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International Journal of Medical and Dental Case Reports (2021), Article ID INS161 380221, 3 Pages

C A S E R E P O R T

An interesting case of odontogenic keratocyst mimicking traumatic bone cystVishwas D. Kadam1, Govind Changule2, Lalitkumar P. Gade3, Sneha H. Choudhary3

1Department of Oral Medicine and Radiology, C.S.M.S.S. Dental College and Hospital, Aurangabad, Maharashtra, India, 2Department of Oral Pathology, SMBT Institute of Dental Sciences and Research Centre, Nandi Hills, Dhamangaon, Nashik, Maharashtra, India, 3Department of Oral Medicine and Radiology, Teerthanker Mahaveer Dental College and Research Centre, Moradabad, Uttar Pradesh, India

AbstractOdontogenic keratocyst (OKC) is the cyst arising from the cell rests of dental lamina. Due to its aggressive nature, high recurrence, and possible malignant transformation, OKC has been added to the benign odontogenic tumors category, with the new term of keratocystic odontogenic tumor. Since the clinical features and radiological appearance are not characteristic, this condition is often misdiagnosed. In the present article, we have reported a case of OKC in a 46-year-old man, which was initially misdiagnosed as traumatic bone cyst, who presented with the chief complaint of generalized sensitivity of teeth. Further, the clinical, radiological, and histological features of this entity, along with its surgical management, have been discussed in this article.

Keywords: Developmental cyst, keratocystic odontogenic tumor, odonotogenic keratocyst

Correspondence: Sneha H. Choudhary, Department of Oral Medicine and Radiology, Teerthanker Mahaveer Dental College and Research Centre, Moradabad, Uttar Pradesh, India. Phone: +91-8240731631. E-mail: snehachy1606@gmail.com

Received 06 January 2021; Accepted 10 February 2021

doi: 10.15713/ins.ijmdcr.154

How to cite the article: Kadam VD, Changule G, Gade LP, Choudhary SH. An interesting case of odontogenic keratocyst mimicking traumatic bone cyst. Int J Med Dent Case Rep 2021;7:1-3.

Introduction

Odontogenic keratocyst (OKC) was first explained by Phillipsen in 1956.[1] It is one of the most aggressive odontogenic cysts due to its relatively high recurrence rate, fast growth potential, and tendency to invade adjacent tissues.[2] In the latest World Health Organization (WHO) classification, the former OKC was added to the benign odontogenic tumors category and the new term is “keratocystic odontogenic tumor” (KCOT). The change in terminology was based on the observation that OKC behaves as a neoplasm and not like a benign cystic lesion.[3] Here, we present a case of OKC in a 46-year-old male patient.

Case Report

A 46-year-old male patient reported to the department of oral medicine and radiology with the chief complaint of sensitivity in all his teeth on hot and cold fluid intake. Although the sensitivity was generalized with all the teeth but were prominent with his maxillary and mandibular right side posterior teeth. The patient’s intraoral examination revealed cervical abrasion with maxillary and mandibular right posterior teeth. There were no signs of caries, fracture, swelling, or expansion in

the same region. The patient was advised intraoral periapical radiographs of maxillary and mandibular right posterior teeth region. On radiographic examination, to our surprise, we found a large, well defined, corticated, interdentally scalloped radiolucency at periapical regions of mandubular right premolars and molars (44, 45, 46, 47, and 48) [Figure 1]. The patient was then advised an orthopantomogram (OPG) and mandibular cross-sectional occlusal radiograph. The OPG revealed a single, well defined unilocular corticated radiolucency that showed interdental scalloping and was extending mesiodistally from periapical region of mandibular right second premolar (45) till periapical region of mandibular right third molar (48) and superoinferiorly from alveolar crest margin of mandibular right first and second molar (46, 47) till 0.5 cm above inferior border of mandible of size approximately 4 × 3.5 cm [Figure 2]. The mandibular occlusal radiograph showed no signs of buccal or lingual cortical expansion. During vitality test 45, 46, 47, and 48 responded positively to electric pulp testing. The fine-needle aspiration cytology revealed clear red colored fluid [Figure 3]. Based on all these findings, a provisional diagnosis of traumatic bone cyst with mandibular right posterior region was given and the patient was posted for surgery. Marsupialization with window formation was

Figure  6: Post-operative orthopantomogram after root canals treatment of 46 and 47

Figure 2: Orthopantomogram radiograph showing well defined, unilocular, scalloped, and corticated radiolucency

Figure 3: Fine-needle aspiration cytology showing presence of blood

Figure 4: Marsupialization with window formation

Figure 5: Histopathologic picture of the lesion

Kadam, et al. OKC mimicking traumatic bone cyst

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performed [Figure 4] and the excised tissue was sent for histopathological examination which revealed a cystic cavity which was lined by thin, uniform lining of stratified squamous epithelium; a surface layer of parakeratin with a spinous cell layer of 4–6 cells in thickness and a flat epithelial-fibrous tissue junction devoid of epithelial rete pegs. Furthermore, there was lack of inflammatory cell infiltrate [Figure 5]. All these findings were suggestive of OKC, and hence, the cyst was enucleated. After post-operative healing, root canal treatment of 46 and 47 was performed and a post-operative OPG was taken at 3 month interval which suggested healing at the site of the lesion [Figure 6]. The patient is under regular follow-up with no signs of recurrence till date.

Discussion

The OKC is one of the most aggressive odontogenic cysts due to its relatively high recurrence rate and tendency to invade

Figure 1: (a and b) Intraoral periapical radiograph showing interdental scalloped radiolucency

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OKC mimicking traumatic bone cyst Kadam, et al

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adjacent tissues.[4] OKC is developmental in nature which is generally thought to be derived from either the cell rests of dental lamina, or the basal cell layer of the surface epithelium or from the stellate reticulum of the enamel organ.[2] OKC has been renamed as “KCOT” by the WHO in 2005.[5] Ide et al., in 2003, were the first to describe the solid variant of KCOT.[4]

Mikulicz, in 1876, first described OKC as a part of a familial condition affecting the jaws, which was later named as “Cholesteatoma” in 1926.[6] Philipsen, in 1956, named it as OKC, whereas Pindborg and Hansen were the first to describe its aggressive nature.[7] Toller,[8] in 1967, considered it as a benign neoplasm on the basis of its clinical behavior. Later on, Ahlfors and others, in 1984, have classified it as a true benign cystic epithelial neoplasm.[9] Shear renamed it as keratocystoma, in 2003, which was later modified by Philipsen in 2005 as KCOT, which was accepted by the WHO.[6]

OKC is more common in males than females and occurs over a wide age range with peak in the second and third decades of life. However, in the present case, OKC has occurred in a 46-year-old male patient. OKC may occur in any part of the jaws, but is more common in posterior body of the mandible and ramus, as is seen in the present case. The epicenter of OKC is usually superior to the inferior alveolar nerve canal similar to the present case, where the lesion was above the inferior alveolar nerve canal on the right side. OKC often has no symptoms; however, in the present case, the patient had sensitivity in his teeth. Although, sensitivity may be due to the presence of cervical abrasion in the teeth in that region. In some cases, little swelling may be evident clinically.[3] The clinical feature and radiographic appearance of OKC are not characteristic which may lead to misdiagnosis especially when the lesion is in relation to a non-vital tooth.[10]

Radiographically, OKC can be observed as a multilocular or unilocular, well-defined corticated margin, rarely causing root resorption, except in basal cell nevus syndrome, or nevoid basal cell carcinoma, which may be associated with multiple keratocysts, along with cutaneous nevi, basal cell carcinomas, rib anomalies, among other manifestations of the syndrome characteristics.[11] In the present case, OKC appeared radiographically as a well-defined, corticated, and interdentally scalloped radiolucency in the periapical regions of 44, 45, 46, 47, and 48.

The various differential diagnosis that may be considered includes lateral periodontal cyst, traumatic bone cyst, central giant cell tumor, radicular cyst, ameloblastoma, a benign bone tumor, and adenomatoid odontogenic tumor.[10] Histologically, OKC shows an epithelial lining which consists of a uniform layer

of six to eight cells thick stratified squamous epithelium. Cystic lumen of OKC may contain either a clear liquid or it may be filled with a cheesy substance that consists of keratinaceous debris. Basal layer of epithelium consists of a palisaded layer of cuboidal or columnar epithelial cells, which are hyperchromatic. Small daughter cysts, cords, or islands of odontogenic epithelium (satellite cyst) may be observed within fibrous wall.[10]

The various treatment options for OKC include simple curettage, enucleation often in combination with cryotherapy or Carnoy`s solution, marsupialization, decompression and secondary enucleation, and resection (marginal or segmental). Decompression and marsupialization are the most commonly used procedures in case of a large mandibular and maxillary tumor since they can save the anatomical structures including adjacent teeth, maxillary sinus, or inferior alveolar nerve.[4] In the present, case also enucleation of the cystic lesion was done.

References

1. Gnanaselvi UP, Kamatchi D, Sekar K, Narayanan BS. Odontogenic keratocyst in anterior mandible: An interesting case report. J Indian Acad Dent Spec Res 2016;3:22-4.

2. Myoung H, Hong SP, Hong SD, Lee JI, Lim CY, Choung PH, et al. Odontogenic keratocyst: Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:328-33.

3. Reichart PA, Philipsen HP, Sciubba JJ. The new classification of head and neck tumours (WHO)-any changes. Oral Oncol 2006;42:757-8.

4. Siddique S, Varghese ST, Mathew T. Odontogenic keratocyst of the mandible-a case report. WJPMR 2016;2:213-5.

5. Belmehdi A, Chbicheb S, El Wady W. Odontogenic keratocyst tumor: A case report and literature review. Open J Stomatol 2016;6:171-8.

6. Kesidi S, Maloth KN, Kundoor VK, Thakur M. Unusual presentation of keratocystic odontogenic tumor: Two case reports. J Indian Acad Oral Med Radiol 2016;28:74-8.

7. Pindborg JJ, Hansen J. Studies on odontogenic cyst epithelium. 2. Clinical and roentgenologic aspects of odontogenic keratocysts. Acta Pathol Microbiol Scand 1963;58:283-94.

8. Nayak MT, Singh A, Singhvi A, Sharma R. Odontogenic keratocyst: What is in the name? J Nat Sci Biol Med 2013;4:282-5.

9. Ahlfors E, Larsson A, Sjogren S. The odontogenic keratocyst: A benign cystic tumor? J Oral Maxillofac Surg 1984;42:10-9.

10. Jardim EC, Rossi AC, Faverani LP, Ferreira GR, Ferreira MB, Vicentes LM, et al. Odontogenic keratocyst tumor: Report of two cases. Int J Odontostomat 2013;7:33-8.

11. Shear M, Speight P. Cysts of the Oral and Maxillofacial Region. 4th ed. Singapore: Blackwell Munksgaard; 2007. p. 223.

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