SUBLINGUAL DERMOID CYST OF MANDIBULAR REGION : A CASE REPORT. Case Report. University. Journal of. Dental Sciences

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1 SUBLINGUAL DERMOID CYST OF MANDIBULAR REGION : A CASE REPORT Journal of University Dental Sciences Jeevan Lata, Jaideep Marya, Puja 1 Professor and Head, Deptt. of Oral and Maxillofacial Surgery, Govt Dental College and Hospital, Amritsar 2 Assistant Professor, Deptt. of Pathology, Govt. Medical College, Amritsar 3 Junior Resident, Dept of Oral and Maxillofacial Surgery, Govt. Dental College and Hospital, Amritsar ABSTRACT : Dermoid cyst of the floor of the mouth is a rare, benign lesion requiring surgical intervention. Typically they present as slow growing mass causing elevation of the tongue, interference with speech and swallowing. Their etiology is not yet clear and can be associated with entrapment of ectodermal and mesodermal components when the 1st and 2nd branchial arches of each side fuse in the midline. The entrapped tissues then undergo proliferation and cystic transformation.they are uncommon in the head and neck region. Extensive understanding about this slow growing painless mass is essential because of the symptoms it produces and its malignant potential. Surgical enucleation is the treatment of choice for this type of lesions according to literature. A case of sublingual dermoid cyst in an eighteen year old female patient is presented. As MRI help us to identify the cyst and its precise location, extension and relationship with the surrounding structures and they also enable the surgeon to choose the most appropriate surgical approach. So MRI was done for preoperative diagnosis in our case in addition to FNAC. The size of the lesion was less than 6 cm in our patient, so we used intraoral approach. The lesion was successfully treated by surgical enucleation and patient showed no signs of reccurence on 6 months follow up. Case Report Key words: Dermoid cyst, Dysembryogenetic, Teratoid cyst Source of support : Nil Conflict of interest : None INTRODUCTION Dermoid cysts of the floor of mouth are dysembryogenetic lesions derived from entrapement and subsequent growth of epithelial cells during the midline fusion between the first and second branchial arches in the third and fourth embryonic weeks. Acquired forms are derived from iatrogenic or traumatic inclusion of epithelium and skin appendages.[1]. These benign lesions are encountered throughout the body and rarely occur in the head and neck region, 1.6 to 7%, and represent less than 0.01% of all oral cavity cysts.[2] The floor of the mouth is the second most common site in the head and neck region after the lateral eyebrow as these are the sites of embryonic fusion. The occurrence in oral cavity is approximately 1.6 %.The vast majority of dermoid cysts of the floor of mouth (DCFOM) are located in the midline (sublingual 52%, submental 26%), 16% involve more than 1 of the 3 possible spaces in the floor of the mouth region (submental, sublingual, submandibular), and only 6% are situated exclusively in the submandibular space where they appear to be lateral neck cysts [3]. Dermoid cysts generally present with slow and progressive growth, and even if they are congenital, the diagnosis is usually possible in the second or third decade of life.[4] They usually present as a painless swelling in the floor of the mouth and may increase in size at the onset of puberty when there is an increase in the secretion of sebum from the sebaceous glands.[5] We report a case of dermoid cyst of floor of mouth in a young adult female, which was successfully treated by surgical enucleation. University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 80

CASE REPORT An eighteen year old female patient reported with a cheif complaint of gradually increasing painless midline swelling in the floor of mouth since 1 year.

On extraoral examination there was diffuse non tender swelling in the neck. The swelling extended from 1finger below the chin to 1 finger away from the thyroid cartilage.

The swelling was compressible, non translucent, cystic in consistency,non pulsatile, non reducible, the mass was bimanually palpable and on digital palpation over submental area the swelling

thrill and impulse on coughing. Patient was unable to close the lips completely. On intraoral examination there was an oval shaped non tender, solitary,sessile swelling of about 5 3.

2 CASE REPORT An eighteen year old female patient reported with a cheif complaint of gradually increasing painless midline swelling in the floor of mouth since 1 year. The swelling appeared spontaneously.no history of trauma was present. She was thin built with no history of any systemic illness. On extraoral examination there was diffuse non tender swelling in the neck. The swelling extended from 1finger below the chin to 1 finger away from the thyroid cartilage. There was no movement of swelling with the respiration, deglutition and protrusion of tongue. The swelling was compressible, non translucent, cystic in consistency,non pulsatile, non reducible, the mass was bimanually palpable and on digital palpation over submental area the swelling protrudes in to the oral cavity overlying skin was normal in colour and texture,temperature was normal, surface was smooth, not fixed to overlying skin, borders were indistinct,no evidence of fluid thrill and impulse on coughing. Patient was unable to close the lips completely. On intraoral examination there was an oval shaped non tender, solitary,sessile swelling of about cm in size in the floor of mouth (Fig. 1). Extending from the lingual aspect of mandibular anterior teeth upto mandibular molars bilaterally. The edges were well defined, does not slipped rather yielded, surface was smooth, doughy in consistency, and overlying mucosa was normal in colour with slight yellowish tinge, temperature was normal. It was compressible, moulded on pressure, non pulsatile, non reducible and was not fixed to overlying mucosa, fluctuant on bimanual palpation, fluid thrill was absent, translucency was absent..tongue movements were restricted. Dysphagia and dysphonia was present. There was no history of dysnoea. The mass was displacing the tongue superiorly and posteriorly. The oral mucosa and warthins ducts were healthy. Mouth opening was normal. Lymph nodes were not palpable.no secondary changes occurred in the swelling. There was no history of discharge or associated sinus in relation to the swelling. No other lump was present in the body. Fig.1. showing raised tongue due to swelling The large sublingual swelling raised the tongue upwards(fig.1).on aspiration thick cheesy white fluid was present.fine needle aspiration cytology was done which yielded thick cheesy white aspirate. The smear showed many mature looking squamous cells, a few anucleate squames and scattered foamy histiocytes. No malignant cells were seen, no inflammation was noted. The clinical and F.N.A.C findings indicated a provisional diagnosis of dermoid cyst, while the differential diagnosis included a plunging ranula, epidermoid cyst, thyroglossal duct cyst,cystic hygroma, sublingual/submental space infection, lymph node enlargement, teratoid cyst, sublingual salivary gland infection or tumor. After doing F.N.A.C fever appeared. For this antipyretics and antibiotics were given, after two days fever got relieved. Fig (2) Sagittal, Axial and Coronal sections of MRI showing well circumscribed radio-opacity in the sublingual region MRI (Fig 2) was done which showed cm well circumscribed cystic mass in the floor of mouth extending from mandible anteriorly to base of tongue posteriorly. MRI showed it to be ranula but FNAC suggested it to be dermoid cyst. All routine laboratory investigations were done which were within normal limits,. Surgical excision under general anaesthesia was planned. Oral intubation was done.before giving incision aspiration of the swelling was done to decompress it so as to prevent rupturing. University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 81

On gross examination of the specimen, the lumen contained pultaceous material. The inner surface was skin like rough.

No other heterologus elements were seen. Fig ( 3) Incision was placed in the floor of mouth Fig. 7-a.

Sebaceous gland is also seen at one place in the underlying connective tissue Fig (4) Total enucleation of the cystic lining T shaped incision was made in the midline of

Whole of the cystic lining was removed Fig(4&5). After enucleation of the cyst layerwise closure was done Fig (6) after ensuring haemostasis.

H&E Section 20X showing acute inflammation in the skeletal muscle surrounding the cyst wall There was acute inflammation in the skeletal muscle and fibroadipose tissue,

Healing was satisfactory except for contracture at the incision line with slight restriction on protrusion of tongue.

3 On gross examination of the specimen, the lumen contained pultaceous material. The inner surface was skin like rough.on microscopic examination (Fig 7a&b) the lining epithelium was stratified squamous with orthokeratosis.the underlying stroma exhibited sebaceous gland at one place. No other heterologus elements were seen. Fig ( 3) Incision was placed in the floor of mouth Fig. 7-a. H&E Section 10X showing keratin desquamating from the orthokeratinized stratified squamous epithelial lining of the dermoid cyst. Sebaceous gland is also seen at one place in the underlying connective tissue Fig (4) Total enucleation of the cystic lining T shaped incision was made in the midline of floor of mouth.(fig 3) Sharp and blunt dissection was done. Pultaceous material came out during dissection. Finally the cyst was separated from mylohyoid muscle. Whole of the cystic lining was removed Fig(4&5). After enucleation of the cyst layerwise closure was done Fig (6) after ensuring haemostasis. Specimen was sent for histopathological examination. Fig (5) Cystic lining Fig(6) layerwise suturing done Fig. 7b. H&E Section 20X showing acute inflammation in the skeletal muscle surrounding the cyst wall There was acute inflammation in the skeletal muscle and fibroadipose tissue, surrounding the cyst wall. No malignancy was seen. Thus the histopathological examination confirmed that it was dermoid cyst. Healing was satisfactory except for contracture at the incision line with slight restriction on protrusion of tongue. Patient complained of excessive salivation postoperatively which resolved after some time. On follow up of patient at 4 months postoperatively no complication was noted except deviation of tongue on protrusion due to contracture. Tongue exercises were advised for this. On 6 months followup deviation of tongue on protrusion decreased but still persisted. No sign of reoccurrence was noticed during 6 months follow up period. Patient was satisfactory with the outcome. University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 82

4 DISCUSSION : Dermoid cysts are most frequently manifested between the second and sixth decade of life, although cases of younger and older patients have been reported. In our case the age of patient was eighteen years. The most common location in the head and neck area is the periorbital region followed by intraoral sites. The intraoral cyst generally develops in the floor of mouth and it can be found either lateral to tongue or in the midline. The cyst shows no gender predilection, grows slowly, is well circumscribed, and unilocular. The size of the cyst usually varies from several millimeters to as much as 12 cm [6]. Depending on the size of the lesion, it can displace the tongue and cause dysphagia, dysphonia, and dyspnea[7]. In our case dysphagia and dysphonia was present. The cysts in the midline are hypothesized to be caused due to one of the following: 1. Entrapment of ectodermal tissues of first (mandibular) and second (Hyoid) branchial arches, which is called dysdontogenic hypothesis. 2. Traumatic implantation of epithelial cells into deeper tissues which suggests traumatic cause. 3. Third theory considers dermoid cyst as a variant of thyroglossal cyst [8] The cysts of the midline can be classified according to its relationship to the muscles of the floor of the mouth. Sublingual cysts; cysts presenting as swelling in the floor of the mouth and situated above the mylohyoid and genioglossus. Submental cysts present as a swelling below the chin and is situated between the mylohyoid and geniohyoid muscle. The third variant would be a cyst occupying both sublingual and submental spaces, perforating the oral diaphragm which would be a large cyst.[8] Based on the histopathologic picture, Meyer divided the floor of the mouth cysts into following types: (1) Epidermoid cysts, which are simple cysts, lined with simple squamous epithelium, with a fibrous wall and no skin appendages or adnexals like hair follicles, sebaceous or sweat glands in their connective tissue wall. As these cysts develop from the upper part of the pilosebaceous unit, they are incapable of producing sebum and do not contain any skin appendages. b) True Dermoid cysts, also known as compound cysts, which are lined with keratinizing stratified squamous epithelium with skin adnexals in the connective tissue wall. The lumen will contain keratin, sebum, varying amounts of fat and occasionally, hair. c) Teratoid cysts, also known as complex cysts, which are lined with epithelium ranging from simple stratified keratinized squamous to stratified columnar respiratory type of epithelium and containing derivatives from all the three germ layers (ectoderm, mesoderm and endoderm) within their lumen. The cystic cavity in addition to skin appendages also encloses mesodermal derivatives such as bone, muscle, gastrointestinal and respiratory tissue[9]. Our case of cyst fall in to Dermoid type. A conclusive preoperative diagnosis may be difficult. CT scan, MRI, Ultrasound, and FNAC can be performed. CT scan and MRI can show the exact location and size of mass and relationship to adjacent structures, but cannot give a definitive preoperative diagnosis[10]. MRI help us to identify the cyst and its relationship between geniohyoid and mylohyoid muscles. Also this information clarifies the surgical approach[11]. Ultrasound may be helpful in differentiating between solid vascular and cystic lesions. The final diagnosis cannot be established until histopathological study of surgical specimen is performed. In patient presenting with lesion in floor of mouth, FNAC should be the first line of diagnostic procedures. Dermoid cysts of the floor of the mouth have been successfully diagnosed by this method.[11] While aspirating, extreme caution should be exercised for sterilization and asepsis. Secondary infection may have adverse consequences. Literature supports this statement and also suggests that aspiration should only be done in hospital setting Once a provisional diagnosis of a dermoid cyst is formulated, incisional biopsy is not recommended. There may be chances of secondary infection, collapse of cyst due to drainage of contents making the surgical excision difficult and formation of draining sinus in relation to biopsy site. As surgical excision is the treatment of choice, and recurrence is extremely rare, thus we followed same principle in management of the patient..[12] In our case before giving incision aspiration of the swelling was done to decompress it.although collapsing a cyst by needle aspiration is a commonsense approach, surprisingly this technique has not been adequately highlighted in the literature. Only on five previous occasions has partial cyst decompression been reported in the literature.[13] University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 83

5 While intraoral incisions are sufficient in accessing most cases of dermoid occurring in the midline, extraoral incisions may be required for cysts occurring inferior to the mylohyiod, in the submandibular regions and for large cystic lesions which extend across the mylohyoid[10]. Some authors claim that lesions that are less than 6 cm in diameter and above the mylohoid muscle are suitable for intraoral excision. Also they claim that dermoid cysts larger than 6 cm in diameter and located sublingually should be excised by extraoral approach.[11] MRI showed it to be ranula but histopathology confirmed it to be Dermoid cyst; all the other possibilities of sublingual lesions were ruled out on the basis of histopathology and MRI. [14]. The recurrence rates for the dermoid cyst are low[10]. Malignant changes have been recorded in dermoid cysts by New and Erich but not in the floor of the mouth, although a 5% rate of malignant transformation of oral dermoid cysts of the teratoid type has been reported by other authors.[14] Differential diagnosis of cystic lesions of the floor of the mouth is important because the recommended surgical technique is not exactly the same in all of them. There are several lesions which can present as a cyst or pseudocyst of the floor of the mouth with submental repercussion and these include neoplasm, infections and developmental processes i.e. cystic hygroma, acute infection, neurofibroma, haemangioma, sublingual ranula, lipomas, Ludwig's angina and lymphangioma.[15] Treatment comprises total surgical excision. Caution should be taken not to rupture the cyst, as cystic contents may act as irritants to fibrovascular tissues, causing postoperative inflammation.[9] CONCLUSION : Dermoid cysts found on the floor of the mouth are rare. Extensive understanding about this slow growing painless mass is essential because of the symptoms it produces and its malignant potential. When dermoid cysts occur on the floor of the mouth, they may enlarge to such an extent that they can interfere with deglutition and can produce respiratory obstruction. Early diagnosis and treatment are essential for these cystic entities. Appropriate imaging techniques is necessary for the diagnosis of cysts of the floor of the mouth. Surgical enucleation is the treatment of choice. REFRENCES : 1. Hemaraju N, Nanda SK, Medikeri SB. Sub-lingual dermoid cyst. Indian Journal of Otolaryngology and Head and Neck Surgery Jul 1;56(3): Vieira EM, Borges AH, Volpato LE, Porto AN, Carvalhosa AA, Botelho GD, Bandeca MC. Unusual Dermoid Cyst in Oral Cavity. Case reports in Pathology Apr 10; Sahoo NK, Choudhary AK, Srinivas V, Tomar K. Dermoid cysts of maxillofacial region. Medical Journal Armed Forces India Dec 31;71:S Kandogan T, Koç M, Vardar E, Selek E, Sezgin O. Sublingual epidermoid cyst: a case report. Journal of Medical Case Reports Sep 17;1(1):1. 5. Chukwuneke FN, Akaji C, Onyeka TC, Udeagha P. Surgical excision of intra-oral dermoid cyst under local anaesthesia: a review of nine cases. Journal of Maxillofacial and Oral Surgery Mar 1;9(1): Yilmaz T, Unal OF, Altinok G Pathology quiz case 2. Epidermal inclusion cyst. Arch Otolaryngol Head Neck Surg ; 127(11): Seah TE, Sufyan W, Singh B. Case report of a dermoid cyst at the floor of the mouth. Annals- Academy of Medicine Singapore Jul 1;33: Sheshadri P, Kalappa TM, Krishna BP, Kumaran S, Biddappa ML. Dermoid Cyst of Submental Region Mimicking Pilomatricoma. Journal of Maxillofacial and Oral Surgery. 2015: Jeyaraj P, Sahoo BN. A case of an unusually large sublingual dermoid cyst of the maxillofacial region. J. Dent. Med. Med. Sci. 2012;2(2): Cramer H, Lampe H, Downing P. Dermoid cyst of the floor of the mouth diagnosed by fine needle aspiration cytology. Acta Cytologica Jul 1;40(2): Aydýn S, Demir MG, Demir N, ªahin S, Kayýpmaz ÞS A Giant Plunging Sublingual Dermoid Cyst Excised by Intraoral Approach J. Maxillofac. Oral Surg.2016;15(2): Jain H, Singh S, Singh A. Giant sublingual dermoid cyst in floor of the mouth. Journal of Maxillofacial and Oral surgery Jun 1;11(2): King RC, Smith BR, Burk JL. Dermoid cyst in the University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 84

6 floor of the mouth: review of the literature and case reports. Oral surgery, Oral medicine, Oral pathology Nov 30;78(5): Gulati U, Mohanty S, Augustine J, Gupta SR. Potentially Fatal Supramylohyoid Sublingual Epidermoid Cyst. Journal of Maxillofacial and Oral Surgery Mar 1;14(1): Verma S, Kushwaha JK, Sonkar AA, Kumar R, Gupta R. Giant sublingual epidermoid cyst resembling plunging ranula. National Journal of Maxillofacial Surgery Jul 1;3(2):211. CORRESPONDING AUTHOR: Dr. Puja H.N.O 762/3, Topkhana Road, Patiala India pujatopkhana4@gmail.com University Journal of Dental Sciences, An Official Publication of Aligarh Muslim University, Aligarh. India 85

PHISIS: THE COMBINED SURGICAL APPROACH

ISSN: 2250-0359 Volume 5 Issue 3 2015 A CASE OF GIANT SUBLINGUAL DERMOID CYST ORIGIN FROM THE MANDIBULAR SYM- PHISIS: THE COMBINED SURGICAL APPROACH Selçuk Güneş,Mustafa Çelik,Yakup Yegin,Kamil Hakan Kaya,Mustafa