, generalized congenital osteosclerosis, ivory bones, marble bone disease, or osteosclerosis fragilis generalisata. It is classified into two main phenotypes: the infantile (malignant) and adult (benign) forms. These OP phenotypes are divided into three types: • Benign or adult OP is an autosomal dominant form with no bone marrow compromise and a good vital prognosis. The serum acid phosphatase is increased because of augmented release by defective osteoclasts.1 • Malignant or infantile OP is autosomal recessive, and has severe bone marrow involvement and poor vital prognosis. It is usually diagnosed before the age of 1 year.2 1
Head, Department of Oral and Maxillofacial Surgery, University Clinic of Navarra, Pamplona, Spain; and Associate Professor, University of Navarra, Pamplona, Spain.
2
Staff Member, Department of Oral and Maxillofacial Surgery, University of Navarra, Pamplona, Spain.
3
Clinician, Private Practice, Clínica Naval, Madrid, Spain.
Correspondence: Dr Beatriz Naval, Clínica Naval, C/ Alcántara 71, 28006 Madrid, Spain. Email: [email protected]
VOLUME 45 • NUMBER 9 • OCTOBER 2014
•
Intermediate OP is an autosomal recessive form with no bone marrow compromise and poor vital prognosis. Bone marrow transplantation must be considered as a treatment option.2
Depending on the severity of the OP, the following signs or symptoms can appear: bone pain, frequent fractures of the long bones, osteomyelitis, nerve compression leading to headache, blindness, deafness, pancytopenia with enlarged spleen, and stroke.1,3 Adult OP can often be asymptomatic for a long period of time, and it has a normal life expectancy. It is characterized by a generalized increase in skeletal mass, headache, frequent bone fractures, and mandibular or maxillary osteomyelitis. Although adult OP has an autosomal dominant inheritance it can also originate because of an isolated mutation. The actual incidence is unknown. However, it is thought to be between 1 in 100,000 and 1 in 500,000. Bone biopsy is not essential for diagnosis. The diagnosis of OP is often secondary to other diseases and findings of specific
765
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
a
b
Fig 1 Panoramic radiograph after removing the failed implant (second left mandibular molar position). Bone sclerosis of the jaws is evident.
Figs 2a and 2b Skeletal radiograph: osteoclerosis of the vertebrae (a) and iliac bones (b).
radiographic features.4 Maxillofacial changes include sclerosis of the facial bones, delayed dental eruption, early tooth loss, congenitally missing or malformed teeth, and thickened lamina dura. A common complication that arises after tooth extraction is osteomyelitis of the mandible. Osteomyelitis of the maxilla is extremely rare.3-5 In the literature, most of the cases of OP with osteomyelitis presented osteomyelitis after dental extraction.6-8 In OP, hypocalcemia can occur due to elevation of the parathyroid hormone (secondary hyperparathyroidism). Furthermore, acid phosphatase can increase as well as brain-type creatine kinase (CK-BB) because of the elevated release from defective osteoclasts. The present article reports a case of adult OP complicated with mandibular osteomyelitis after dental implant treatment. It is the first reported case of OP treated with dental implants.
integration period. It was removed without incident. However, the patient had since suffered from pain and fistula with active suppuration. The soft tissue exhibited gingival swelling. Despite several antibiotic courses the clinical manifestation did not improve over a period of 6 months. The panoramic radiograph (Fig 1) showed bone depression and radiolucency at the site of the failed implant and an augmented bone density through the whole mandible, similar to those seen in patients treated with bisphosphonates. The medical records of the patient were reevaluated for bisphosphonate intake or other pathologic findings. The patient had no other oral complaints. His dental records showed only some “delays and problems” in extraction socket healing. The patient had previously undergone surgery at the Department of Orthopedic Surgery for calcifying tendonitis, both subacromial and patellar. A more extensive radiographic study clearly showed a case of OP (Figs 2 to 4). The diagnosis at this time was mandibular osteomyelitis with poor response to antibiotic therapy in a patient that suffered from adult OP after dental implant surgery. Following the protocols of the Spanish Society of Oral and Maxillofacial Surgery for osteomyelitis,9 the patient was treated with antibiotics for 4 weeks (amoxicillin/clavulonate 875/125 mg every 8 hours), and two curettages of the site (Figs 5 and 6). The second curettage was carried out under sedation and local anesthesia using a piezoelectric device until a clear, clean, and
CASE REPORT A 71-year-old man, suffering from adult OP without prior family history of the disease, came to the Clinic University of Navarra complaining of pain, oral discharge, and fistula on the left side of the mandible (failed implant site). He had been treated with one maxillary and four mandibular implants 2 years previously, in order to improve his masticatory ability. The most distal mandibular implant failed at the end of the
766
VOLUME 45 • NUMBER 9 • OCTOBER 2014
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
Fig 3 Chest radiograph showing osteoclerosis of the ribs and other bones with obliteration of cancellous bone marrow. Two old fractures can be observed on the right side.
Fig 4 Lateral radiograph showing calcifying tendinitis of the patella.
Fig 5
Fig 6 Panoramic radiograph after first curettage and antibiotic treatment for four weeks.
Intraoral aspect at the first curettage.
Fig 7 After piezoelectric scraping until a healthy looking and bleeding bone was reached. Both alveolar canals are narrow because of the osteopetrosis. The left alveolar canal is very close to the bone defect.
Fig 8 Biopsy showed necrotic bone with empty osteocytes lacunae and no bone marrow spaces (hematoxylin-eosin stain).
bleeding bone was obtained. Neither mucosal detachment nor waterproof sutures were performed. The follow-up showed uneventful development for 1 year (Fig 7).
A biopsy showed sclerotic bone without osteocytes or bone marrow spaces (Fig 8). Calcium levels were normal, but parathyroid hormone and creatine kinase (CK) levels were elevated.
VOLUME 45 • NUMBER 9 • OCTOBER 2014
767
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
DISCUSSION
CONCLUSION
The differential diagnosis that can be considered includes other sclerosing bone dysplasias, such as pycnodysostosis, craniometaphyseal dysplasia, diaphyseal dysplasia, melorheostosis, osteopoikilosis, and osteopathia striata. Fluoride poisoning and secondary hyperparathyroidism from renal osteodystrophy may also produce a diffuse osteosclerosis.8 The treatment of osteomyelitis when secondary to OP is controversial. Different treatment regimens include local hygiene, systemic antibiotics, debridement of necrotic bone, and primary closure of soft tissues, if possible. On other occasions hyperbaric oxygen has been used for the treatment of chronic osteomyelitis, especially if related with radionecrosis, but there are no references for this particular bone disease.7 The mechanism of the development of osteomyelitis in OP is the same as for osteonecrosis of the jaw (ONJ) related with drugs10,11 such as antiresorptive medications (bisphosphonates [bisphosphonaterelated ONJ, BRONJ], or denosumab): the loss of function of osteoclasts. Several protocols have been developed in order to prevent or even treat these situations of BRONJ or ONJ. Many of them contraindicated oral surgery (mainly dental extractions or bone curettage) even for the treatment of osteomyelitis itself. Above all, the use of dental implants in these patients has been often criticized or even contraindicated, leaving these patients with no alternative other than very conservative therapies, which often achieve poor results.10-12 Recent reports, however, show that some cases of osteoclast dysfunction diseases can be treated with careful oral surgery, dental extraction, and bone curettage. In carefully selected cases, explaining to the patient the potential risks, advantages, and disadvantages, dental implants can be placed.11,12 Thus the proposal of this surgical piezoelectric scraping treatment can be a surgical compliment or alternative to the failure of antibiotics or hyperbaric oxygen therapy. Some authors suggest the use of Er-YAG laser or careful debridement to remove the avascular and necrotic bone.3,4,6,7,12
This study is the first presented case of dental implants placed in a patient with OP. Circumscribed osteomyelitis of one of the sites was observed after one mandibular implant failure. Careful case selection and consideration of this type of bone pathology must be taken into account before implant placement. The surgical protocol to treat the osteomyelitis in cases with osteoclast dysfunction disease (OP) was described. Nevertheless, more cases must be reported before this treatment can be recommended.
768
REFERENCES 1. de Oliveira Hdo C, Pereira Filho VA, Gabrielli MF, Gabrielli MA, Vieira EH. Marginal resection for treatment of mandibular osteomyelitis associated with osteopetrosis: case report. J Craniomaxillofac Surg 2011;39:525–529. 2. Yamada T, Mishima K, Imura H, et al. Osteomyelitis of the mandible secondary to infantile osteopetrosis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e25–e29. 3. Öğütcen-Toller M, Tek M, Sener I, Bereket C, Inal S, Özden B. Intractable bimaxillary osteomyelitis in osteopetrosis: review of the literature and current therapy. J Oral Maxillofac Surg 2010;68:167–175. 4. Krithika C, Neelakandan RS, Sivapathasundaram B, Koteeswaran D, Rajaram PC, Shetkar GS. Osteopetrosis-associated osteomyelitis of the jaws: a report of 4 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e56–e65. 5. Barry CP, Ryan CD, Stassen LFA. Osteomyelitis of the maxilla secondary to osteopetrosis: a report of 2 cases in sisters. J Oral Maxillofac Surg 2007;65:144–147. 6. Albuquerque MA, Melo ES, Jorge WA, Cavalcanti MG. Osteomyelitis of the mandible associated with autosomal dominant osteopetrosis: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:94–98. 7. García CM, Garcıa M A, Garcıa RG, Gil FM. Osteomyelitis of the mandible in a patient with osteopetrosis. case report and review of the literature. J Maxillofac Oral Surg 2013;12:94–99. 8. Kant P, Sharda N, Bhowate RR. Clinical and radiological findings of autosomal dominant osteopetrosis type II: a case report. Case Rep Dent 2013;2013:707343. 9. García Marín F (ed). Protocolos Clínicos de la Sociedad Española de Crugía Oral y Maxilofacial. Madrid: SECOM, 2006. 10. Zadik Y, Benoliel R, Fleissig Y, Casap N. Painful trigeminal neuropathy induced by oral bisphosphonate-related osteonecrosis of the jaw: a new etiology for the numb-chin syndrome. Quintessence Int 2012;43:97–104. 11. Goss A, Bartold M, Sabrook P, Hawker P. The nature and frequency of bisphosphonate-associates osteonecrosis of the jaws in dental implant patients: a South Australian case series. J Oral Maxillofac Surg 2010;68:337–343. 12. Vescovi P, Merigo E, Meleti M, et al. Conservative surgical management of stage I bisphosphonate-related osteonecrosis of the jaw. Int J Dent 2014;2014:107690.
VOLUME 45 • NUMBER 9 • OCTOBER 2014
IMPLANTOLOGY
Luis Naval
Dental implants and osteomyelitis in a patient with osteopetrosis Luis Naval, DDS, PhD, DMD1/Manuel Sanchez Molini2/Gonzalo Herrera 2/Beatriz Naval, DDS, MSc 3 Osteopetrosis (OP) is a rare genetic metabolic bone disorder characterized by osteoclast dysfunction impairing bone resorption. Together with normal osteoblastic activity this dysfunction promotes intense bone sclerosis with reduction of marrow spaces as well as bone turnover. Maxillofacial complications are common after tooth extraction. Osteomyelitis (OM) of the mandible occurs in around 10% of the cases with OP. The pur-
pose of this paper is to report the first case of OP treated with dental implants, and to describe the protocol used to treat OM that developed after failure of one implant. The treatment of OM included long-term use of antibiotics and marginal bone resection with a piezoelectric device. Similar cases to confirm the efficacy of this treatment are required. (Quintessence Int 2014;45:765–768; doi: 10.3290/j.qi.a32443)
Key words: bone resorption, dental implants, osteomyelitis, osteopetrosis
Osteopetrosis (OP) is also known as Albers-Schönberg disease, generalized congenital osteosclerosis, ivory bones, marble bone disease, or osteosclerosis fragilis generalisata. It is classified into two main phenotypes: the infantile (malignant) and adult (benign) forms. These OP phenotypes are divided into three types: • Benign or adult OP is an autosomal dominant form with no bone marrow compromise and a good vital prognosis. The serum acid phosphatase is increased because of augmented release by defective osteoclasts.1 • Malignant or infantile OP is autosomal recessive, and has severe bone marrow involvement and poor vital prognosis. It is usually diagnosed before the age of 1 year.2 1
Head, Department of Oral and Maxillofacial Surgery, University Clinic of Navarra, Pamplona, Spain; and Associate Professor, University of Navarra, Pamplona, Spain.
2
Staff Member, Department of Oral and Maxillofacial Surgery, University of Navarra, Pamplona, Spain.
3
Clinician, Private Practice, Clínica Naval, Madrid, Spain.
Correspondence: Dr Beatriz Naval, Clínica Naval, C/ Alcántara 71, 28006 Madrid, Spain. Email: [email protected]
VOLUME 45 • NUMBER 9 • OCTOBER 2014
•
Intermediate OP is an autosomal recessive form with no bone marrow compromise and poor vital prognosis. Bone marrow transplantation must be considered as a treatment option.2
Depending on the severity of the OP, the following signs or symptoms can appear: bone pain, frequent fractures of the long bones, osteomyelitis, nerve compression leading to headache, blindness, deafness, pancytopenia with enlarged spleen, and stroke.1,3 Adult OP can often be asymptomatic for a long period of time, and it has a normal life expectancy. It is characterized by a generalized increase in skeletal mass, headache, frequent bone fractures, and mandibular or maxillary osteomyelitis. Although adult OP has an autosomal dominant inheritance it can also originate because of an isolated mutation. The actual incidence is unknown. However, it is thought to be between 1 in 100,000 and 1 in 500,000. Bone biopsy is not essential for diagnosis. The diagnosis of OP is often secondary to other diseases and findings of specific
765
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
a
b
Fig 1 Panoramic radiograph after removing the failed implant (second left mandibular molar position). Bone sclerosis of the jaws is evident.
Figs 2a and 2b Skeletal radiograph: osteoclerosis of the vertebrae (a) and iliac bones (b).
radiographic features.4 Maxillofacial changes include sclerosis of the facial bones, delayed dental eruption, early tooth loss, congenitally missing or malformed teeth, and thickened lamina dura. A common complication that arises after tooth extraction is osteomyelitis of the mandible. Osteomyelitis of the maxilla is extremely rare.3-5 In the literature, most of the cases of OP with osteomyelitis presented osteomyelitis after dental extraction.6-8 In OP, hypocalcemia can occur due to elevation of the parathyroid hormone (secondary hyperparathyroidism). Furthermore, acid phosphatase can increase as well as brain-type creatine kinase (CK-BB) because of the elevated release from defective osteoclasts. The present article reports a case of adult OP complicated with mandibular osteomyelitis after dental implant treatment. It is the first reported case of OP treated with dental implants.
integration period. It was removed without incident. However, the patient had since suffered from pain and fistula with active suppuration. The soft tissue exhibited gingival swelling. Despite several antibiotic courses the clinical manifestation did not improve over a period of 6 months. The panoramic radiograph (Fig 1) showed bone depression and radiolucency at the site of the failed implant and an augmented bone density through the whole mandible, similar to those seen in patients treated with bisphosphonates. The medical records of the patient were reevaluated for bisphosphonate intake or other pathologic findings. The patient had no other oral complaints. His dental records showed only some “delays and problems” in extraction socket healing. The patient had previously undergone surgery at the Department of Orthopedic Surgery for calcifying tendonitis, both subacromial and patellar. A more extensive radiographic study clearly showed a case of OP (Figs 2 to 4). The diagnosis at this time was mandibular osteomyelitis with poor response to antibiotic therapy in a patient that suffered from adult OP after dental implant surgery. Following the protocols of the Spanish Society of Oral and Maxillofacial Surgery for osteomyelitis,9 the patient was treated with antibiotics for 4 weeks (amoxicillin/clavulonate 875/125 mg every 8 hours), and two curettages of the site (Figs 5 and 6). The second curettage was carried out under sedation and local anesthesia using a piezoelectric device until a clear, clean, and
CASE REPORT A 71-year-old man, suffering from adult OP without prior family history of the disease, came to the Clinic University of Navarra complaining of pain, oral discharge, and fistula on the left side of the mandible (failed implant site). He had been treated with one maxillary and four mandibular implants 2 years previously, in order to improve his masticatory ability. The most distal mandibular implant failed at the end of the
766
VOLUME 45 • NUMBER 9 • OCTOBER 2014
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
Fig 3 Chest radiograph showing osteoclerosis of the ribs and other bones with obliteration of cancellous bone marrow. Two old fractures can be observed on the right side.
Fig 4 Lateral radiograph showing calcifying tendinitis of the patella.
Fig 5
Fig 6 Panoramic radiograph after first curettage and antibiotic treatment for four weeks.
Intraoral aspect at the first curettage.
Fig 7 After piezoelectric scraping until a healthy looking and bleeding bone was reached. Both alveolar canals are narrow because of the osteopetrosis. The left alveolar canal is very close to the bone defect.
Fig 8 Biopsy showed necrotic bone with empty osteocytes lacunae and no bone marrow spaces (hematoxylin-eosin stain).
bleeding bone was obtained. Neither mucosal detachment nor waterproof sutures were performed. The follow-up showed uneventful development for 1 year (Fig 7).
A biopsy showed sclerotic bone without osteocytes or bone marrow spaces (Fig 8). Calcium levels were normal, but parathyroid hormone and creatine kinase (CK) levels were elevated.
VOLUME 45 • NUMBER 9 • OCTOBER 2014
767
Q U I N T E S S E N C E I N T E R N AT I O N A L Naval et al
DISCUSSION
CONCLUSION
The differential diagnosis that can be considered includes other sclerosing bone dysplasias, such as pycnodysostosis, craniometaphyseal dysplasia, diaphyseal dysplasia, melorheostosis, osteopoikilosis, and osteopathia striata. Fluoride poisoning and secondary hyperparathyroidism from renal osteodystrophy may also produce a diffuse osteosclerosis.8 The treatment of osteomyelitis when secondary to OP is controversial. Different treatment regimens include local hygiene, systemic antibiotics, debridement of necrotic bone, and primary closure of soft tissues, if possible. On other occasions hyperbaric oxygen has been used for the treatment of chronic osteomyelitis, especially if related with radionecrosis, but there are no references for this particular bone disease.7 The mechanism of the development of osteomyelitis in OP is the same as for osteonecrosis of the jaw (ONJ) related with drugs10,11 such as antiresorptive medications (bisphosphonates [bisphosphonaterelated ONJ, BRONJ], or denosumab): the loss of function of osteoclasts. Several protocols have been developed in order to prevent or even treat these situations of BRONJ or ONJ. Many of them contraindicated oral surgery (mainly dental extractions or bone curettage) even for the treatment of osteomyelitis itself. Above all, the use of dental implants in these patients has been often criticized or even contraindicated, leaving these patients with no alternative other than very conservative therapies, which often achieve poor results.10-12 Recent reports, however, show that some cases of osteoclast dysfunction diseases can be treated with careful oral surgery, dental extraction, and bone curettage. In carefully selected cases, explaining to the patient the potential risks, advantages, and disadvantages, dental implants can be placed.11,12 Thus the proposal of this surgical piezoelectric scraping treatment can be a surgical compliment or alternative to the failure of antibiotics or hyperbaric oxygen therapy. Some authors suggest the use of Er-YAG laser or careful debridement to remove the avascular and necrotic bone.3,4,6,7,12
This study is the first presented case of dental implants placed in a patient with OP. Circumscribed osteomyelitis of one of the sites was observed after one mandibular implant failure. Careful case selection and consideration of this type of bone pathology must be taken into account before implant placement. The surgical protocol to treat the osteomyelitis in cases with osteoclast dysfunction disease (OP) was described. Nevertheless, more cases must be reported before this treatment can be recommended.
768
REFERENCES 1. de Oliveira Hdo C, Pereira Filho VA, Gabrielli MF, Gabrielli MA, Vieira EH. Marginal resection for treatment of mandibular osteomyelitis associated with osteopetrosis: case report. J Craniomaxillofac Surg 2011;39:525–529. 2. Yamada T, Mishima K, Imura H, et al. Osteomyelitis of the mandible secondary to infantile osteopetrosis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e25–e29. 3. Öğütcen-Toller M, Tek M, Sener I, Bereket C, Inal S, Özden B. Intractable bimaxillary osteomyelitis in osteopetrosis: review of the literature and current therapy. J Oral Maxillofac Surg 2010;68:167–175. 4. Krithika C, Neelakandan RS, Sivapathasundaram B, Koteeswaran D, Rajaram PC, Shetkar GS. Osteopetrosis-associated osteomyelitis of the jaws: a report of 4 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e56–e65. 5. Barry CP, Ryan CD, Stassen LFA. Osteomyelitis of the maxilla secondary to osteopetrosis: a report of 2 cases in sisters. J Oral Maxillofac Surg 2007;65:144–147. 6. Albuquerque MA, Melo ES, Jorge WA, Cavalcanti MG. Osteomyelitis of the mandible associated with autosomal dominant osteopetrosis: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:94–98. 7. García CM, Garcıa M A, Garcıa RG, Gil FM. Osteomyelitis of the mandible in a patient with osteopetrosis. case report and review of the literature. J Maxillofac Oral Surg 2013;12:94–99. 8. Kant P, Sharda N, Bhowate RR. Clinical and radiological findings of autosomal dominant osteopetrosis type II: a case report. Case Rep Dent 2013;2013:707343. 9. García Marín F (ed). Protocolos Clínicos de la Sociedad Española de Crugía Oral y Maxilofacial. Madrid: SECOM, 2006. 10. Zadik Y, Benoliel R, Fleissig Y, Casap N. Painful trigeminal neuropathy induced by oral bisphosphonate-related osteonecrosis of the jaw: a new etiology for the numb-chin syndrome. Quintessence Int 2012;43:97–104. 11. Goss A, Bartold M, Sabrook P, Hawker P. The nature and frequency of bisphosphonate-associates osteonecrosis of the jaws in dental implant patients: a South Australian case series. J Oral Maxillofac Surg 2010;68:337–343. 12. Vescovi P, Merigo E, Meleti M, et al. Conservative surgical management of stage I bisphosphonate-related osteonecrosis of the jaw. Int J Dent 2014;2014:107690.
VOLUME 45 • NUMBER 9 • OCTOBER 2014
