The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
Sagittal Split Ramus Osteotomy for Aneurysmal Bone Cyst of the Mandibular Condyle Amit Bhandari, BDS,* Rohit Sharma, MDS,* Chiyyarath Gopalan Muralidharan, MD, DNB† Abstract: Aneurysmal bone cyst is a benign pseudocystic osseous lesion characterized by a fibrous connective tissue stroma with cellular fibrous tissue, multinucleated giant cells, and large bloodfilled spaces with no endothelial lining. The entity is uncommon in facial bones, and it rarely involves the mandibular condyle. Resection of the lesion is the most accepted treatment. The present case is the 11th reported case of aneurysmal bone cyst of the mandibular condyle in the existing literature and the first where, rather than using conventional extra oral approach, sagittal split ramus osteotomy was used to excise the lesion successfully with no recurrence after 3 years of follow-up. Key Words: Expansive osteolytic lesions, sagittal split ramus osteotomy
B
enign and malignant tumors of the temporomandibular joint are rare. An aneurysmal bone cyst (ABC) of the condyle is even more unusual, with only 10 cases reported in the existing literature.1 Aneurysmal bone cyst are relatively rare osteolytic lesions. They are mainly found within the long bones, and only 2% occur in the jaws. Jaffe and Lichtenstein recognized this cystic lesion for the first time in 1942. Since then, much debate and confusion have ensued regarding its nature and pathogenesis. The World Health Organization defined ABCs as expansive osteolytic lesions consisting of blood-filled spaces and channels divided by connective tissue septa that can contain osteoid tissue and osteoclast-like giant cells. In the long bones, ABCs are characterized radiologically as well-defined, expansive, radiolucent lesions surrounded by a thin overlying cortex. The characteristic radiologic features of jaw ABCs are contradictory and can vary from mainly unilocular radiolucencies to bloated multilocular radiolucencies with honeycomb or soap bubble–like appearances. To date, no consensus has been reached regarding the pathogenesis of ABCs.2 One of the most controversially discussed issues pertains to its origin being primary or secondary. The treatment of ABCs usually includes complete removal by curettage and/or excision. Recurrence, although uncommon, has been reported and might be related to inappropriate access with incomplete removal of the lesion.3 Aneurysmal bone cyst of the mandibular condyle, coronoid, and ramal unit necessitates extraoral approach for complete excision and for better access. This is the first case report until date of ABC involving mandibular condyle where sagittal split From the *Oral & Maxillofacial Surgery, Armed Forces Medical College; and †Department of Radiology, Command Hospital Southern Command, Pune, India. Received July 4, 2014. Accepted for publication August 19, 2014. Address correspondence and reprint requests to Amit Bhandari, BDS, Department of Oral & Maxillofacial Surgery, Armed Forces Medical College, Pune- 411040, India; E-mail: [email protected] No funding was received for this study. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001297
e38
ramus osteotomy (SSRO) was used to address the lesion. The lesion was excised in toto, and no recurrence was noticed after 3 years of follow-up.
CLINICAL REPORT An otherwise healthy 17-year-old male adolescent was referred to our center with a complaint of painless swelling in the right preauricular region which had developed during the last 6 months. History revealed trauma to the chin region 3 years back. Extraoral examination revealed a hard swelling over the right preauricular region. The individual had restricted range of mandibular motion in all directions with no clicking or crepitation. On palpation, the swelling moved along with the sliding condyle. Occlusion was stable, and no abnormality was detected on intraoral examination. Panoramic radiograph revealed a solitary expansile lytic lesion involving the right mandibular condyle, the sigmoid notch, coronoid process, and right ramus with a narrow zone of transition (Fig. 1). Computed tomography with three-dimensional reconstructions revealed expansile lytic lesion with thinning of the cortical margins and absence of any arc like calcifications. Multiplanar magnetic resonance imaging was done on a 1.5-T Signa Excite HD (GE Healthcare, Buckinghamshire, UK) using T1, T2 short-tau inversion recovery sequences followed by contrast-enhanced magnetic resonance imaging. Magnetic resonance angiogram revealed multiseptate expansile lesion involving the right mandibular condyle, the mandibular notch, coronoid process; right ramus measuring 42.9 41.8 41.3 mm (anterioposterior transverse craniocaudal) isointense to muscle on T1-weighted imaging and hyperintense on the T2-weighted imaging, short-tau inversion recovery sequences. There were multiple small fluid-filled levels seen within the lesion on the T1- and T2-weighted imaging. The lesion caused separation of the masseter and parotid gland laterally, and the pterygoid muscles medially. It also resulted in splaying of terminal branches of external carotid artery, that is, the superficial temporal and the internal maxillary artery. Postcontrast scans revealed evidence of enhancement of the internal septations. Clinicoradiologic diagnosis of ABC was made, and the patient was counseled for the surgery accordingly. Patient refused any surgery involving cutaneous incision for aesthetic reasons. Under general anaesthesia, SSRO of the mandible was carried out on the right side after taking approval from the institutional ethical committee. The inferior alveolar nerve was teased out and preserved. Proximal segment harboring the lesion was removed in toto with careful dissection (Fig. 2). The defect was then inspected for any leftover pathology. Hemostasis was achieved. The uninvolved bone with adequate margins was osteotomized and refixed with the distal segment of the mandible using a 4-hole miniplate and screws. The postoperative period was uneventful, and the occlusion was stable. Deviation to right side persisted on mouth opening. Histopathology report confirmed the clinicoradiologic diagnosis of ABC. The patient was assessed on regular recall visits for 3 years, and no recurrence has been noticed to date.
DISCUSSION Aneurysmal bone cysts of the jaws are relatively rare osteolytic lesions that occur in patients of all ages with slight prevalence in
FIGURE 1. Solitary expansile lesion on the right mandibular condyle-coronoid-ramus region with narrow zone of transition on orthopantomogram.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
FIGURE 2. Aneurysmal bone cyst on the proximal segment of SSRO.
the first 2 decades similar to the case reported in this article. Usually, no sex predilection is found. The mandible is more prone to being affected, with incident rates of 3:1 to 11:9. More than 90% of jaw ABCs occur in the posterior regions of the jaws.4 In the present case, the lesion involved the mandibular condyle extending to involve the coronoid, sigmoid notch, and the ramal region. It is the 11th reported case of the ABC involving mandibular condyle in existing literature. The ABC in the jaw region can have considerably varied clinical features ranging from asymptomatic lesions occasionally discovered as radiolucencies on routine radiography to sometimes expansive and destructive patterns. The main symptom according to the published data is a painless swelling. Varying degree of limited mouth opening can occur if the lesion involves mandibular condyle or ramus. In our case, both painless swelling and restricted range of mandibular motion in all directions were present. Aneurysmal bone cyst frequently appears as a well-circumscribed bloated multilocular lesion on the radiograph. In the reported case, all the typical radiographic features of ABC were present. The accepted treatment of ABC is surgical resection as high rate of recurrence is seen with enucleation.5 The access to the condylar region is possible through transparotid or anteroparotid transmasseteric, preauricular, Risdon, or submandibular and intraoral approach. The combination of preauricular and submandibular incision is most commonly used for condylar pathology resection and reconstruction. In our case, this approach was not used because the patient denied consent for any cutaneous approach for the resection of the lesion. Intraoral resection of the lesion without any reconstruction was the only available choice. Sagittal split ramus osteotomy has been described in the literature for approaches to mandibular cysts,6 myxomas,7 keratocyst odontogenic tumor, odontomas,8,9 and deeply impacted third molars.10 The procedure is simple and leads to increased accessibility to the lesion without sacrificing bone and causing damage to inferior alveolar nerve. Because of the overlap of proximal and distal segment after SSRO, the union/healing of the osteotomized segment is better if adequate osteosynthesis is used. In our case, the lesion was above the lingula, and adequate bone was present for SSRO. The advantage other than of carrying out resection of the pathology intraorally and avoiding any extraoral scar was that even without reconstruction, enough height of the mandible was preserved lingually, and the uninvolved buccal cortex was refixed using titanium miniplate and screws. The drawback is limited access to dissect out soft tissue attachments from the condylar head and removal of the thinned-out lesion in toto. Aneurysmal bone cyst was excised in toto, and no clinical or radiological recurrence was noticed after 3 years of follow-up. The approach is technique sensitive but can be used to address condylar pathologies, avoiding any facial scar or nerve damage.
REFERENCES 1. Zadik Y, Aktaş A, Drucker S, et al. Aneurysmal bone cyst of mandibular condyle: a case report and review of the literature. J Craniomaxillofac Surg 2012;40:e243–e248
Brief Clinical Studies
2. Cottalorda J, Bourelle S. Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 2007;127:105–114 3. Kumar VV, Malik NA, Kumar DB. Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous intralesional embolization as an adjunct to resection. Int J Oral Maxillofac Surg 2009;38:671–676 4. Sun ZJ, Zhao YF, Yang RL, et al. Aneurysmal bone cysts of the jaws: analysis of 17 cases. J Oral Maxillofac Surg 2010;68:2122–2128 5. Choi BJ, Choi SC, Kwon YD, et al. Aneurysmal bone cyst causing a pathologic fracture of the mandibular condyle. J Oral Maxillofac Surg 2011;69:2995–3000 6. Rittersma J, van Gool AV. Surgical access to multicystic lesions, by sagittal splitting of the lower jaw. J Maxillofac Surg 1979;7:246–250 7. Wong GB. Large odontogenic myxoma of the mandible treated by sagittal ramus osteotomy and peripheral ostectomy. J Oral Maxillofac Surg 1992;50:1221–1224 8. Casap N, Zeltser R, Abu-Tair J, et al. Removal of odontoma by sagittal split osteotomy. J Oral Maxillofac Surg 2006;64:1833–1836 9. Blinder D, Peleg M, Taicher S. Surgical considerations in cases of large mandibular odontomas located in the mandibular angle. Int J Oral Maxillofac Surg 1993;22:163–165 10. Toffanin A, Zupi A, Cicognini A. Sagittal split osteotomy in removal of impacted third molar. J Oral Maxillofac Surg 2003;61:638–640
Simultaneous Transsphenoidal and Transventricular Endoscopic Approaches for Giant Pituitary Adenoma With Hydrocephalus Ender Koktekir, MD,* Hakan Karabagli, MD,* Kayhan Ozturk, MD† Abstract: The surgical management of giant pituitary adenomas is challenging. Although most pituitary adenomas, even those with suprasellar extension, can be resected using the transsphenoidal surgery alone, the transcranial approach is still needed for approximately 1% to 4% of these tumors. The transcranial approach is usually used in large adenomas with hourglass configuration and adenomas with firm consistency impeding the adjunctive measures, which are used for delivering the suprasellar part of the tumor into the sellar area and thereby obscure the tumor resection by transsphenoidal route. In this report, we describe the successful use of transventricular endoscope as an adjunctive measure to remove giant pituitary adenoma from transsphenoidal route and discuss the limitations of this new technique. We concluded that this technique would be used safely in selected cases. Case selection and surgical strategies should be based on preoperative magnetic resonance imaging findings, ventricular size, and the availability of experienced surgeons. From the Departments of *Neurosurgery and †Otolaryngology, Selcuk University, Konya, Turkey. Received July 5, 2014. Accepted for publication August 19, 2014. Address correspondence and reprint requests to Ender Koktekir, MD, Department of Neurosurgery, Selcuk Universitesi Tip Fakultesi Neurosirurji Anabilim Dali, Alaeddin Keykubat Kampusu 42050, Konya, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001298
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e39
Brief Clinical Studies
Sagittal Split Ramus Osteotomy for Aneurysmal Bone Cyst of the Mandibular Condyle Amit Bhandari, BDS,* Rohit Sharma, MDS,* Chiyyarath Gopalan Muralidharan, MD, DNB† Abstract: Aneurysmal bone cyst is a benign pseudocystic osseous lesion characterized by a fibrous connective tissue stroma with cellular fibrous tissue, multinucleated giant cells, and large bloodfilled spaces with no endothelial lining. The entity is uncommon in facial bones, and it rarely involves the mandibular condyle. Resection of the lesion is the most accepted treatment. The present case is the 11th reported case of aneurysmal bone cyst of the mandibular condyle in the existing literature and the first where, rather than using conventional extra oral approach, sagittal split ramus osteotomy was used to excise the lesion successfully with no recurrence after 3 years of follow-up. Key Words: Expansive osteolytic lesions, sagittal split ramus osteotomy
B
enign and malignant tumors of the temporomandibular joint are rare. An aneurysmal bone cyst (ABC) of the condyle is even more unusual, with only 10 cases reported in the existing literature.1 Aneurysmal bone cyst are relatively rare osteolytic lesions. They are mainly found within the long bones, and only 2% occur in the jaws. Jaffe and Lichtenstein recognized this cystic lesion for the first time in 1942. Since then, much debate and confusion have ensued regarding its nature and pathogenesis. The World Health Organization defined ABCs as expansive osteolytic lesions consisting of blood-filled spaces and channels divided by connective tissue septa that can contain osteoid tissue and osteoclast-like giant cells. In the long bones, ABCs are characterized radiologically as well-defined, expansive, radiolucent lesions surrounded by a thin overlying cortex. The characteristic radiologic features of jaw ABCs are contradictory and can vary from mainly unilocular radiolucencies to bloated multilocular radiolucencies with honeycomb or soap bubble–like appearances. To date, no consensus has been reached regarding the pathogenesis of ABCs.2 One of the most controversially discussed issues pertains to its origin being primary or secondary. The treatment of ABCs usually includes complete removal by curettage and/or excision. Recurrence, although uncommon, has been reported and might be related to inappropriate access with incomplete removal of the lesion.3 Aneurysmal bone cyst of the mandibular condyle, coronoid, and ramal unit necessitates extraoral approach for complete excision and for better access. This is the first case report until date of ABC involving mandibular condyle where sagittal split From the *Oral & Maxillofacial Surgery, Armed Forces Medical College; and †Department of Radiology, Command Hospital Southern Command, Pune, India. Received July 4, 2014. Accepted for publication August 19, 2014. Address correspondence and reprint requests to Amit Bhandari, BDS, Department of Oral & Maxillofacial Surgery, Armed Forces Medical College, Pune- 411040, India; E-mail: [email protected] No funding was received for this study. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001297
e38
ramus osteotomy (SSRO) was used to address the lesion. The lesion was excised in toto, and no recurrence was noticed after 3 years of follow-up.
CLINICAL REPORT An otherwise healthy 17-year-old male adolescent was referred to our center with a complaint of painless swelling in the right preauricular region which had developed during the last 6 months. History revealed trauma to the chin region 3 years back. Extraoral examination revealed a hard swelling over the right preauricular region. The individual had restricted range of mandibular motion in all directions with no clicking or crepitation. On palpation, the swelling moved along with the sliding condyle. Occlusion was stable, and no abnormality was detected on intraoral examination. Panoramic radiograph revealed a solitary expansile lytic lesion involving the right mandibular condyle, the sigmoid notch, coronoid process, and right ramus with a narrow zone of transition (Fig. 1). Computed tomography with three-dimensional reconstructions revealed expansile lytic lesion with thinning of the cortical margins and absence of any arc like calcifications. Multiplanar magnetic resonance imaging was done on a 1.5-T Signa Excite HD (GE Healthcare, Buckinghamshire, UK) using T1, T2 short-tau inversion recovery sequences followed by contrast-enhanced magnetic resonance imaging. Magnetic resonance angiogram revealed multiseptate expansile lesion involving the right mandibular condyle, the mandibular notch, coronoid process; right ramus measuring 42.9 41.8 41.3 mm (anterioposterior transverse craniocaudal) isointense to muscle on T1-weighted imaging and hyperintense on the T2-weighted imaging, short-tau inversion recovery sequences. There were multiple small fluid-filled levels seen within the lesion on the T1- and T2-weighted imaging. The lesion caused separation of the masseter and parotid gland laterally, and the pterygoid muscles medially. It also resulted in splaying of terminal branches of external carotid artery, that is, the superficial temporal and the internal maxillary artery. Postcontrast scans revealed evidence of enhancement of the internal septations. Clinicoradiologic diagnosis of ABC was made, and the patient was counseled for the surgery accordingly. Patient refused any surgery involving cutaneous incision for aesthetic reasons. Under general anaesthesia, SSRO of the mandible was carried out on the right side after taking approval from the institutional ethical committee. The inferior alveolar nerve was teased out and preserved. Proximal segment harboring the lesion was removed in toto with careful dissection (Fig. 2). The defect was then inspected for any leftover pathology. Hemostasis was achieved. The uninvolved bone with adequate margins was osteotomized and refixed with the distal segment of the mandible using a 4-hole miniplate and screws. The postoperative period was uneventful, and the occlusion was stable. Deviation to right side persisted on mouth opening. Histopathology report confirmed the clinicoradiologic diagnosis of ABC. The patient was assessed on regular recall visits for 3 years, and no recurrence has been noticed to date.
DISCUSSION Aneurysmal bone cysts of the jaws are relatively rare osteolytic lesions that occur in patients of all ages with slight prevalence in
FIGURE 1. Solitary expansile lesion on the right mandibular condyle-coronoid-ramus region with narrow zone of transition on orthopantomogram.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
FIGURE 2. Aneurysmal bone cyst on the proximal segment of SSRO.
the first 2 decades similar to the case reported in this article. Usually, no sex predilection is found. The mandible is more prone to being affected, with incident rates of 3:1 to 11:9. More than 90% of jaw ABCs occur in the posterior regions of the jaws.4 In the present case, the lesion involved the mandibular condyle extending to involve the coronoid, sigmoid notch, and the ramal region. It is the 11th reported case of the ABC involving mandibular condyle in existing literature. The ABC in the jaw region can have considerably varied clinical features ranging from asymptomatic lesions occasionally discovered as radiolucencies on routine radiography to sometimes expansive and destructive patterns. The main symptom according to the published data is a painless swelling. Varying degree of limited mouth opening can occur if the lesion involves mandibular condyle or ramus. In our case, both painless swelling and restricted range of mandibular motion in all directions were present. Aneurysmal bone cyst frequently appears as a well-circumscribed bloated multilocular lesion on the radiograph. In the reported case, all the typical radiographic features of ABC were present. The accepted treatment of ABC is surgical resection as high rate of recurrence is seen with enucleation.5 The access to the condylar region is possible through transparotid or anteroparotid transmasseteric, preauricular, Risdon, or submandibular and intraoral approach. The combination of preauricular and submandibular incision is most commonly used for condylar pathology resection and reconstruction. In our case, this approach was not used because the patient denied consent for any cutaneous approach for the resection of the lesion. Intraoral resection of the lesion without any reconstruction was the only available choice. Sagittal split ramus osteotomy has been described in the literature for approaches to mandibular cysts,6 myxomas,7 keratocyst odontogenic tumor, odontomas,8,9 and deeply impacted third molars.10 The procedure is simple and leads to increased accessibility to the lesion without sacrificing bone and causing damage to inferior alveolar nerve. Because of the overlap of proximal and distal segment after SSRO, the union/healing of the osteotomized segment is better if adequate osteosynthesis is used. In our case, the lesion was above the lingula, and adequate bone was present for SSRO. The advantage other than of carrying out resection of the pathology intraorally and avoiding any extraoral scar was that even without reconstruction, enough height of the mandible was preserved lingually, and the uninvolved buccal cortex was refixed using titanium miniplate and screws. The drawback is limited access to dissect out soft tissue attachments from the condylar head and removal of the thinned-out lesion in toto. Aneurysmal bone cyst was excised in toto, and no clinical or radiological recurrence was noticed after 3 years of follow-up. The approach is technique sensitive but can be used to address condylar pathologies, avoiding any facial scar or nerve damage.
REFERENCES 1. Zadik Y, Aktaş A, Drucker S, et al. Aneurysmal bone cyst of mandibular condyle: a case report and review of the literature. J Craniomaxillofac Surg 2012;40:e243–e248
Brief Clinical Studies
2. Cottalorda J, Bourelle S. Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 2007;127:105–114 3. Kumar VV, Malik NA, Kumar DB. Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous intralesional embolization as an adjunct to resection. Int J Oral Maxillofac Surg 2009;38:671–676 4. Sun ZJ, Zhao YF, Yang RL, et al. Aneurysmal bone cysts of the jaws: analysis of 17 cases. J Oral Maxillofac Surg 2010;68:2122–2128 5. Choi BJ, Choi SC, Kwon YD, et al. Aneurysmal bone cyst causing a pathologic fracture of the mandibular condyle. J Oral Maxillofac Surg 2011;69:2995–3000 6. Rittersma J, van Gool AV. Surgical access to multicystic lesions, by sagittal splitting of the lower jaw. J Maxillofac Surg 1979;7:246–250 7. Wong GB. Large odontogenic myxoma of the mandible treated by sagittal ramus osteotomy and peripheral ostectomy. J Oral Maxillofac Surg 1992;50:1221–1224 8. Casap N, Zeltser R, Abu-Tair J, et al. Removal of odontoma by sagittal split osteotomy. J Oral Maxillofac Surg 2006;64:1833–1836 9. Blinder D, Peleg M, Taicher S. Surgical considerations in cases of large mandibular odontomas located in the mandibular angle. Int J Oral Maxillofac Surg 1993;22:163–165 10. Toffanin A, Zupi A, Cicognini A. Sagittal split osteotomy in removal of impacted third molar. J Oral Maxillofac Surg 2003;61:638–640
Simultaneous Transsphenoidal and Transventricular Endoscopic Approaches for Giant Pituitary Adenoma With Hydrocephalus Ender Koktekir, MD,* Hakan Karabagli, MD,* Kayhan Ozturk, MD† Abstract: The surgical management of giant pituitary adenomas is challenging. Although most pituitary adenomas, even those with suprasellar extension, can be resected using the transsphenoidal surgery alone, the transcranial approach is still needed for approximately 1% to 4% of these tumors. The transcranial approach is usually used in large adenomas with hourglass configuration and adenomas with firm consistency impeding the adjunctive measures, which are used for delivering the suprasellar part of the tumor into the sellar area and thereby obscure the tumor resection by transsphenoidal route. In this report, we describe the successful use of transventricular endoscope as an adjunctive measure to remove giant pituitary adenoma from transsphenoidal route and discuss the limitations of this new technique. We concluded that this technique would be used safely in selected cases. Case selection and surgical strategies should be based on preoperative magnetic resonance imaging findings, ventricular size, and the availability of experienced surgeons. From the Departments of *Neurosurgery and †Otolaryngology, Selcuk University, Konya, Turkey. Received July 5, 2014. Accepted for publication August 19, 2014. Address correspondence and reprint requests to Ender Koktekir, MD, Department of Neurosurgery, Selcuk Universitesi Tip Fakultesi Neurosirurji Anabilim Dali, Alaeddin Keykubat Kampusu 42050, Konya, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001298
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e39
