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Authors Dr. Jagadish V. Hosmani, Dr. Deepa Hugar, Dr. Ramakant S. Nayak

Abstract:
A case of a dentigerous cyst with sebaceous glands differentiation seen at the interface between the epithelium and connective tissue in a 13 year old female patient is reported here.

Introduction
Jaw cysts with sebaceous elements are rare and various interpretations of such cysts have been given in the literature. Some authors have preferred to consider these lesions as intraosseous dermoid cysts due to the virtue of sebaceous glands being dermal adnexal structures.1 Hofrath, Gorlin, and Spouge documented the occurrence of sebaceous glands in dentigerous cyst. 2,3,4 Here we report a case of dentigerous cyst with sebaceous differentiation in a 13 year old female.

Case History
A female patient aged 13 years reported to our institution with the chief complaint of a painless swelling in the lower left posterior tooth region since 6 months. Extraoral examination revealed slight facial asymmetry. Intraoral examination revealed single, lobulated, intraosseous swelling with obliteration of buccal vestibule with respect to primary mandibular second molar region. On palpation the swelling was firm and non tender. Primary mandibular second molar was carious and was grossly destructed.
Radiological examination by OPG revealed grossly decayed primary mandibular second molar showing resorption of roots. Well defined unilocular radiolucency around the crown of impacted mandibular second premolar was seen. The radiolucency extended from the distal surface of root of first premolar to the mesial surface of root of first permanent molar. Radiologically the lesion mimicked to be radicular cyst associated with primary mandibular second molar. (Figure 1)
A provisional diagnosis of dentigerous cyst was made. All routine lab investigations followed by FNAC were done. Red colored fluid was drawn. FNAC smear revealed sheets of RBCs and neurtrophils. Patient was referred to oral surgery where the lesion was surgically excised and specimen was subjected for histopathological examination.
Microscopically, the lesion showed 3-4 layered non keratinized stratified squamous epithelium with focal areas of hyperplasia. The underlying connective tissue was inflamed characterized by diffuse infiltration of lymphocytes, plasma cells and macrophages. Focal sebaceous cells were identified in association with the connective tissue. (Figure 2 & 3)
A diagnosis of infected dentigerous cyst with sebaceous differentiation within the connective tissue was given.

Discussion:
Sebaceous glands are holocrine glands that produce an oily product called sebum. Normal sebaceous glands typically are found in conjunction with hair follicles as part of a pilosebaceous unit. Therefore, the distribution of normal sebaceous glands roughly corresponds to that of hair that is, widely distributed throughout the skin. 4,5 Within the oral cavity, sebaceous glands may present as small, subtle, yellowish spots called Fordyce granules, which exhibit a predilection for the buccal mucosa. Estimated to occur in more than 80% of the population,6 Fordyce granules are such a frequent finding that they can be considered a normal anatomic variation rather than an ectopic phenomenon.7 In addition, aberrant or ectopic sebaceous glands have been described in various locations, including the parotid gland, orbit, larynx, and esophagus. Proposed theories to explain the occurrence of aberrant or ectopic sebaceous glands include development from sequestered multipotent epithelial cells and metaplasia of existing mature epithelium.4

Intraosseous jaw cysts with sebaceous elements are rare, and various interpretations of such cysts have been given in the literature. Some authors have described these lesions as orthokeratinized odontogenic cysts (OOCs) exhibiting sebaceous differentiation, whereas others have preferred to consider these lesions intraosseous dermoid cysts or unusual variants of dentigerous cysts.1 Chi et al reported 5 cases of jaw cysts with sebaceous elements and reviewed the literature concerning these unusual lesions. In particular they examined the apparent controversy as to whether these lesions are best considered odontogenic or nonodontogenic in origin. In the jaw cysts which the authors reviewed many were interpreted to represent odontogenic cyst with sebaceous elements formed by metaplasia of the epithelial lining. Whereas sebaceous glands located deeper within the cyst wall potentially could originate from metaplasia of sequestered epithelial rests.1
Other authors have interpreted intraosseous jaw cysts with sebaceous glands as dermoid cysts and have rejected theories of odontogenic origin.1 Dermoid cysts commonly occur in soft tissues and reports of intraosseous dermoid cysts of the jaw are exceedingly rare.8 Characteristic microscopic features of the dermoid cyst include an epidermis like lining, intraluminal keratinaceous debris, and one or more skin appendages. The presence of hair follicles and/ or sweat glands could be considered evidence in favor of an epidermal rather than odontogenic origin of such cysts. On the other hand, if it is possible for sebaceous differentiation to occur in odontogenic cyst, then perhaps it is possible for odontogenic cyst to exhibit the formation of hair follicles or sweat glands as well.8 Many investigators have commented on the pluripotentiality of the odontogenic epithelium, which apparently may have the capacity to differentiate into sebaceous cells, mucous cells, respiratory epithelial cells, and other cell types. But well formed adenexal structures other than sebaceous glands arising from odontogenic epithelium have not been conclusively demonstrated.3, 9
It seems reasonable to postulate that some factors in the oral mesenchyme could interact with epithelium of almost any source to produce wide variability in structure and potential of the affected epithelium. The multipotentiality of the oral epithelium may be secondary to the influence of the mesenchyme, resulting in the differentiation of sebaceous glands in odontogenic cysts. 10

In our case presented here adnexal structures other than sebaceous gland was absent in the lining epithelium and connective tissue. Thus we believe that the lesion is odontogenic in origin and we can exclude dermoid cyst. Some authors have preferred to use more descriptive, noncommittal terminology for these lesions, such as “intraosseous mandibular cyst with sebaceous differentiation”, as proposed by Cristensen and Propper.
To summarize, it is important to note that dentigerous cyst with sebaceous differentiation do occur and must be separated from other non odontogenic cysts in a clinical and histological differential diagnosis.
Figure Legends
Figure 1: Orthopantomograph showing well defined unilocular radiolucency around the crown of impacted left mandibular second premolar. Figure 2: Photomicrograph showing typical cystic lining of dentigerous cyst having 2-3 layered non keratinized epithelial lining. Focal collection of sebaceous elements is seen in the underlying connective tissue. (Hematoxylin & Eosin, X 40) Figure 3: Photomicrograph showing sebaceous cells as well as chronic inflammatory cells within the connective tissue. (Hematoxylin & Eosin, X 100)
References
  1. Chi AC, Neville BW, McDonald TA, Trayham RT, Byram J, Peacock EH. Jaw cysts with sebaceous differentiation: report of 5 cases and a review of the literature. J Oral Maxillofac Surg 2007; 65: 2568-2574
  2. Hofrath H. Uber das vorkommen Von Talgdrussen in der Wandung einer Zahncyste, Zugelich ein Beitrag zur Pathogenese der kiefer-Zahncysten, Dtsch Monatsschr. Zahn heilkd. 1930; 2:65-76.
  3. Gorlin RJ. Potentialities of oral epithelium manifested by mandibular dentigerous cyst. Oral Surg 1957; 10:271-84.
  4. Spouge JD. Sebaceous metaplasia in the oral cavity occurring in association with dentigerous cyst epithelium. Oral Surg 1966; 21: 492-8.
  5. Downie MMT, Guy R, Kealey T. Advances in sebaceous gland research: Potential new approaches to acne management. Int J Cosmet Sci 2004; 26: 291
  6. Halperin V, Kolas SR, Huddleston So, Robinson Hb. The occurrence of Fordyce spots, benign migratory glossitis, median rhomboid glossitis, and fissured tongue in 2,478 dental patients. Oral Surg Oral Med Oral Pathol. 1953 Sep; 6(9):1072-7.
  7. Neville BW, Damm DD, Allen CM. Fordyces Granules, in Oral & Maxillofacial Pathology (ed 2). Philadelphia, WB Saunders, 2002, p6.
  8. Neville BW, Damm DD, Allen CM. Fordyces Granules, in Oral & Maxillofacial Pathology (ed 2). Philadelphia, WB Saunders, 2002, p32.
  9. Brannon RB. The odontogenic keratocyst: A clinicopathologic study of 312 cases. Part II. Histologic features. Oral Surg Oral Med Oral Pathol 1977; 43: 233.
  10. Shamim T, Varghese I, Shameena PM, Sudha S. Sebaceous differentiation in odontogenic keratocyst. Indian J Pathol Microbiol 2008; 51(1): 83-84.

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