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Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm
YIJOM-3712; No of Pages 9
Int. J. Oral Maxillofac. Surg. 2017; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2017.05.024, available online at http://www.sciencedirect.com
Original Article
Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm
M. Hanisch1, D. Baumhoer2, S. Elges3, L. F. Fro¨hlich1, J. Kleinheinz1, S. Jung1 1
Department of Cranio-Maxillofacial Surgery, University Hospital Mu¨nster, Mu¨nster, Germany; 2Bone Tumour Reference Centre ¨ SAK Registry, Institute of Pathology, and DO University Hospital Basel, University of Basel, Basel, Switzerland; 3Gerhard-DomagkInstitute of Pathology, University Hospital Mu¨nster, Mu¨nster, Germany
M. Hanisch, D. Baumhoer, S. Elges, L.F. Fro¨hlich, J. Kleinheinz, S. Jung: Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm. Int. J. Oral Maxillofac. Surg. 2017; xxx: xxx– xxx. ã 2017 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Abstract. Sclerosing odontogenic carcinoma (SOC) is a primary intraosseous carcinoma of the jaws that has been listed as a separate entity for the first time in the latest version of the World Health Organization classification of Head and Neck Tumours (2017). The aim of this study was to analyse and interpret the existing literature on SOC in the context of a clinical case treated in the authors’ department. A systematic search of the PubMed database was performed in accordance with the PRISMA guidelines, yielding nine cases of SOC reported so far. In summary, characteristic clinical and radiological features of SOC include asymptomatic swelling, location predominantly in the mandible, tumour primarily lytic in appearance, presence of cortical bone destruction, and lack of metastatic spread. Due to the rarity of the disease, close collaboration between oral/maxillofacial surgeons and pathologists is crucial to avoid misdiagnosis. With complete excision, no recurrence of SOC should be expected.
Sclerosing odontogenic carcinoma (SOC) is defined as a primary intraosseous carcinoma of the jaws with a bland cytology, a markedly sclerotic stroma, and a locally aggressive and infiltrative growth pattern1. Whether SOC should be regarded as a separate entity or as part of a morphological spectrum of other odontogenic carcinomas remains controversial2. Nevertheless, in the latest version of the World Health Organi0901-5027/000001+09
zation Classification of Head and Neck Tumours (2017), SOC has been introduced as an independent subtype. SOC was first described by Landwehr and Allen in 19963. Twelve years later, Koutlas et al. reported three cases of SOC4, including the case reported by Landwehr and Allen. Histologically, SOC appears as single file thin cords, nests, and strands of epithelium in a
Key words: oral oncology; sclerosing odontogenic carcinoma; odontogenic tumour; immunohistochemistry; WHO Classification of Head and Neck Tumours 2017. Accepted for publication 31 May 2017
densely sclerotic stroma1. Differential diagnoses include metastatic carcinoma, primary intraosseous carcinoma, clear cell odontogenic carcinoma, and ameloblastic carcinoma5, as well as desmoplastic ameloblastoma, intraosseous mucoepidermoid carcinoma, and (the epithelium-rich variant of) odontogenic fibroma6. The aim of this study was to analyse and interpret the existing literature on SOC
ã 2017 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
YIJOM-3712; No of Pages 9
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with a focus on clinical, radiological, and histological features and therapeutic options. The results are discussed in the context of a recent clinical case. Materials and methods
A literature search of the PubMed database was performed. The following medical subject heading (MeSH) terms were used: ‘‘odontogenic sclerotic carcinoma’’, ‘‘sclerosing odontogenic carcinoma’’, ‘‘sclerotic odontogenic carcinoma’’. In accordance with the PRISMA guidelines7, all records identified as PubMed database entries were surveyed for duplicates. After removing duplicates, the abstracts of the remaining records were screened against the exclusion criteria (no case reported, not describing SOC). Subsequently, full-text articles were retrieved and assessed for eligibility. The references included in the full-text articles were reviewed for further case reports on SOC. Articles identified in the references search were retrieved and the full-texts assessed for eligibility. Finally, all records were analysed according to the aims of this study. The flow diagram of the literature search is given in Fig. 1. Furthermore, a clinical case provided by the authors is reported. Results
The first literature search of the PubMed database with the key words listed above identified 39 entries. After removing duplicates; 20 articles remained. The abstracts of these articles were screened; following which nine articles were excluded: four were letters to the editor and nine did not describe reports of SOC. Finally; seven full-text articles were selected. Screening of the reference lists of these seven selected articles led to the identification of a further 12 articles; of which five were rejected either due to not representing case reports (n = 3) or not describing SOC (n = 2). Two of the remaining seven articles from the reference search were clearly appropriate for inclusion; suitability for inclusion was unclear for five questionable articles. In total; nine reports representing SOC could be included in the summary below (Table 1). The same cases were reported several times by different authors. Wood et al. 20162 and Gordon et al. 20158 reported the same case; as did Koutlas et al. in 20059 and 20084; who also reported other cases that had already been reported by Chaisuparat et al. in 200610 or Landwehr and Allen in
Fig. 1. Flow diagram of the literature search and identification of articles on sclerosing odontogenic carcinoma (SOC), according to the PRISMA guidelines.
19963. One case was reported two times by different authors2,8.
Demographic characteristics
Overall, the cases of nine patients were reported, five of whom were female2–4,6,8– 11 and four of whom were male4,5,9,12,13. The female patients ranged in age from 31 to 73 years (mean 48 years) and the male patients from 42 to 72 years (mean 58.75 years).
Treatment
The most frequently described treatment was surgical resection alone (n = 3)2– 4,8,9,12 , followed by resection in combination with neck dissection (n = 2)4,9,11. Resection in combination with neck dissection and radiation was performed once5, as was resection in combination with radiation4,9,10 and enucleation6. Multiple surgeries were necessary only in one patient, due to recurrent disease13. Clinical outcome and follow-up
Location affected
The most frequently affected location was the mandible (n = 6)3–6,9,11,13. The maxilla was involved in the remaining three cases2,4,8–10,12.
Clinical signs and symptoms
The most commonly reported clinical symptom of SOC was swelling (n = 6)2,4–6,8,9,11,12, followed by paresthesia (n = 2)4,9,13, pain (n = 1)3,4, and sensitivity of a tooth12. One case was completely asymptomatic4,9,10.
No recurrence after initial surgery was reported for eight cases2–6,8–12. Only one author reported a local recurrence, which occurred at 8 months after initial surgery; no further recurrence was recorded 15 months after the second surgery13. Follow-up ranged from 12 to 72 months (mean 36.5 months). Initial diagnosis
Initially, the tumours were interpreted as squamous cell carcinoma12, primary intraosseous carcinoma4,9,10, primary intraosseous carcinoma with dentinoid11,
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Author
Sex, age (years)
Initial diagnosis
Wood et al. 20162 (Gordon et al. 20158) Tan et al. 20146
F, 43
Adenocarcinoma
F, 31
None
Saxena et al. 20135
M, 42
Metastatic tumour, PIOC, ameloblastic carcinoma, CCOC
Hussain et al. 201312
M, 54
SCC
Irie´ et al. 201013
M, 67
BFOL
Ide et al. 200911
F, 47
PIOC with dentinoid
Chaisuparat et al. 200610 (Koutlas et al. 20059, Koutlas et al. 20084) Koutlas et al. 20059 (Koutlas et al. 20084)
F, 73
PIOC
M, 72
Odontogenic tumour
Landwehr and Allen 19963 (Koutlas et al. 20084)
F, 46
SOC
Symptoms, site Asymptomatic firm lump on right anterior hard palate Lump at the site of the lower right first permanent molar (tooth extracted 10 years before) Extraoral firm to bony-hard swelling in the left parasymphyseal area involving the canine and premolar region Sensitivity of the upper right canine tooth, expansile swelling with a firm consistency Paresthesia in the left mental region
History of a 2-cm mass on the left lower lingual gingiva surrounding the roots of the second premolar and first molar Asymptomatic
Enlarging left mandibular mass in the area of the canine and the premolars with a long duration, mental nerve paresthesia Pain in the right mandible
Treatment
Follow-up/outcome
Radiology
Maxillectomy with wide margin Enucleation
No recurrence after 17 months No recurrence after 12 months
Soft tissue mass arising from the right hard palate with no bone destruction Well-circumscribed round radiolucent lesion measuring 1 cm, with scattered specks of radiopacity and a distinct sclerotic peripheral margin Well-defined unilocular lytic lesion extending from the left mandibular central incisor to the second premolar, with a smooth outline and no sclerotic margins
Hemimandibulectomy, radical neck dissection, radiotherapy
No recurrence, 10 months
Resection with 5-mm margins
No recurrence, 19 months
First surgery: curettage Second surgery: segmental mandibulectomy and chemotherapy Mandibular resection, cervical lymph node dissection
Recurrence 8 months after first surgery No recurrence 15 months after second surgery No recurrence, 6 years
Unilocular radiolucency with sclerotic inferior borders
Right hemimaxillectomy, radiotherapy
No recurrence, 3.5 years
Radiolucency with ill-defined margins showing bone destruction and perforation of the cortical bone in the right maxilla
Extensive surgery, ipsilateral neck dissection
No recurrence, 5 years
Radiolucency affecting the area of the mandibular canine and premolars
Resection with 1-cm margins
No recurrence, 5 years
Poorly defined osteolytic lesion at the right angle of the mandible, buccal cortical plate perforated, lingual cortical plate thinned
Well-demarcated radiolucency related to the upper right lateral incisor and canine teeth with loss of the lamina dura around the roots, resorption of the canine tooth Focally expansile lesion with thinning or disappearance of the buccal side of the cortical bone and contained admixed radiolucent and radio-opaque areas
BFOL, benign fibro-osseous lesion; CCOC, clear cell odontogenic carcinoma; F, female; M, male; PIOC, primary intraosseous odontogenic carcinoma; SCC, squamous cell carcinoma; SOC, sclerosing odontogenic carcinoma.
Sclerosing odontogenic carcinoma
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
Table 1. Analysis of the cases of sclerosing odontogenic carcinoma identified in the literature and fulfilling the eligibility criteria.
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odontogenic tumour not otherwise specified4,9, adenocarcinoma2,8, or as a benign fibro-osseous lesion13. In one case, various differential diagnoses were described5, while Landwehr and Allen3 regarded the tumour as a separate entity for the first time and suggested the term ‘sclerosing odontogenic tumour’. Radiology
In all reported cases, the appearance in radiographs and computed tomography (CT) scans was primarily lytic. Additionally, mixed radio-opaque or sclerotic areas were seen in two cases6,11. Perforation of the cortical bone was described in four cases3,4,9,12,13. Histopathology
Perineural or intraneural infiltration was described as a striking feature in seven cases2–5,8–10,12,13. Additionally, soft tissue and/or vascular infiltration was reported by some authors4,5,9,10. Two authors found no signs of perineural, soft tissue, or vascular infiltration6,11. Necrosis was found by Saxena et al.5. A dense, fibrous or sclerotic stroma was reported in seven cases2–6,8–10,12, while one author noticed the deposition of dentinoid11. Immunohistochemistry
Immunohistochemical results were reported for eight of the cases analysed2–6,8–10,12,13. Staining for P63 was positive in seven of eight immunohistochemical analyses2–6,8– 10,13 . Staining was positive for CK5/6 in six of eight cases3–6,9,10,12, while only one author confirmed positive staining for CK613. A positive signal for CK19 was found in seven of eight cases2–4,6,8–10,12,13.
Fig. 2. Clinical intraoral view showing slight swelling in the premolar/molar region of the left mandible compared with the contralateral side.
formation with signs of active remodelling, but no signs of a neoplastic process. Upon first consultation in the clinic, the patient presented with a swelling in the premolar/molar region of the left mandible (Fig. 2). No perforation or ulceration of the soft tissue was observed and no hypoesthesia or paresthesia was noted. CT revealed ill-defined lytic osseous changes with expansion, erosion, and perforation, highly suspicious of a malignant tumour (Figs 3 and 4). Consequently, it was decided that an incisional biopsy should be performed under general anaesthesia. Histopathology revealed cords of epithelial tumour cells infiltrating preexisting lamellar bone (Figs 5 and 6). Immunohistochemistry confirmed the
epithelial nature of the neoplasm, demonstrating consistent and strong positivity for CK5/6, p40, p63, and MNF116 (Figs 7– 10). The findings were initially interpreted as low-grade squamous cell carcinoma. Based on this diagnosis, tumour staging was performed without any signs of metastasis or suspicious lymph nodes. Another panoramic radiograph and a postero-anterior radiograph were obtained and these showed that the tumour had increased considerably in size over the intervening 3 months. A left hemimandibulectomy including radical ipsilateral neck dissection was performed under general anaesthesia. Due to the uncertain histological classification, a primary reconstruction was done using an individual computer-aided design and manufacturing
Molecular genetic analyses
Molecular studies (fluorescence in situ hybridization (FISH)) were performed in only two cases2,6, and neither case showed EWS RNA-binding protein 1 (EWSR1) gene rearrangements. Case report
A 60-year-old male was referred to the craniomaxillofacial surgery clinic in May 2016. Four weeks earlier, his dentist had removed the left lower third molar. The dentist reported a conspicuous radiolucency with a defined border surrounding the tooth in the panoramic radiograph. A biopsy of this area showed fibrotic tissue, giant cells, and reactive new bone
Fig. 3. Three-dimensional reconstructed CT image demonstrating an irregular bony structure and slight swelling of the mandible.
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Sclerosing odontogenic carcinoma
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Fig. 4. Coronal CT scan revealing a non-corticated, ill-defined lytic change in the left mandible.
(CAD/CAM) endoprosthesis, including replacement of the temporomandibular joint. After verification of the final diagnosis, reconstruction with a microvascular fibula transplant is planned in a second operation.
Given the unusual histopathological findings showing relatively sparse, scattered epithelial cords and nests, the case ¨ SAK registry (registry was sent to the DO of the German–Austrian–Swiss Working Group on Tumours of the Face and Jaws)
for a second opinion; the final diagnosis was SOC with perineural infiltration (Fig. 11). No lymph node metastases were found and all margins were tumour-free. The patient is still free of disease 9 months after surgery.
Fig. 5. Histology of the resection specimen, demonstrating infiltration of the pre-existing lamellar bone (haematoxylin and eosin, 2.5).
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Fig. 6. Higher magnification image of the resection specimen, showing small epithelial tumour cell cords and nests (haematoxylin and eosin, 20).
Fig. 7. Immunohistochemistry for the expression of CK5/6, revealing uniform positivity in the tumour cells (20).
Fig. 8. Immunohistochemistry for the expression of p40, revealing uniform positivity in the tumour cells (20).
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Sclerosing odontogenic carcinoma
Fig. 9. Immunohistochemistry for the expression of p63, revealing uniform positivity in the tumour cells (10).
Discussion
Only nine reported cases2–6,8–13 of SOC and five disputable reports14–18 underline the scarcity of the data on this tumour. Five female and four male patients were reported in these nine articles, indicating the absence of a significant gender disparity. No patient younger than 31 years was reported and only two patients were over 70 years of age, so SOC seems to be most common in the middle-aged generation, with a calculated average age of 52.8 years.
According to Wood et al.2, SOC is a low-grade malignant tumour that should be treated in a similar way to calcifying epithelial odontogenic tumours. Only one author described a recurrence after first surgery13, and no recurrence was reported after second surgery. Metastases were not noted in the literature either2. Saxena et al. assumed that the sclerosing nature of the stroma may contribute to the lack of apparent distant metastatic potential5. Recurrence, as reported by Irie´ et al.13, could be the result of incomplete resection. Hence, resection with 5-mm margins
Fig. 10. Immunohistochemistry for the expression of MNF116, revealing uniform positivity in the tumour cells (10).
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in combination with periodic follow-up, as recommended by Hussain et al.12, could be regarded as the treatment of choice. As no report to date has disclosed lymph node metastases, neck dissection might be unnecessary. Additional treatment, such as chemotherapy or radiotherapy, has not been recommended so far. Risk factors for developing SOC are unknown. In the case presented herein there was an impacted third molar, but a direct relationship with the development of this rare kind of tumour can of course not be proven. The exclusion of other tumours or metastases can be difficult, particularly with small core needle biopsies1. Desmoplastic ameloblastoma with characteristic compressed strands of tumour cells and heavily collagenized stroma exhibits features similar to those of SOC6. Nevertheless, perineural infiltration, as a typical feature of SOC, is not seen in desmoplastic ameloblastoma. Another differential diagnosis includes odontogenic fibroma, which is usually less collagenized and also lacks perineural infiltration. Since not all reported SOC cases showed perineural infiltration, this phenomenon alone cannot exclude all differential diagnoses. However, the radiology of SOC is generally far more aggressive than for desmoplastic ameloblastoma and odontogenic fibroma, and therefore should always be taken into account. The clinical features of SOC were mostly described as being non-specific and ranged from completely asymptomatic to cases where the patient had noticed a lump or swelling2,4–6,8–11. Pain was reported in only one case3,4. Although perineural infiltration was seen in seven SOC cases2–5,8–10,12,13, paresthesia was only verified in two cases4,9,13. Furthermore, Irie´ et al. described invasion into the mandibular canal13. In the case reported herein, perineural infiltration was also observed without paresthesia. Hence, asymptomatic swelling mainly in the mandible seems to be a common form of presentation for SOC, and pain and paresthesia are less common. Radiographically, SOC most commonly appears lytic. Additionally, frequent cortical bone destruction with thinning or perforation is described3,4,9,10,12,13, while sclerotic margins are less common6,11. Root resorption was only reported by Hussain et al.12. Histopathologically, SOC shows cords, nests, and strands of epithelial cells embedded in a densely sclerotic stroma. Cytologically, individual epithelial cells appear bland or slightly hyperchromatic with infrequent mitoses. The cytoplasm
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Fig. 11. Immunohistochemistry for the expression of S100, showing perineural infiltration by the tumour cells (20).
may show vacuolation or partial clearing1. Besides perineural infiltration, which was reported in most cases of SOC2–5,8–10,12,13, soft tissue or vascular infiltration was also reported4,5,9,10. However, a lack of perineural, soft tissue, and vascular infiltration can occur6,11. Necrosis was only mentioned by Saxena et al.5. In summary, typical single file or thin cords, nests, and strands of epithelium in a densely sclerotic stroma, in association with perineural infiltration, seem to be the characteristic histopathological features of SOC. Immunohistochemically, epithelial cells generally stain positive for CK19, CK5/6, and p632–6,9–13, as validated by the case report presented. Up to now, no specific molecular alterations have been identified. Sclerosing odontogenic carcinoma might mimic a benign tumour. Thus, an interdisciplinary approach taking into consideration all clinical, radiological, and histopathological features is highly recommended to avoid misdiagnosis and incorrect treatment.
Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Competing interests
The authors declare that they have no competing interests.
Ethical approval
Ethical approval for this study was obtained from the ethics review commit¨ rztekammer tee, Ethikkommission der A Westfalen-Lippe und der Westfa¨lischen Wilhelms-Universita¨t, Mu¨nster, Germany (Ref. No. 2017-051-f-N). Patient consent
Written informed consent to participate was obtained from the patient. Acknowledgement. We acknowledge Mrs Mu¨nster-Erkeling for the clinical photography.
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sive odontogenic neoplasm without apparent metastatic potential. Am J Surg Pathol 2008;32:1613–9. 5. Saxena S, Kumar S, Rawat S, Arun Kumar KV. An indolent swelling of the parasymphyseal area. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:528–33. 6. Tan SH, Yeo JF, Kheem Pang BN, Petersson F. An intraosseous sclerosing odontogenic tumor predominantly composed of epithelial cells: relation to (so-called) sclerosing odontogenic carcinoma and epithelial-rich central odontogenic fibroma. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;118:119–25. 7. Moher D, Liberati A, Tetzlaff J, Altmann DG, PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 2009;6: e1000097. 8. Gordon P, Morrison J, Wood A, Conn B. A case report of sclerosing odontogenic carcinoma of the hard palate. Br J Oral Maxillofac Surg 2015;53:e49. 9. Koutlas IG, Warnock G, Manivel JC. ‘‘Intraosseous’’ sclerosing carcinoma of possible odontogenic origin: report of two cases. Oral Surg Oral Med Oral Pathol Oral Radiol 2005;100:187. 10. Chaisuparat R, Coletti D, Kolokythas A, Ord RA, Nikitakis NG. Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathologic study of six new cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101: 194–200. 11. Ide F, Kikuchi K, Kusama K. Sclerosing odontogenic carcinoma. Pathol Int 2011; 61:259–61. 12. Hussain O, Rendon AT, Orr RL, Speight PM. Sclerosing odontogenic carcinoma in the maxilla: a rare primary intraosseous carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:e283–6. 13. Irie´ T, Ogawa I, Takata T, Toyosawa S, Saito N, Akiba M, Isobe T, Hokazono C, Tachikawa T, Suzuki Y. Sclerosing odontogenic carcinoma with benign fibro-osseous lesion of the mandible: an extremely rare case report. Pathol Int 2010;60:694–700. 14. Cercadillo-Ibarguren I, Berini-Ayte´s L, Marco-Molina V, Gay-Escoda C. Locally aggressive central odontogenic fibroma associated to an inflammatory cyst: a clinical, histological and immunohistochemical study. J Oral Pathol Med 2006;35:513–6. 15. Bennett JH, Jones J, Speight PM. Odontogenic squamous cell carcinoma with osseous metaplasia. J Oral Pathol Med 1993;22: 286–8. 16. Gu¨nhan O, Gu¨rbu¨zer B, Gardner DG, Demiriz M, Finci R. A central odontogenic fibroma exhibiting pleomorphic fibroblasts and numerous calcifications. Br J Oral Maxillofac Surg 1991;29:42–3. 17. Jones GM, Eveson JW, Shepherd JP. Central odontogenic fibroma: a report of two controversial cases illustrating diagnostic
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Address: Marcel Hanisch Department of Cranio-Maxillofacial Surgery University Hospital Mu¨nster Albert-Schweitzer-Campus 1 Geba¨ude W 30
D-48149 Mu¨nster Germany Tel: +49 (0)2 51/83-4 70 02 Fax: +49 (0)2 51/83-4 71 84 E-mail: [email protected]
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Int. J. Oral Maxillofac. Surg. 2017; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2017.05.024, available online at http://www.sciencedirect.com
Original Article
Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm
M. Hanisch1, D. Baumhoer2, S. Elges3, L. F. Fro¨hlich1, J. Kleinheinz1, S. Jung1 1
Department of Cranio-Maxillofacial Surgery, University Hospital Mu¨nster, Mu¨nster, Germany; 2Bone Tumour Reference Centre ¨ SAK Registry, Institute of Pathology, and DO University Hospital Basel, University of Basel, Basel, Switzerland; 3Gerhard-DomagkInstitute of Pathology, University Hospital Mu¨nster, Mu¨nster, Germany
M. Hanisch, D. Baumhoer, S. Elges, L.F. Fro¨hlich, J. Kleinheinz, S. Jung: Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm. Int. J. Oral Maxillofac. Surg. 2017; xxx: xxx– xxx. ã 2017 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Abstract. Sclerosing odontogenic carcinoma (SOC) is a primary intraosseous carcinoma of the jaws that has been listed as a separate entity for the first time in the latest version of the World Health Organization classification of Head and Neck Tumours (2017). The aim of this study was to analyse and interpret the existing literature on SOC in the context of a clinical case treated in the authors’ department. A systematic search of the PubMed database was performed in accordance with the PRISMA guidelines, yielding nine cases of SOC reported so far. In summary, characteristic clinical and radiological features of SOC include asymptomatic swelling, location predominantly in the mandible, tumour primarily lytic in appearance, presence of cortical bone destruction, and lack of metastatic spread. Due to the rarity of the disease, close collaboration between oral/maxillofacial surgeons and pathologists is crucial to avoid misdiagnosis. With complete excision, no recurrence of SOC should be expected.
Sclerosing odontogenic carcinoma (SOC) is defined as a primary intraosseous carcinoma of the jaws with a bland cytology, a markedly sclerotic stroma, and a locally aggressive and infiltrative growth pattern1. Whether SOC should be regarded as a separate entity or as part of a morphological spectrum of other odontogenic carcinomas remains controversial2. Nevertheless, in the latest version of the World Health Organi0901-5027/000001+09
zation Classification of Head and Neck Tumours (2017), SOC has been introduced as an independent subtype. SOC was first described by Landwehr and Allen in 19963. Twelve years later, Koutlas et al. reported three cases of SOC4, including the case reported by Landwehr and Allen. Histologically, SOC appears as single file thin cords, nests, and strands of epithelium in a
Key words: oral oncology; sclerosing odontogenic carcinoma; odontogenic tumour; immunohistochemistry; WHO Classification of Head and Neck Tumours 2017. Accepted for publication 31 May 2017
densely sclerotic stroma1. Differential diagnoses include metastatic carcinoma, primary intraosseous carcinoma, clear cell odontogenic carcinoma, and ameloblastic carcinoma5, as well as desmoplastic ameloblastoma, intraosseous mucoepidermoid carcinoma, and (the epithelium-rich variant of) odontogenic fibroma6. The aim of this study was to analyse and interpret the existing literature on SOC
ã 2017 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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with a focus on clinical, radiological, and histological features and therapeutic options. The results are discussed in the context of a recent clinical case. Materials and methods
A literature search of the PubMed database was performed. The following medical subject heading (MeSH) terms were used: ‘‘odontogenic sclerotic carcinoma’’, ‘‘sclerosing odontogenic carcinoma’’, ‘‘sclerotic odontogenic carcinoma’’. In accordance with the PRISMA guidelines7, all records identified as PubMed database entries were surveyed for duplicates. After removing duplicates, the abstracts of the remaining records were screened against the exclusion criteria (no case reported, not describing SOC). Subsequently, full-text articles were retrieved and assessed for eligibility. The references included in the full-text articles were reviewed for further case reports on SOC. Articles identified in the references search were retrieved and the full-texts assessed for eligibility. Finally, all records were analysed according to the aims of this study. The flow diagram of the literature search is given in Fig. 1. Furthermore, a clinical case provided by the authors is reported. Results
The first literature search of the PubMed database with the key words listed above identified 39 entries. After removing duplicates; 20 articles remained. The abstracts of these articles were screened; following which nine articles were excluded: four were letters to the editor and nine did not describe reports of SOC. Finally; seven full-text articles were selected. Screening of the reference lists of these seven selected articles led to the identification of a further 12 articles; of which five were rejected either due to not representing case reports (n = 3) or not describing SOC (n = 2). Two of the remaining seven articles from the reference search were clearly appropriate for inclusion; suitability for inclusion was unclear for five questionable articles. In total; nine reports representing SOC could be included in the summary below (Table 1). The same cases were reported several times by different authors. Wood et al. 20162 and Gordon et al. 20158 reported the same case; as did Koutlas et al. in 20059 and 20084; who also reported other cases that had already been reported by Chaisuparat et al. in 200610 or Landwehr and Allen in
Fig. 1. Flow diagram of the literature search and identification of articles on sclerosing odontogenic carcinoma (SOC), according to the PRISMA guidelines.
19963. One case was reported two times by different authors2,8.
Demographic characteristics
Overall, the cases of nine patients were reported, five of whom were female2–4,6,8– 11 and four of whom were male4,5,9,12,13. The female patients ranged in age from 31 to 73 years (mean 48 years) and the male patients from 42 to 72 years (mean 58.75 years).
Treatment
The most frequently described treatment was surgical resection alone (n = 3)2– 4,8,9,12 , followed by resection in combination with neck dissection (n = 2)4,9,11. Resection in combination with neck dissection and radiation was performed once5, as was resection in combination with radiation4,9,10 and enucleation6. Multiple surgeries were necessary only in one patient, due to recurrent disease13. Clinical outcome and follow-up
Location affected
The most frequently affected location was the mandible (n = 6)3–6,9,11,13. The maxilla was involved in the remaining three cases2,4,8–10,12.
Clinical signs and symptoms
The most commonly reported clinical symptom of SOC was swelling (n = 6)2,4–6,8,9,11,12, followed by paresthesia (n = 2)4,9,13, pain (n = 1)3,4, and sensitivity of a tooth12. One case was completely asymptomatic4,9,10.
No recurrence after initial surgery was reported for eight cases2–6,8–12. Only one author reported a local recurrence, which occurred at 8 months after initial surgery; no further recurrence was recorded 15 months after the second surgery13. Follow-up ranged from 12 to 72 months (mean 36.5 months). Initial diagnosis
Initially, the tumours were interpreted as squamous cell carcinoma12, primary intraosseous carcinoma4,9,10, primary intraosseous carcinoma with dentinoid11,
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Author
Sex, age (years)
Initial diagnosis
Wood et al. 20162 (Gordon et al. 20158) Tan et al. 20146
F, 43
Adenocarcinoma
F, 31
None
Saxena et al. 20135
M, 42
Metastatic tumour, PIOC, ameloblastic carcinoma, CCOC
Hussain et al. 201312
M, 54
SCC
Irie´ et al. 201013
M, 67
BFOL
Ide et al. 200911
F, 47
PIOC with dentinoid
Chaisuparat et al. 200610 (Koutlas et al. 20059, Koutlas et al. 20084) Koutlas et al. 20059 (Koutlas et al. 20084)
F, 73
PIOC
M, 72
Odontogenic tumour
Landwehr and Allen 19963 (Koutlas et al. 20084)
F, 46
SOC
Symptoms, site Asymptomatic firm lump on right anterior hard palate Lump at the site of the lower right first permanent molar (tooth extracted 10 years before) Extraoral firm to bony-hard swelling in the left parasymphyseal area involving the canine and premolar region Sensitivity of the upper right canine tooth, expansile swelling with a firm consistency Paresthesia in the left mental region
History of a 2-cm mass on the left lower lingual gingiva surrounding the roots of the second premolar and first molar Asymptomatic
Enlarging left mandibular mass in the area of the canine and the premolars with a long duration, mental nerve paresthesia Pain in the right mandible
Treatment
Follow-up/outcome
Radiology
Maxillectomy with wide margin Enucleation
No recurrence after 17 months No recurrence after 12 months
Soft tissue mass arising from the right hard palate with no bone destruction Well-circumscribed round radiolucent lesion measuring 1 cm, with scattered specks of radiopacity and a distinct sclerotic peripheral margin Well-defined unilocular lytic lesion extending from the left mandibular central incisor to the second premolar, with a smooth outline and no sclerotic margins
Hemimandibulectomy, radical neck dissection, radiotherapy
No recurrence, 10 months
Resection with 5-mm margins
No recurrence, 19 months
First surgery: curettage Second surgery: segmental mandibulectomy and chemotherapy Mandibular resection, cervical lymph node dissection
Recurrence 8 months after first surgery No recurrence 15 months after second surgery No recurrence, 6 years
Unilocular radiolucency with sclerotic inferior borders
Right hemimaxillectomy, radiotherapy
No recurrence, 3.5 years
Radiolucency with ill-defined margins showing bone destruction and perforation of the cortical bone in the right maxilla
Extensive surgery, ipsilateral neck dissection
No recurrence, 5 years
Radiolucency affecting the area of the mandibular canine and premolars
Resection with 1-cm margins
No recurrence, 5 years
Poorly defined osteolytic lesion at the right angle of the mandible, buccal cortical plate perforated, lingual cortical plate thinned
Well-demarcated radiolucency related to the upper right lateral incisor and canine teeth with loss of the lamina dura around the roots, resorption of the canine tooth Focally expansile lesion with thinning or disappearance of the buccal side of the cortical bone and contained admixed radiolucent and radio-opaque areas
BFOL, benign fibro-osseous lesion; CCOC, clear cell odontogenic carcinoma; F, female; M, male; PIOC, primary intraosseous odontogenic carcinoma; SCC, squamous cell carcinoma; SOC, sclerosing odontogenic carcinoma.
Sclerosing odontogenic carcinoma
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
Table 1. Analysis of the cases of sclerosing odontogenic carcinoma identified in the literature and fulfilling the eligibility criteria.
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odontogenic tumour not otherwise specified4,9, adenocarcinoma2,8, or as a benign fibro-osseous lesion13. In one case, various differential diagnoses were described5, while Landwehr and Allen3 regarded the tumour as a separate entity for the first time and suggested the term ‘sclerosing odontogenic tumour’. Radiology
In all reported cases, the appearance in radiographs and computed tomography (CT) scans was primarily lytic. Additionally, mixed radio-opaque or sclerotic areas were seen in two cases6,11. Perforation of the cortical bone was described in four cases3,4,9,12,13. Histopathology
Perineural or intraneural infiltration was described as a striking feature in seven cases2–5,8–10,12,13. Additionally, soft tissue and/or vascular infiltration was reported by some authors4,5,9,10. Two authors found no signs of perineural, soft tissue, or vascular infiltration6,11. Necrosis was found by Saxena et al.5. A dense, fibrous or sclerotic stroma was reported in seven cases2–6,8–10,12, while one author noticed the deposition of dentinoid11. Immunohistochemistry
Immunohistochemical results were reported for eight of the cases analysed2–6,8–10,12,13. Staining for P63 was positive in seven of eight immunohistochemical analyses2–6,8– 10,13 . Staining was positive for CK5/6 in six of eight cases3–6,9,10,12, while only one author confirmed positive staining for CK613. A positive signal for CK19 was found in seven of eight cases2–4,6,8–10,12,13.
Fig. 2. Clinical intraoral view showing slight swelling in the premolar/molar region of the left mandible compared with the contralateral side.
formation with signs of active remodelling, but no signs of a neoplastic process. Upon first consultation in the clinic, the patient presented with a swelling in the premolar/molar region of the left mandible (Fig. 2). No perforation or ulceration of the soft tissue was observed and no hypoesthesia or paresthesia was noted. CT revealed ill-defined lytic osseous changes with expansion, erosion, and perforation, highly suspicious of a malignant tumour (Figs 3 and 4). Consequently, it was decided that an incisional biopsy should be performed under general anaesthesia. Histopathology revealed cords of epithelial tumour cells infiltrating preexisting lamellar bone (Figs 5 and 6). Immunohistochemistry confirmed the
epithelial nature of the neoplasm, demonstrating consistent and strong positivity for CK5/6, p40, p63, and MNF116 (Figs 7– 10). The findings were initially interpreted as low-grade squamous cell carcinoma. Based on this diagnosis, tumour staging was performed without any signs of metastasis or suspicious lymph nodes. Another panoramic radiograph and a postero-anterior radiograph were obtained and these showed that the tumour had increased considerably in size over the intervening 3 months. A left hemimandibulectomy including radical ipsilateral neck dissection was performed under general anaesthesia. Due to the uncertain histological classification, a primary reconstruction was done using an individual computer-aided design and manufacturing
Molecular genetic analyses
Molecular studies (fluorescence in situ hybridization (FISH)) were performed in only two cases2,6, and neither case showed EWS RNA-binding protein 1 (EWSR1) gene rearrangements. Case report
A 60-year-old male was referred to the craniomaxillofacial surgery clinic in May 2016. Four weeks earlier, his dentist had removed the left lower third molar. The dentist reported a conspicuous radiolucency with a defined border surrounding the tooth in the panoramic radiograph. A biopsy of this area showed fibrotic tissue, giant cells, and reactive new bone
Fig. 3. Three-dimensional reconstructed CT image demonstrating an irregular bony structure and slight swelling of the mandible.
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Sclerosing odontogenic carcinoma
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Fig. 4. Coronal CT scan revealing a non-corticated, ill-defined lytic change in the left mandible.
(CAD/CAM) endoprosthesis, including replacement of the temporomandibular joint. After verification of the final diagnosis, reconstruction with a microvascular fibula transplant is planned in a second operation.
Given the unusual histopathological findings showing relatively sparse, scattered epithelial cords and nests, the case ¨ SAK registry (registry was sent to the DO of the German–Austrian–Swiss Working Group on Tumours of the Face and Jaws)
for a second opinion; the final diagnosis was SOC with perineural infiltration (Fig. 11). No lymph node metastases were found and all margins were tumour-free. The patient is still free of disease 9 months after surgery.
Fig. 5. Histology of the resection specimen, demonstrating infiltration of the pre-existing lamellar bone (haematoxylin and eosin, 2.5).
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Fig. 6. Higher magnification image of the resection specimen, showing small epithelial tumour cell cords and nests (haematoxylin and eosin, 20).
Fig. 7. Immunohistochemistry for the expression of CK5/6, revealing uniform positivity in the tumour cells (20).
Fig. 8. Immunohistochemistry for the expression of p40, revealing uniform positivity in the tumour cells (20).
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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Sclerosing odontogenic carcinoma
Fig. 9. Immunohistochemistry for the expression of p63, revealing uniform positivity in the tumour cells (10).
Discussion
Only nine reported cases2–6,8–13 of SOC and five disputable reports14–18 underline the scarcity of the data on this tumour. Five female and four male patients were reported in these nine articles, indicating the absence of a significant gender disparity. No patient younger than 31 years was reported and only two patients were over 70 years of age, so SOC seems to be most common in the middle-aged generation, with a calculated average age of 52.8 years.
According to Wood et al.2, SOC is a low-grade malignant tumour that should be treated in a similar way to calcifying epithelial odontogenic tumours. Only one author described a recurrence after first surgery13, and no recurrence was reported after second surgery. Metastases were not noted in the literature either2. Saxena et al. assumed that the sclerosing nature of the stroma may contribute to the lack of apparent distant metastatic potential5. Recurrence, as reported by Irie´ et al.13, could be the result of incomplete resection. Hence, resection with 5-mm margins
Fig. 10. Immunohistochemistry for the expression of MNF116, revealing uniform positivity in the tumour cells (10).
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in combination with periodic follow-up, as recommended by Hussain et al.12, could be regarded as the treatment of choice. As no report to date has disclosed lymph node metastases, neck dissection might be unnecessary. Additional treatment, such as chemotherapy or radiotherapy, has not been recommended so far. Risk factors for developing SOC are unknown. In the case presented herein there was an impacted third molar, but a direct relationship with the development of this rare kind of tumour can of course not be proven. The exclusion of other tumours or metastases can be difficult, particularly with small core needle biopsies1. Desmoplastic ameloblastoma with characteristic compressed strands of tumour cells and heavily collagenized stroma exhibits features similar to those of SOC6. Nevertheless, perineural infiltration, as a typical feature of SOC, is not seen in desmoplastic ameloblastoma. Another differential diagnosis includes odontogenic fibroma, which is usually less collagenized and also lacks perineural infiltration. Since not all reported SOC cases showed perineural infiltration, this phenomenon alone cannot exclude all differential diagnoses. However, the radiology of SOC is generally far more aggressive than for desmoplastic ameloblastoma and odontogenic fibroma, and therefore should always be taken into account. The clinical features of SOC were mostly described as being non-specific and ranged from completely asymptomatic to cases where the patient had noticed a lump or swelling2,4–6,8–11. Pain was reported in only one case3,4. Although perineural infiltration was seen in seven SOC cases2–5,8–10,12,13, paresthesia was only verified in two cases4,9,13. Furthermore, Irie´ et al. described invasion into the mandibular canal13. In the case reported herein, perineural infiltration was also observed without paresthesia. Hence, asymptomatic swelling mainly in the mandible seems to be a common form of presentation for SOC, and pain and paresthesia are less common. Radiographically, SOC most commonly appears lytic. Additionally, frequent cortical bone destruction with thinning or perforation is described3,4,9,10,12,13, while sclerotic margins are less common6,11. Root resorption was only reported by Hussain et al.12. Histopathologically, SOC shows cords, nests, and strands of epithelial cells embedded in a densely sclerotic stroma. Cytologically, individual epithelial cells appear bland or slightly hyperchromatic with infrequent mitoses. The cytoplasm
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Fig. 11. Immunohistochemistry for the expression of S100, showing perineural infiltration by the tumour cells (20).
may show vacuolation or partial clearing1. Besides perineural infiltration, which was reported in most cases of SOC2–5,8–10,12,13, soft tissue or vascular infiltration was also reported4,5,9,10. However, a lack of perineural, soft tissue, and vascular infiltration can occur6,11. Necrosis was only mentioned by Saxena et al.5. In summary, typical single file or thin cords, nests, and strands of epithelium in a densely sclerotic stroma, in association with perineural infiltration, seem to be the characteristic histopathological features of SOC. Immunohistochemically, epithelial cells generally stain positive for CK19, CK5/6, and p632–6,9–13, as validated by the case report presented. Up to now, no specific molecular alterations have been identified. Sclerosing odontogenic carcinoma might mimic a benign tumour. Thus, an interdisciplinary approach taking into consideration all clinical, radiological, and histopathological features is highly recommended to avoid misdiagnosis and incorrect treatment.
Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Competing interests
The authors declare that they have no competing interests.
Ethical approval
Ethical approval for this study was obtained from the ethics review commit¨ rztekammer tee, Ethikkommission der A Westfalen-Lippe und der Westfa¨lischen Wilhelms-Universita¨t, Mu¨nster, Germany (Ref. No. 2017-051-f-N). Patient consent
Written informed consent to participate was obtained from the patient. Acknowledgement. We acknowledge Mrs Mu¨nster-Erkeling for the clinical photography.
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Address: Marcel Hanisch Department of Cranio-Maxillofacial Surgery University Hospital Mu¨nster Albert-Schweitzer-Campus 1 Geba¨ude W 30
D-48149 Mu¨nster Germany Tel: +49 (0)2 51/83-4 70 02 Fax: +49 (0)2 51/83-4 71 84 E-mail: [email protected]
Please cite this article in press as: Hanisch M, et al. Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.05.024
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