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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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  Table of Contents    
CASE REPORT
Year : 2022  |  Volume : 40  |  Issue : 1  |  Page : 86-89
 

Pedunculated cavernous lymphangioma of the newborn oral cavity


Department of Paediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

Date of Submission26-May-2021
Date of Acceptance13-Jan-2022
Date of Web Publication13-Apr-2022

Correspondence Address:
Dr. Manoj Kumar Mohanty
Department of Paediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar - 751 019, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jisppd.jisppd_189_21

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   Abstract 


Oral swellings and ulcers in neonates are a spectrum of diseases often creating a sense of anxiety among parents. Early detection, high index of suspicion, proper investigations, and prompt diagnosis can aid in accurate management of the same. Lymphangiomas are benign hamartomas which are basically malformed lymphatics do not drain into other lymphatics or veins and hence there is lymphatic accumulation and enlargement following cystically dilated spaces. We hereby present a rare case of a neonate born with a cystic mucosal swelling at the upper gingival sulcus since birth.


Keywords: Lymphatic malformation, oral cyst, newborn oral lesions, benign cyst


How to cite this article:
Manekar AA, Janjala N, Sahoo SK, Tripathy BB, Mohanty MK. Pedunculated cavernous lymphangioma of the newborn oral cavity. J Indian Soc Pedod Prev Dent 2022;40:86-9

How to cite this URL:
Manekar AA, Janjala N, Sahoo SK, Tripathy BB, Mohanty MK. Pedunculated cavernous lymphangioma of the newborn oral cavity. J Indian Soc Pedod Prev Dent [serial online] 2022 [cited 2022 Jun 25];40:86-9. Available from: https://www.jisppd.com/text.asp?2022/40/1/86/343010





   Introduction Top


The first case of alveolar ridge lymphangioma in a neonate was reported first in 1976 in a study involving more than 2000 infants.[1] Just like other vascular malformations, lymphangiomas are lymphatic malformations showing abnormal proliferation of the lymphatic vessels. They are benign hamartomatous tumors, mostly localized in about 75% of cases to the head-and-neck region.[2] Clinically, they are slow-growing, painless soft-tissue masses. Extension of the lymphangioma defines the clinical appearance of the lesion. Superficial lesions present as elevated nodules with pink or yellowish color or seen as transparent grouped vesicles, which can be red or purple due to secondary hemorrhages.[3] Deeper lesions appear as diffuse masses with normal color and are soft in consistency.[4] Anterior two-third of the dorsum of the tongue is the most common site, followed by the lips, buccal mucosa, soft palate, and floor of the mouth.[5],[6] Surgical excision is the preferred treatment when vital structures are not involved.[5],[7] Other procedures such as sclerotherapy, laser therapy, electrocautery, radiation therapy, cryotherapy, embolization, ligation, and steroid administration are also used.[5],[8]


   Case Report Top


A 1 day newborn, registered inborn male child, was referred to pediatric surgery for the complaint of a swelling in the upper gingival sulcus noticed by the mother since birth while she was breastfeeding the baby. There was no history of difficulty in feeding the baby and also no history of respiratory distress. There was no evidence of any active bleeding from the lesion.

General and systemic examination revealed normal findings. Examination revealed a single, round, and 2 cm × 2 cm pedunculated swelling in the left upper gingival sulcus which was soft in consistency, partially compressible, and of the same color as the surrounding mucosa [shown in [Figure 1]]. There was no evidence of any other cyst in the oral cavity as well as the pharynx.
Figure 1: Clinical photograph of mucosal cyst of the newborn

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Doppler ultrasound of the left cheek with the lesion showed multiple dilated spaces with a hypoechoic content, with slow flow of the blood or lymph, labelling it to be a slow-flow lymphovascular malformation. Magnetic resonance angiography (MRA) revealed a hyperintense swelling on T2 plates, suggesting it to be a vascular malformation shown in [Figure 2].
Figure 2: Magnetic resonance angiography showing the mucosal lesion

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After proper preparation, the child was taken up for surgery where the cystic mass was excised at the base using bipolar cautery shown in [Figure 3].
Figure 3: Intraoperative photographs showing the pedunculated cyst with another small cyst behind the previous one

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Postoperative recovery was uneventful, and the baby was discharged the next day. The surgical site was healthy on follow-up, and histopathology report showed the presence of a cavernous lymphangioma shown in [Figure 4]. The child has no recurrence even after 1 year of age.
Figure 4: Histopathology showing a cavernous lymphangioma

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   Discussion Top


The diseases of the oral cavity form an important arena of the pediatric surgical and dental specialty. Due to lack of resources and parental education, many are left untreated. Although the lesions are usually confined to the oral cavity, they might provide certain clues to the underlying more serious systemic conditions. A broad spectrum of diseases manifests with oral features in the neonates–which include cysts, infections, traumatic lesions, and tumors of the oral cavity structures. Majority of the oral lesions are asymptomatic and benign, commonly resolve, and no intervention is needed. However, a thorough clinical examination and knowledge of the various lesions are essential for precise diagnosis, management, as well as parental counselling.

Lymphangiomas are benign neoplasms of the lymphatic system with 50% of cases noted at birth and 90% developing before the age of 2 years, prevalent in 1–3/10,000 live births, affecting both genders equally, most commonly present in the head-and-neck region (75%), followed by trunk, abdomen, and extremities.[9],[10] Oral sites include the dorsum of the tongue, lips, buccal mucosa, soft palate, and floor of the mouth in the descending order of preference. Clinically, it is a slow, progressive lesion with superficial blue-black or red hemorrhagic elevated nodules, the deeper lesions presenting as soft, diffuse growths with normal color, and cystic forms manifesting as soft, painless fluid-filled lesions, characteristically located in the neck.[11] In our case, the baby presented with the lesion since birth, and the lesion was arising from the left upper gingival sulcus and was typically a pink and pedunculated lesion.

Radiographic investigations such as ultrasonography (US), computed tomography (CT), and magnetic resonance imaging (MRI) may be required to clinch the diagnosis. US combined with Doppler study is routinely to evaluate the size and extension of the lesion.[12] Both CT and US have proved to be beneficial to evaluate the solid or cystic nature of the lesion, whereas MRI has been most useful in evaluating the extension of the lesion.[13] We used US and MRI as a diagnostic investigation for the mucosal cyst which labelled our lesion as a slow-flow vascular malformation. However, MRA revealed no feeding artery and a well-circumscribed lesion in the upper gingival region.

Various treatment modalities have been employed for the treatment of lymphangioma of the tongue. The various treatment modalities for lymphangioma are surgical excision, radiation therapy, cryotherapy, electrocautery, sclerotherapy, steroid administration, embolization, ligation, laser surgery with Nd-YAG, CO2, and radiofrequency tissue ablation technique. Surgical excision is the treatment of choice when lymphangiomas are encapsulated or partially circumscribed.[14] Surgical excision to be successful requires the inclusion of a surrounding border of normal tissue, taking care that vital structures are not harmed.[15],[16] Nd-YAG laser surgery has become the choice of treatment because of its advantages as less bleeding and edema versus standard methods of surgical resection.[17] Sclerosing agents do not prove effective due to discontinuous basement membrane of the lymphatic vessels.[18] Ablative therapy of lymphangiomas requires exact knowledge of anatomy. The lymphangioma in our case was in the gingivobuccal sulcus and a pedunculated mobile mass. There was a very high risk of avulsion of the polyp and sudden aspiration into the respiratory tract leading to the respiratory emergency; therefore, we chose the surgical mode of treatment, i.e., excision of the mass.

The recurrence of a lymphangioma is common because of its infiltrative nature. Orvidas and Kasperbauer stated a 39% recurrence rate for lymphangiomas.[19] As the lesion in our case was pedunculated and of a benign nature, we went forward with surgical management for the same to prevent potential complications of aspiration and bleeding. There is no evidence of recurrence as of now at 1-year of follow-up.


   Conclusions Top


Lymphangiomas although common in newborns, the presence of a pedunculated lymphangioma in a newborn oral cavity is a rare entity with the risk of avulsion and aspiration into the airway. MRI, although difficult, is the most useful tool for the diagnosis and knowing the extent of the lesion. Prompt surgical excision should be the preferred mode of treatment in such cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Levin LS, Jorgenson RJ, Jarvey BA. Lymphagiomas of the alveolar ridges in neonates. Pediatrics 1976;58:881-4.  Back to cited text no. 1
    
2.
Mandel L. Parotid area lymphangioma in an adult: Case report. J Oral Maxillofac Surg 2004;62:1320-3.  Back to cited text no. 2
    
3.
Weiss SW, Goldblum JR. Tumors of lymph vessels. In: Soft Tissue Tumors. 4th ed. St. Louis: Mosby; 2001. p. 955-67.  Back to cited text no. 3
    
4.
Pinto A. Pediatric soft tissue lesions. Dent Clin North Am 2005;49:241-58.  Back to cited text no. 4
    
5.
Bhayya H, Pavani D, Avinash Tejasvi ML, Geetha P. Oral lymphangioma: A rare case report. Contemp Clin Dent 2015;6:584-7.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Gupta S, Ahuja P, Rehani U, Singh V. Lymphangioma of cheek region – An unusual presentation. J Oral Biol Craniofac Res 2011;1:47-9.  Back to cited text no. 6
    
7.
Sunil S, Gopakumar D, Sreenivasan BS. Oral lymphangioma – Case reports and review of literature. Contemp Clin Dent 2012;3:116-8.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Babu DB, Kumar BR, Boinepally NH, Gannepalli A. A case of intraoral lymphangioma circumscripta – A diagnostic dilemma. J Clin Diagn Res 2015;9:D11-3.  Back to cited text no. 8
    
9.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 3rd ed. New Delhi: Saunders, Elsevier; 2009. p. 984.  Back to cited text no. 9
    
10.
Kolay SK, Jha PK, Sinha RK, Kumar P. Oral lymphangioma: A review. J Clin Den Res Edu 2012;1:42-6.  Back to cited text no. 10
    
11.
Kennedy TL, Whitaker M, Pellitteri P, Wood WE. Cystic hygroma/lymphangioma: A rational approach to management. Laryngoscope 2001;111:1929-37.  Back to cited text no. 11
    
12.
Grasso DL, Pelizzo G, Zocconi E, Schleef J. Lymphangiomas of the head and neck in children. Acta Otorhinolaryngol Ital 2008;28:17-20.  Back to cited text no. 12
    
13.
Alqahtani A, Nguyen LT, Flageole H, Shaw K, Laberge JM. 25 years' experience with lymphangiomas in children. J Pediatr Surg 1999;34:1164-8.  Back to cited text no. 13
    
14.
Kennedy TL. Cystic hygroma-lymphangioma: A rare and still unclear entity. Laryngoscope 1989;99:1-10.  Back to cited text no. 14
    
15.
Fung K, Poenaru D, Soboleski DA, Kamal IM. Impact of magnetic resonance imaging on the surgical management of cystic hygromas. J Pediatr Surg 1998;33:839-41.  Back to cited text no. 15
    
16.
Schefter RP, Olsen KD, Gaffey TA. Cervical lymphangioma in the adult. Otolaryngol Head Neck Surg 1985;93:65-9.  Back to cited text no. 16
    
17.
Tasar F, Tümer C, Sener BC, Sençift K. Lymphangioma treatment with Nd-YAG laser. Turk J Pediatr 1995;37:253-6.  Back to cited text no. 17
    
18.
Brennan TD, Miller AS, Chen SY. Lymphangiomas of the oral cavity: A clinicopathologic, immunohistochemical, and electron-microscopic study. J Oral Maxillofac Surg 1997;55:932-5.  Back to cited text no. 18
    
19.
Orvidas LJ, Kasperbauer JL. Pediatric lymphangiomas of the head and neck. Ann Otol Rhinol Laryngol 2000;109:411-21.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    Abstract
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