| Abstract|| |
Hemangiomas are the most common benign vascular tumours, often reported in very young. Though the head and neck regions have high predilection, intra-oral hemangiomas (IH) are very rare. In spite of numerous treatment options, the IH are ideal candidates for surgical exoneration. The reasons for this are manifold. This manuscript intends to present a case of IH arising in the tongue along with subsequent macroglossia. The treatment and surgical strategy based on cardinal principles of macroglossia corrections are described. A note on the challenges associated with treatment is presented.
Keywords: Benign vascular tumours, hemangioma, macroglossia, surgical treatment
|How to cite this article:|
Balaji S M, Balaji P. Large hemangioma of the tongue. Indian J Dent Res 2020;31:979-82
| Introduction|| |
Hemangiomas are a spectrum of congenital, benign vascular tumours which are noticed in newborns, infants and children. These lesions are characterised by excessive localized proliferation of endothelial cells with a central lumen. Clinically, such hemangiomas are classified into infantile and congenital hemangiomas. They also can be classified into several types depending on histology. These lesions commonly involve the skin of the head and neck while the oral cavity and oropharynx are often uncommon. Intra-oral hemangioma (IH) occurs more commonly in the lips but any intra-oral subsites including the maxillary and mandibular bones could be affected. Depending on the site, histology and size, the clinical presentation could change. The natural history of the IH is also influenced by the type, site, and age of onset. It typically initiates as a small area of discoloration that progress to a patch with age-dependent proportional growth with increasing grades of morbidity. Treatment goal in managing IH would be to restore the anatomical continuity and physiological function. To execute this, several strategies have been reported in the literature. They include wait and see approach (for smaller lesions), drug therapy (corticosteroids and beta blockers), sclerotherapy (steroids, bleomycin), cryotherapy, isotope radiotherapy, laser therapy, and surgery., The treatment algorithm is heavily influenced by type, size, location, natural history, and degree of morbidity, anatomical complexities and perfusion. Small IH in non-critical, non-masticatory zones often do not require active treatment but would need active observation. Those IH that rapidly enlarge or those that do not involute or arise in critical or masticatory zone, should be treated early to prevent potentially fatal complications. The quality of life post-removal is a serious concern. This manuscript aims to present yet another case of giant IH of tongue in a young adult male who was treated surgically.
| Case Report|| |
An 18-year-old otherwise healthy male patient reported with an abnormally large, irregularly disproportionate tongue for many years. History revealed that it was a result of progressive enlargement of the tongue over the years. The patient reported difficulty in chewing, swallowing, and inability to close the mouth with the tongue hindering the regular path of closure. There was no history of pain, fever, or bleeding from the tongue. On general examination, the build of the patient was adequate and all vitals were normal. No relevant medical, dental and family history was present. On local examination, a swelling involving most of the tongue was observed. Closer inspection revealed that the superficial surface was observed to be smooth and granular with well-defined borders. The swelling was bluish-purple with a normal coloured adjacent area. On palpation, the swelling was soft to firm in consistency, ovoid to elliptical along the lateral border of the tongue measuring around 4.5 cm × 3 cm [Figure 1]. It was non mobile, non-tender, afebrile with no palpable thrills. It disappears momentarily on digital compression (diascopy test). It was sessile with no underlying attachment or relation with the muscles. Complete blood picture was within normal limits. On ultrasound, a well-defined solid echogenic lesion was found along the lateral borders of the tongue, with central areas of hypoechoicity, with a few small specks of echogenic calcification within the lesion. On Doppler, there was no significant arterial flow. However, on compression, augmentation of venous flow is noted. On CT scan, the lesion appears as a well-circumscribed, heterogeneous soft tissue density mass with 2-3 small foci of calcification within it. MRI revealed a well-defined round to oval slightly lobulated heterogeneous lesion in the lateral aspect of the tongue involving intrinsic muscles and superior portion of floor of the mouth with intense peripheral enhancement, delayed filling, multiple serpiginous flow voids with strong homogenous enhancement in contrast with a marked enhancement of intra-tumoural vessels is noted at the lateral border of the tongue [Figure 2]a,[Figure 2]b,[Figure 2]c. The patient underwent digital subtraction angiography that revealed supply from the right lingual artery [Figure 2]d. A differential diagnosis of hemangioma or arterio-venous malformation was made. There was absence of thrill or bruit, a working diagnosis of cavernous hemangioma was made. Surgical planning for ligation of the lingual artery and excision in Toto was planned.
|Figure 1: (a-c) Abnormally large and irregularly disproportionate tongue with bluish-purple coloured swelling along the lateral border of tongue|
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|Figure 2: MRI imaging of the large Hemangioma of the tongue. (a) Coronal Sagittal T2 fat-saturated image showed high signal swelling of the tongue, (b) Post-processed coronal coloured image of T2 fat saturation showed the location of the hemangioma, (c) Sagittal T2 fat-saturated image showed high signal swelling, (d) Vascular blush as seen in digital subtraction angiography|
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Nasotracheal intubation under endoscopic guidance was used to administer general anesthesia. The surgery started with intralesional injection of triamcinolone and ligating all feeder vessels, particularly the right lingual artery and other vessels identified in the imaging were ligated. The lesion was completely excised after stabilizing the ends of the lesion. The lesion was huge involving the ventral tongue and resulting in macroglossia. For complete rehabilitation, reduction glossoplasty was needed. Considering the local anatomy and the need to remove excess tissues along the lateral edges and hemangiomatous lobules, the flap design was carefully chosen using previous literature: A combined approach of debulking with marginal reduction. Later closure was achieved with 3-0 Vicryl [Figure 3]. Standard prescriptions to prevent infection, control pain and swelling were provided to the patient. The postoperative hospital stay was uneventful with no complications. After an initial nasogastric feed, oral feeding started after 3 days and the patient was discharged [Figure 4]. The excised specimen was sent for histopathological examination. The lesion presented with variably sized vascular spaces lined by flat endothelial cells and myxoid or fibrous stroma. Variable thrombosis, calcifications and phleboliths were also present, confirming the diagnosis [Figure 5]. The patient now has proper swallowing and respiratory function, improved speech and aesthetic issues, and thus a better quality of life. The patient is followed up with annual consultation.
|Figure 3: (a) Excision of lesion, (b) Postoperative view of the tongue following excision, (c) Excised lesion|
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|Figure 4: (a) Preoperative view of hemangioma of tongue (b) Full postsurgical apposition of lips|
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|Figure 5: (a) High power H and E section showing the fine small blood vessels, (b) Low power histopathology showing fine blood vessels in H and E section, (c) H and E section showing dilated capillaries with blood|
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| Discussion|| |
Macroglossia resulting from an IH has to be carefully approached. The diagnosis has to be made after a study of all imaging modalities. The feeder vessels and hemodynamic status have to be carefully weighed. The depth of soft tissue involvement assessment is a critical factor. Compartmental assessment of the tongue tissues and IH involvement are key influencers. Similarly, adequate knowledge of the various approaches to glossectomy would be needed to plan the surgery. Surgical approach would be only considered when the IH is large and conventional treatments like systemic corticosteroids, beta-blockers, intra-lesional sclerosing agents are contraindicated, have failed or are ruled out.
When surgical approach is finalized, the possibility of presurgical embolization should be considered. Such a procedure helps to reduce intraoperative bleeding and offers a better surgical field. However, such a procedure has substantial risks like iatrogenic embolization of intracranial vessels via external and internal carotid arteries. The gravest complication of macroglossia is airway compromise, but the quality of life related morbidities such as speech function, feeding and cosmetics might warrant a surgical solution. In the present case, though the former was not present, the latter set of problems warranted the need for surgical rehabilitation.
We have previously described the criteria and guidelines for approaching macroglossia. Using the principles and guidelines as described, a margin reduction procedure with evisceration of the lesional tissue that was occupying the compartments in the tongue. Careful blunt dissection of the lesional tissue is the key. As the lesion was centrally located in the dorsum of the tongue inside the muscle layers, dissection and evisceration was possible. There is a report of trapezoidal-shaped resection utilizing anterior wedge resection of the tongue to manage a giant cavernous hemangioma of the oral cavity as well as other procedures.,, The elective ligation of right lingual artery and preoperative triamcinolone injection helped to maintain the surgical field clarity during the procedure.
| Conclusion|| |
Successful rehabilitation of a giant haemangioma of the tongue has been presented. Surgical approach remains the only option when the lesion is present in critical sites and other treatments are not a viable option. Surgical outcomes are often predictable given the nature of the surgery but carry substantial risk. However, when successfully planned, it could give the best result.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Ferri G, Fermi M, Maccarrone F, Mattioli F, Persutti L. Giant hemangioma involving the tongue: A surgical strategy to improve quality of life. J Oral MaxillofacSurg MedPathol 2020;32:190-94.
George A, Mani V, Noufal A. Update on the classification of hemangioma. J Oral MaxillofacPathol 2014;18(Suppl S1):117-20.
Barrón-Peña A, Martínez-Borras MA, Benítez-Cárdenas O, Pozos-Guillén A, Garrocho-Rangel A. Management of the oral hemangiomas in infants and children: Scoping review. Med Oral Patol Oral Cir Bucal 2020;25:e252-61.
Balaji SM. Reduction glossectomy for large tongues. Ann MaxillofacSurg 2013;3:167-72.
Calabrese L, Giugliano G, Bruschini R, Ansarin M, Navach V, Grosso E, et al
. Compartmental surgery in tongue tumours: description of a new surgical technique. ActaOtorhinolaryngolItal 2009;29:259-64.
Shuker ST. Resection of giant hemangioma of the tongue utilizing a miniature tourniquet technique. J CraniofacSurg 2016;27:e447-50.
Pranitha V, Puppala N, Deshmukh SN, Jagadesh B, Anuradha S. Cavernous hemangioma of tongue. Management of two cases. J ClinDiagn Res 2014;8:ZD15-17.
Dr. S M Balaji
Director and Consultant Oral and Maxillofacial Surgeon, Balaji Dental and Craniofacial Hospital, 30, KB Dasan Road, Teynampet, Chennai - 600 018, Tamil Nadu
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]