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An atypical presentation of a solitary bone cyst of the mandibular ramus: A case report
Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 145e147
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
An atypical presentation of a solitary bone cyst of the mandibular ramus: A case report E.M. Strabbing a, R.A.Th. Gortzak a, J.G. Vinke b, C.P. Saridin c, J.P.R. van Merkesteyn a, * a
Department of Oral and Maxillofacial Surgery (Head: Prof. Dr. J.P.R. van Merkesteyn), Albinusdreef 2, PO Box 9600, 2300 RC Leiden, The Netherlands Department of Otorhinolaryngology, HAGA Hospital, The Hague, The Netherlands c Department of Oral and Maxillofacial Surgery, HAGA Hospital, The Hague, The Netherlands b
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 23 January 2010 Accepted 21 October 2010
This report describes a large solitary bone cyst involving the mandibular ramus, presenting with a history of recurrent hypoaesthesia of the lower lip and a multilocular, multilobular radiological aspect with cortical expansion and possibly cortical perforation. Ó 2010 European Association for Cranio-Maxillo-Facial Surgery.
Keywords: Radiolucent lesion Mandibular ramus Case report Solitary bone cyst Atypical Subjective recurrent hypoaesthesia
1. Introduction Synonyms of a solitary bone cyst are simple bone cyst, traumatic bone cyst, hemorrhagic bone cyst, hemorrhagic cyst, idiopathic bone cavity and unicameral bone cyst. The international histological classification of tumours by the World Health Organization (WHO) recommends use of the term solitary bone cyst. A solitary bone cyst is an intraosseous pseudocyst having a tenuous lining of connective tissue without epithelium, either empty or filled with serous or sanguinous fluid (Kramer et al., 1992). The cause of solitary bone cysts is still unclear, however, several theories regarding the aetiology have been suggested. A recent review of the aetiopathogenic hypotheses of solitary bone cysts of the jaws discusses the major etiologic processes often quoted but which are still under debate, being tumoural degeneration, trauma, and defects linked to periods of bone growth (Harnet et al., 2008). Xanthinaki reported a case with iatrogenic trauma suggested as the principal aetiological factor (Xanthinaki et al., 2006). Solitary bone cysts are predominately diagnosed in the first two decades of life. Most often they are asymptomatic and discovered accidentally on a panoramic radiograph. A radiographic review of 44 cases of solitary bone cysts showed that in nearly two-thirds of cases solitary bone cysts were cone-
* Corresponding author. Tel.: þ31 71 526 2371; fax: þ31 71 526 6766. E-mail address: [email protected] (J.P.R. van Merkesteyn).
shaped (Copete et al., 1998). The location is predominantly the premolaremolar area of the mandible, with scalloping of the margin between the roots (Copete et al., 1998). In some instances, the lesions crossed the midline (Copete et al., 1998). Histopathologically, the walls of the defect may be lined by a thin band of vascular fibrous connective tissue. There is never any evidence of an epithelial lining (Neville et al., 2009). Treatment of solitary bone cysts is surgical exploration and curettage of the bone wall. However, the recurrence rate was found to be greater than 20% (Suei et al., 2007). In most cases healing or recurrence will be confirmed within 3 years after surgery (Suei et al., 2007). This case report describes a clinically and radiographically atypical case of a large radiolucency involving the mandibular ramus. It presented as a multilocular, multilobular lesion with irregular but well-defined borders without a clear sclerotic lining. The differential diagnosis included odontogenic keratocyst, odontogenic tumour (ameloblastoma, odontogenic myxoma), giant cell granuloma, aneurysmal bone cyst and solitary bone cyst. 2. Case report A 22-year-old woman was referred to the department of Oral and Maxillofacial Surgery of the Leiden University Medical Centre for investigation and treatment of a large well-defined radiolucent structure affecting the right ramus of the mandible. The lesion had been noticed on a panoramic radiograph (Fig. 1), taken because
1010-5182/$ e see front matter Ó 2010 European Association for Cranio-Maxillo-Facial Surgery. doi:10.1016/j.jcms.2010.10.023
146
E.M. Strabbing et al. / Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 145e147
Fig. 1. Panoramic radiograph showing a large radiolucent, multilocular lesion of the right mandibular ramus with scalloped margins, expansion and resorption of the cortices.
Fig. 3. During surgical exploration, a bone cavity with exposure of the inferior alveolar nerve was noticed.
Fig. 2. Computer tomography. This slice shows expansion of the buccal and lingual cortex of the right mandibular ramus and signs of perforation of the cortex.
of slight pain and feelings of ‘pressure’ of the right mandible. The patient sometimes noticed a bad taste. During the periods of pain, the patient experienced hypoaesthesia of the right inferior alveolar nerve. On clinical examination there was no visible swelling, but intraoral palpation revealed a slight expansion of the buccal cortex of the mandibular ramus. The patient was otherwise healthy, with a complete dentition. The first and the second lower molars on the right side were vital on testing. No fistulas were found at examination. The panoramic radiograph (Fig. 1) showed a large, expansive radiolucent lesion of the right mandibular ramus with a multilocular aspect, a scalloped margin and resorption of the cortex. A computerized tomography scan showed expansion of the lingual and buccal cortex of the right mandibular ramus (Fig. 2). Furthermore, there were signs of perforation of the cortex. A radiological differential diagnosis of cystic lesion or tumour was made. The differential diagnosis based on the clinical and radiographical appearances was odontogenic keratocyst, odontogenic tumour (ameloblastoma) and giant cell granuloma. The patient was scheduled for surgical exploration under general anaesthesia. A buccal mucoperiosteal flap was made along the anterior border of the ramus. A window was made with a round bur in order to expose the lesion. A bone cavity with an overlying thin membrane-like structure was noticed (Fig. 3). The cavity itself
Fig. 4. Panoramic radiograph 6 months postoperatively with nearly complete ossification of the defect.
was filled with a small quantity of straw-colored fluid. The walls of the cavity were curetted to induce bleeding. The mucoperiosteal flap was replaced and closed primarily. A clinical diagnosis of solitary bone cyst was made. Histological examination of wall fragments revealed bone without specific characteristics. The operative findings and histologic examination were consistent with a diagnosis of solitary bone cyst. In the third postoperative week the patient presented with a small dehiscence of the wound and swelling of the right cheek. The patient was instructed to rinse the wound and oral antibiotics were administered for 1 week (amoxicillin 500 mg, every 8 h). The following postoperative course was uneventful. At the 3 months follow-up, a panoramic radiograph showed normal bone healing. A panoramic radiograph taken 6 months postoperatively revealed nearly complete ossification of the defect (Fig. 4). 3. Discussion This case describes a clinically and radiographically atypical case of a large radiolucent lesion involving the right mandibular ramus. The patient had periods of pain and during those periods, she reported a hypoaesthesia of the right inferior alveolar nerve. In the literature hypoaesthesia due to a solitary bone cyst is a very rare
E.M. Strabbing et al. / Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 145e147
presenting symptom with only 3 reported cases (Goodstein et al., 1976; Harris et al., 1992; Huebner and Turlington, 1971). In all of these 3 cases the inferior alveolar nerve was lying free within the cavity. Two of the 3 reported cases were free of symptoms after treatment. As in this case, no clear signs of inflammation were found, therefore a good explanation for the hypoaesthesia was not found. Solitary bone cysts of the mandibular ramus are relatively rare. In 7 of the 121 (5.8%) reported cases, with known localisation, the mandibular ramus was involved (Forssell et al., 1988; Shigematsu et al., 1994; Matsumura et al., 1998; Copete et al., 1998). This lesion showed radiographic characteristics of an ameloblastoma or a keratocyst due to its location, size and multilocular appearance. Furthermore there was bucco-lingual bone expansion on the CT-scan which is considered a sign of neoplasm or growth of a cyst. Another atypical characteristic of the lesion was a suggestion of perforation of the cortex. The nearly complete ossification of the defect within 6 months after surgery confirmed the diagnosis. 4. Conclusion An atypical case of a solitary bone cyst is presented. The clinically atypical symptoms were pain associated with hypoaesthesia of the inferior alveolar nerve. The radiologically atypical presentation consisted of the unusual location (the mandibular ramus), size, multilocular appearance and the presence of bucco-lingual bone expansion with signs of cortical perforation. The rapid ossification of this large lesion after treatment was remarkable.
147
Conflict of interest No conflicts of interest are present. References Copete MA, Kawamata A, Langlais RP: Solitary bone cyst of the jaws: radiographic review of 44 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 85: 221e225, 1998 Forssell K, Forssell H, Happonen RP, Neva M: Simple bone cyst: review of the literature and analysis of 23 cases. Int J Oral Maxillofac Surg 17: 21e24, 1988 Goodstein DB, Himmerfarb R, Hempstead NY: Paresthesia and the traumatic bone cyst. Oral Surg 42: 442e446, 1976 Harris SJ, O Carroll MK, Gordy FM: Idiopathic bone cavity (traumatic bone cyst) with the radiographic appearance of a fibro-osseous lesion. Oral Surg Oral Med Oral Pathol 74: 118e123, 1992 Harnet JC, Lombardi T, Klewansky P, Rieger J, Tempe MH, Clavert JM: Solitary bone cyst of the jaws: a review of the etiopathogenic hypotheses. J Oral Maxillofac Surg 66: 2345e2348, 2008 Huebner GR, Turlington EG: So-called traumatic (hemorrhagic) bone cysts of the jaws: review of the literature and report of two unusual cases. Oral Surg Oral Med Oral Pathol 31: 354e365, 1971 Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumours, vol. 33. New York: Springer-Verlag, 1992 Matsumura S, Murakami S, Kakimoto N, Furukawa S, Kishino M, Ishida T, et al: Histopathologic and radiographic findings of the simple bone cyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 85: 619e625, 1998 Neville BW, Damm DD, Allen CM, Bouquot JE: Oral and maxillofacial pathology, Missouri: Saunders Elsevier, 631e633, 2009 Shigematsu H, Fujita K, Watanabe K: Atypical simple bone cyst of the mandible. Int J Oral Maxillofac Surg 23: 298e299, 1994 Suei Y, Taguchi A, Tanimoto K: Simple bone cyst of the jaws: evaluation of treatment outcome by review of 132 cases. J Oral Maxillofac Surg 65: 918e923, 2007 Xanthinaki AA, Choupis KI, Tosios K, Pagkalos VA, Papanikolaou SI: Traumatic bone cyst of the mandible of possible iatrogenic origin: a case report and brief review of the literature. Head Face Med 2: 40, 2006
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
An atypical presentation of a solitary bone cyst of the mandibular ramus: A case report E.M. Strabbing a, R.A.Th. Gortzak a, J.G. Vinke b, C.P. Saridin c, J.P.R. van Merkesteyn a, * a
Department of Oral and Maxillofacial Surgery (Head: Prof. Dr. J.P.R. van Merkesteyn), Albinusdreef 2, PO Box 9600, 2300 RC Leiden, The Netherlands Department of Otorhinolaryngology, HAGA Hospital, The Hague, The Netherlands c Department of Oral and Maxillofacial Surgery, HAGA Hospital, The Hague, The Netherlands b
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 23 January 2010 Accepted 21 October 2010
This report describes a large solitary bone cyst involving the mandibular ramus, presenting with a history of recurrent hypoaesthesia of the lower lip and a multilocular, multilobular radiological aspect with cortical expansion and possibly cortical perforation. Ó 2010 European Association for Cranio-Maxillo-Facial Surgery.
Keywords: Radiolucent lesion Mandibular ramus Case report Solitary bone cyst Atypical Subjective recurrent hypoaesthesia
1. Introduction Synonyms of a solitary bone cyst are simple bone cyst, traumatic bone cyst, hemorrhagic bone cyst, hemorrhagic cyst, idiopathic bone cavity and unicameral bone cyst. The international histological classification of tumours by the World Health Organization (WHO) recommends use of the term solitary bone cyst. A solitary bone cyst is an intraosseous pseudocyst having a tenuous lining of connective tissue without epithelium, either empty or filled with serous or sanguinous fluid (Kramer et al., 1992). The cause of solitary bone cysts is still unclear, however, several theories regarding the aetiology have been suggested. A recent review of the aetiopathogenic hypotheses of solitary bone cysts of the jaws discusses the major etiologic processes often quoted but which are still under debate, being tumoural degeneration, trauma, and defects linked to periods of bone growth (Harnet et al., 2008). Xanthinaki reported a case with iatrogenic trauma suggested as the principal aetiological factor (Xanthinaki et al., 2006). Solitary bone cysts are predominately diagnosed in the first two decades of life. Most often they are asymptomatic and discovered accidentally on a panoramic radiograph. A radiographic review of 44 cases of solitary bone cysts showed that in nearly two-thirds of cases solitary bone cysts were cone-
* Corresponding author. Tel.: þ31 71 526 2371; fax: þ31 71 526 6766. E-mail address: [email protected] (J.P.R. van Merkesteyn).
shaped (Copete et al., 1998). The location is predominantly the premolaremolar area of the mandible, with scalloping of the margin between the roots (Copete et al., 1998). In some instances, the lesions crossed the midline (Copete et al., 1998). Histopathologically, the walls of the defect may be lined by a thin band of vascular fibrous connective tissue. There is never any evidence of an epithelial lining (Neville et al., 2009). Treatment of solitary bone cysts is surgical exploration and curettage of the bone wall. However, the recurrence rate was found to be greater than 20% (Suei et al., 2007). In most cases healing or recurrence will be confirmed within 3 years after surgery (Suei et al., 2007). This case report describes a clinically and radiographically atypical case of a large radiolucency involving the mandibular ramus. It presented as a multilocular, multilobular lesion with irregular but well-defined borders without a clear sclerotic lining. The differential diagnosis included odontogenic keratocyst, odontogenic tumour (ameloblastoma, odontogenic myxoma), giant cell granuloma, aneurysmal bone cyst and solitary bone cyst. 2. Case report A 22-year-old woman was referred to the department of Oral and Maxillofacial Surgery of the Leiden University Medical Centre for investigation and treatment of a large well-defined radiolucent structure affecting the right ramus of the mandible. The lesion had been noticed on a panoramic radiograph (Fig. 1), taken because
1010-5182/$ e see front matter Ó 2010 European Association for Cranio-Maxillo-Facial Surgery. doi:10.1016/j.jcms.2010.10.023
146
E.M. Strabbing et al. / Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 145e147
Fig. 1. Panoramic radiograph showing a large radiolucent, multilocular lesion of the right mandibular ramus with scalloped margins, expansion and resorption of the cortices.
Fig. 3. During surgical exploration, a bone cavity with exposure of the inferior alveolar nerve was noticed.
Fig. 2. Computer tomography. This slice shows expansion of the buccal and lingual cortex of the right mandibular ramus and signs of perforation of the cortex.
of slight pain and feelings of ‘pressure’ of the right mandible. The patient sometimes noticed a bad taste. During the periods of pain, the patient experienced hypoaesthesia of the right inferior alveolar nerve. On clinical examination there was no visible swelling, but intraoral palpation revealed a slight expansion of the buccal cortex of the mandibular ramus. The patient was otherwise healthy, with a complete dentition. The first and the second lower molars on the right side were vital on testing. No fistulas were found at examination. The panoramic radiograph (Fig. 1) showed a large, expansive radiolucent lesion of the right mandibular ramus with a multilocular aspect, a scalloped margin and resorption of the cortex. A computerized tomography scan showed expansion of the lingual and buccal cortex of the right mandibular ramus (Fig. 2). Furthermore, there were signs of perforation of the cortex. A radiological differential diagnosis of cystic lesion or tumour was made. The differential diagnosis based on the clinical and radiographical appearances was odontogenic keratocyst, odontogenic tumour (ameloblastoma) and giant cell granuloma. The patient was scheduled for surgical exploration under general anaesthesia. A buccal mucoperiosteal flap was made along the anterior border of the ramus. A window was made with a round bur in order to expose the lesion. A bone cavity with an overlying thin membrane-like structure was noticed (Fig. 3). The cavity itself
Fig. 4. Panoramic radiograph 6 months postoperatively with nearly complete ossification of the defect.
was filled with a small quantity of straw-colored fluid. The walls of the cavity were curetted to induce bleeding. The mucoperiosteal flap was replaced and closed primarily. A clinical diagnosis of solitary bone cyst was made. Histological examination of wall fragments revealed bone without specific characteristics. The operative findings and histologic examination were consistent with a diagnosis of solitary bone cyst. In the third postoperative week the patient presented with a small dehiscence of the wound and swelling of the right cheek. The patient was instructed to rinse the wound and oral antibiotics were administered for 1 week (amoxicillin 500 mg, every 8 h). The following postoperative course was uneventful. At the 3 months follow-up, a panoramic radiograph showed normal bone healing. A panoramic radiograph taken 6 months postoperatively revealed nearly complete ossification of the defect (Fig. 4). 3. Discussion This case describes a clinically and radiographically atypical case of a large radiolucent lesion involving the right mandibular ramus. The patient had periods of pain and during those periods, she reported a hypoaesthesia of the right inferior alveolar nerve. In the literature hypoaesthesia due to a solitary bone cyst is a very rare
E.M. Strabbing et al. / Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 145e147
presenting symptom with only 3 reported cases (Goodstein et al., 1976; Harris et al., 1992; Huebner and Turlington, 1971). In all of these 3 cases the inferior alveolar nerve was lying free within the cavity. Two of the 3 reported cases were free of symptoms after treatment. As in this case, no clear signs of inflammation were found, therefore a good explanation for the hypoaesthesia was not found. Solitary bone cysts of the mandibular ramus are relatively rare. In 7 of the 121 (5.8%) reported cases, with known localisation, the mandibular ramus was involved (Forssell et al., 1988; Shigematsu et al., 1994; Matsumura et al., 1998; Copete et al., 1998). This lesion showed radiographic characteristics of an ameloblastoma or a keratocyst due to its location, size and multilocular appearance. Furthermore there was bucco-lingual bone expansion on the CT-scan which is considered a sign of neoplasm or growth of a cyst. Another atypical characteristic of the lesion was a suggestion of perforation of the cortex. The nearly complete ossification of the defect within 6 months after surgery confirmed the diagnosis. 4. Conclusion An atypical case of a solitary bone cyst is presented. The clinically atypical symptoms were pain associated with hypoaesthesia of the inferior alveolar nerve. The radiologically atypical presentation consisted of the unusual location (the mandibular ramus), size, multilocular appearance and the presence of bucco-lingual bone expansion with signs of cortical perforation. The rapid ossification of this large lesion after treatment was remarkable.
147
Conflict of interest No conflicts of interest are present. References Copete MA, Kawamata A, Langlais RP: Solitary bone cyst of the jaws: radiographic review of 44 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 85: 221e225, 1998 Forssell K, Forssell H, Happonen RP, Neva M: Simple bone cyst: review of the literature and analysis of 23 cases. Int J Oral Maxillofac Surg 17: 21e24, 1988 Goodstein DB, Himmerfarb R, Hempstead NY: Paresthesia and the traumatic bone cyst. Oral Surg 42: 442e446, 1976 Harris SJ, O Carroll MK, Gordy FM: Idiopathic bone cavity (traumatic bone cyst) with the radiographic appearance of a fibro-osseous lesion. Oral Surg Oral Med Oral Pathol 74: 118e123, 1992 Harnet JC, Lombardi T, Klewansky P, Rieger J, Tempe MH, Clavert JM: Solitary bone cyst of the jaws: a review of the etiopathogenic hypotheses. J Oral Maxillofac Surg 66: 2345e2348, 2008 Huebner GR, Turlington EG: So-called traumatic (hemorrhagic) bone cysts of the jaws: review of the literature and report of two unusual cases. Oral Surg Oral Med Oral Pathol 31: 354e365, 1971 Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumours, vol. 33. New York: Springer-Verlag, 1992 Matsumura S, Murakami S, Kakimoto N, Furukawa S, Kishino M, Ishida T, et al: Histopathologic and radiographic findings of the simple bone cyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 85: 619e625, 1998 Neville BW, Damm DD, Allen CM, Bouquot JE: Oral and maxillofacial pathology, Missouri: Saunders Elsevier, 631e633, 2009 Shigematsu H, Fujita K, Watanabe K: Atypical simple bone cyst of the mandible. Int J Oral Maxillofac Surg 23: 298e299, 1994 Suei Y, Taguchi A, Tanimoto K: Simple bone cyst of the jaws: evaluation of treatment outcome by review of 132 cases. J Oral Maxillofac Surg 65: 918e923, 2007 Xanthinaki AA, Choupis KI, Tosios K, Pagkalos VA, Papanikolaou SI: Traumatic bone cyst of the mandible of possible iatrogenic origin: a case report and brief review of the literature. Head Face Med 2: 40, 2006