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East and Central African Journal of Surgery, Vol. 16, No. 2, July/August, 2011, pp. 157-161 Congenital Sublingual Cyst: A Case Report J. M. Simba1, I. K. Marete2, R.T. Kuremu3 1 Assistant Lecturer Child Health &
Paediatrics Department, 2Lecturer Child Health & Paediatrics
Department,3Associate Professor Surgery &
Anaesthesiology Dept,Moi University, School of Medicine, Eldoret,
Kenya. Code Number: js11045 Congenital sublingual cysts are usually rare. A neonate presented in our facility with feeding difficulties caused by a large sublingual cyst. Excision of the cyst was done and post operative period was uneventful. In this case we discuss the common presentation of such rare cyst and their management. Introduction Congenital sublingual cysts are generally rare. Over a period of 25 years only 83 cases of ranula were documented in Zimbabwe while Roscow et.al (2009) and Ellegard et.al (2010) noted less than 30 cases of cyst arising from duct atresia have been described in English literature. Cyst in the sublingual region could either be true cysts or pseudocyst; benign or malignant1,2,3,4,5,6. A ranula is a pseudocyst that is caused by the extravasation of mucus from the sublingual gland1. It occurs specifically in the floor of the mouth associated with the sublingual or submandibular salivary glands as a fluctuant, unilateral, bluish soft tissue mass. They are cystic and are frequently blue owing to the Tyndall effect, whereby blue light is reflected more than red light at the interface of soft tissue and cyst. Ranula could occur following trauma or obstruction of the outlet of the glands1,7,8. Similar cysts could arise from; an imperforate submandibular or sublingual duct orifice. This may result in a cystic swelling with a characteristic bluish, translucent appearance though it occurs rarely4. Unlike ranula cyst these have epithelial lining1,2,4,9. Other differential diagnoses include dermoid cysts, lymphatic or vascular malformations, minor salivary gland tumors, and mucous retention cysts. Dermoid cyst usually occurs in the midline4,10. Patients usually present with non painful swelling noticed incidentally or by displacement of the tongue. It may also be suspected in a neonate with feeding difficulties. Interference with feeding may lead to failure to thrive if not addressed5. The methods, and the age of intervention are diverse in literature. Observation for 6 months has been suggested as is aspiration upto removal of ipsilateral sublingual gland4,7,8. We present a case of a neonate with a congenital sublingual cyst who presented with feeding difficulties at birth. Case Report A 9 days old male neonate was referred to us from a district hospital. The neonate was born at term to a 21 year old primigravida through spontaneous vertex delivery assisted by a Traditional Birth Attendant at home, cried immediately after birth and was essentially normal at that point save for a swelling in the floor of the mouth that interfered with breastfeeding of the baby. The swelling was noted on day one of life. The swelling progressively increased in size and by the time of being at our facility, the child was only able to feed by cup and spoon. No other reported problems, and the birth was uneventful. The mother had attended clinic from the 7th month of pregnancy with normal antenatal profile and nonreactive HIV screen. She did not smoke nor take alcohol during pregnancy. The pregnancy was uneventful with a normal labour of about 14hours. No history of prolonged rupture of membranes. On arrival to our facility the neonate was in fair general condition, no pallor, cyanosis or edema, but had a tinge of jaundice. No features of dehydration noted. The vital signs were all within normal with pulse rate of 148, respiratory rate of 46, temperature of 37.1oC and had oxygen saturation of 92% in room air. Head examination was normal other than for the swelling in the mouth. Local examination revealed a cystic mass approximately 4cm in diameter on the floor of the mouth arising on the right side and pushing the tongue completely away to the left. The swelling had ulceration, approximately 1/2cm in diameter on the surface. It generally had no colour change compared to surrounding tissues. No sinus noted. The gum and lips were not involved. The submandibular and submental regions were normal. See Figure 1 .All other systems were normal. The baby was reviewed and scheduled for theatre with a diagnosis of sublingual cyst. Preoperatively the complete blood count showed Haemoglobin of 15.6g/dl; White blood count of 9.7*109/L (Granulocytes-26.9% & Lymphocytes-59.1%); Platelets of 383*109/L; Urea/electrolytes and creatinine were normal. Pre-operative supportive treatment included antibiotics and intravenous fluids. Intraoperatively, the child was induced and intubated nasotracheally. Excision of the cyst was done, by dissecting it out, after packing the posterior oral cavity. Closure was done using absorbable suture. There was mucinous material in the cyst. See Figure 2 . Capsule was taken for histology. The neonate was reversed successfully and had restoration of normal anatomy in the oral cavity. See Figure 3 . The immediate post operative period was uneventful and the neonate was slowly reintroduced to feeds after 12hours. The neonate was discharged on the third day postoperatively able to breastfeed. The histology reported a fibrous cyst wall lined by stratified squamous epithelium. There were mucous glands seen in the wall, with a conclusion of a simple cyst. Discussion Cysts in the mouth are rare occurrences. The types of cysts in neonates include vascular malformations/lymphagioma, mucoceles of minor salivary glands, ranulas, mucous retention cyst or even due to imperforate salivary gland duct orifice1,2,4,5,11. Sublingual cyst could be inflammatory, traumatic or neoplastic. Most oral lesions in infancy are benign11. Our patient presented with a lesion at birth. This puts inflammatory/infective conditions low in our differentials. There was neither history of prolonged rupture of membranes, nor birth trauma. There was also no maternal illness. Ranulas incidence increase with HIV seropositivity but our neonate was not seroexposed6,12. Chidzonga6 documented only 83 cases of ranulas over a period of 25 years in Zimbabwe. So far only 29 cases of babies with sublingual/ submandibular duct atresia presenting with sublingual cyst have been described4,5. Ranulas are slightly more common in females and mostly involve the left side while dermoid cysts are reported to be more common in males as are cysts arising from duct orifice atresia4,11,13. Ranula is formed following excretory duct rupture with extravasation and accumulation of saliva into the surrounding tissue. The accumulation of mucus in the surrounding connective tissue forms a pseudocyst that lacks an epithelial lining. Rarely though a ranula could occur without extravasation of mucous. The cause of ranula in neonates is however not known. In older children it is associated with trauma to the salivary duct. When the duct orifice is not patent this may end up with congenital sialocoele which is a true cyst with epithelial lining. This is thought to result from a congenital failure of canalization of the terminal end of the duct2,4,9. Dermoid and epidermoid cysts that appear in the midline floor of the mouth are a result of entrapped ectodermal tissue of the first and second branchial arches, which fuse during the third and fourth weeks in utero10. The presenting complaint is usually a slow growing swelling noticed incidentally rarely causing any feeding difficulty10. Our patient presented with feeding difficulties associated with the large size and displacement of the tongue. Any sublingual swelling large enough could present with feeding difficulties 5,10,14. The clinical examination of the lesion showed a cystic lesion, whose differential diagnoses include ranula, but could also occur with cyst resulting from duct atresia and epidermoid cyst (10). Epidermoid cysts usually are in the midline while the others (ranula and duct atresia cyst) are usually lateral. The mucionus substance contained in the cyst points more towards salivary gland abnormality or related problems likely in this case ranula cyst or submandibular/sublingual duct atresia4,10,13. The early presentation is in support of either a ranula cyst, sublingual duct atresia or epidermoid cyst. Most of cyst associated with duct atresia reported in literature presented at birth or as neonates4,5. Our investigations were limited on account of cost and availability. The clinical assessment satisfied the clinicians to go ahead with the operation. However, imaging is not always necessary in preoperative diagnosis and surgical planning, neither is fine needle aspiration. Investigations such as ultrasound, CT scan and MRI have been done preoperatively in other studies10. Histology done showed a fibrous cyst wall lined by stratified squamous epithelium. There were mucous glands seen in the wall. The conclusion was that of a simple cyst. Strictly speaking, ranulas have no epithelial lining though some literature classifies them into two with simple cystic lesions (true cyst) and the extravasation type which is a pseudocyst. Considering this histology report then salivary duct atresia is very likely, though very rare. Considering the stratified squamous epithelium, epidermoid cyst is a possibility but since no skin appendages were seen it becomes unlikely1,2,4,7,9,15. Treatment of sublingual cyst in neonates includes observation for spontaneous resolution in 6months2,16. This in our case was not feasible since it was already interfering with feeding of the neonate. The excision of the cyst done is among several options for treatment of such lesions. Other methods which can be used include marsupilization, resection of ipsilateral sublingual gland for recurrent ranulas, needle aspiration, cryotherapy or scleroherapy2,4,7,8,11,16. Simple surgical resection is usually successful in treating most benign lesion in the mouth, including epidermal cyst and ranulas, with occasional re-excision being necessary in lymphangiomas4,5,11. Acknowledgement We wish to acknowledge the Director, The Moi Teaching and Referral Hospital for giving us permission to publish this case. References
Copyright 2011 - East and Central African Journal of Surgery The following images related to this document are available:Photo images[js11045f3.jpg] [js11045f2.jpg] [js11045f1.jpg] |
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