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Table of Contents   
CASE REPORT  
Year : 2021  |  Volume : 32  |  Issue : 1  |  Page : 131-133
A very rare case of bilateral capillary hemangioma of lower lip and face: A case report


Department of Oral Medicine and Radiology, Vananchal Dental College and Hospital, Garhwa, Jharkhand, India

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Date of Submission18-Jul-2018
Date of Decision01-Nov-2018
Date of Acceptance25-Apr-2019
Date of Web Publication13-Jul-2021
 

   Abstract 


Hemangioma is a relatively common benign proliferation of blood vessel that primarily develops during childhood. It is a most common benign tumor of vascular origin is the region of head and neck. The common sites are lips, tongue, buccal mucosa, and palate in oral cavity. Due to aesthetic concern and appearance problems of the hemangioma of lower lip and face it requires appropriate management. This is a case report of a rare hemangioma of lower lip and face bilaterally with involvement of mandible, floor of mouth, oropharynx, anterior 2/3rd of tongue parotid/submandibular glands in a 46-years-old female along the course of facial artery bilaterally with arterial feeders from the facial artery bilaterally, and draining into the jugular veins. This type of case is being reported first time in English literature.

Keywords: Angiography, artery, bilateral, capillary, face, head, hemangioma, neck imaging

How to cite this article:
Ujala UA, Diwakar N R. A very rare case of bilateral capillary hemangioma of lower lip and face: A case report. Indian J Dent Res 2021;32:131-3

How to cite this URL:
Ujala UA, Diwakar N R. A very rare case of bilateral capillary hemangioma of lower lip and face: A case report. Indian J Dent Res [serial online] 2021 [cited 2021 Aug 3];32:131-3. Available from: https://www.ijdr.in/text.asp?2021/32/1/131/321376



   Introduction Top


One of the most common congenital and neonatal abnormalities is vascular lesions. If the vascular lesion is due to abnormalities in blood vessels or endothelial cell proliferation it is termed as benign vascular lesion.[1],[2] These are also termed as hemangiomas or vascular malformations. Hemangioma is a term that consists of a heterogeneous group of clinical benign vascular lesions that have similar histological features.[3]

The word “hemangioma” has been widely used in the dental and medical literature with reference to a variety of different vascular anomalies which has traditionally led to a significant amount of confusion regarding the nomenclature of these lesions.[2] The benign, localized tumor of the blood vessels is called as hemangioma.[4] Most of the benign vascular lesions occurring in the head and neck region usually have a malformation, hamartomatous basis.[1],[2],[5] Vascular malformations are categorized into arterial, venous, and capillary types and are subcategorized by their relative flow velocity.

A careful imaging plan is necessary for the evaluation of these lesions. The role of imaging is much more vital in the diagnosis of lesions covered by skin.[6] Magnetic resonance imaging (MRI) techniques should be the initial choice and in follow-up studies.[7],[8] Size and volume of lesion is important in hemangioma, in order to establish growth pattern and response to therapy, if employed. Vascular pattern, including feeding arteries, draining vein, and matrix, is of importance to the interventional radiologist, if specific therapy is envisaged.[6]

Here we present a case report of a rare hemangioma of lower lip and face bilaterally with involvement of mandible, floor of mouth, oropharynx, anterior 2/3rd of tongue parotid/submandibular glands in a 46-years-old female along the course of facial artery bilaterally with arterial feeders from the facial artery bilaterally, and draining into the jugular veins. This report intensively uses interpretations and analysis of radiological images to come to diagnosis and plan a treatment. This type of case is being reported for the first time in english language literature.


   Case Report Top


A 46-year-old female patient reported to Department of Oral Medicine and Radiology of Vananchal Dental College, Garhwa, India for evaluation and treatment of reddish purple discoloration and swelling of lower lips, and chin since birth [Figure 1] and [Figure 2].
Figure 1: Front profile of the patient showing bilateral swelling in lower lips and adjacent areas of chin along with reddish purple discoloration in the areas surrounding lips and chin, lower lip is bulky and hypertrophied, surface appears corrugated

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Figure 2: Side profile of the patient left and right showing drooping of lower lips and extent of the lesion bilaterally. Mandible is enlarged, elongated, and hypertrophied

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According to the patient she had swelling in lower lips, and adjacent areas of chin along with reddish purple discoloration in the areas surrounding lips and chin since birth, initially the swelling was small in size and is gradually progressing and has attained present size. A 23 years back patient visited Manipal Hospital for the same, where lip reconstruction was done, initially the lesion subsided but slowly it recurred back [Figure 3], [Figure 4], [Figure 5].
Figure 3: Childhood photograph of patient at age 9 years

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Figure 4: Photograph after 1s t reconstruction surgery which was done at age 23 years at Manipal Hospital, showing that result was not satisfactory

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Figure 5: Photograph of patient at age 26 years and 30 years showing gradual increase in size and extent of lesion along with increase in size of mandible and lips bulkiness

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There is no relevant past medical and family history recorded. On inspection, reddish-purple discoloration of overlying skin extending from the half of the pinna to the neck at a point 1 cm from the clavicle extending to chin bilaterally [Figure 1] and [Figure 2]. Almost all the area supplied by the facial artery is having the discoloration. The pattern of the discoloration is bilaterally symmetrical. The lower lip is hypertrophied, bulky, appears swollen and is drooping. The lower anterior teeth are visible. The surface of the skin on the affected area is rough and corrugated [Figure 1] and [Figure 2]. There is no functional loss of the lower lip present. The mandible appears to be hypertrophied and enlarged [Figure 2] on palpation, the lower lip is non-tender bulky, and normal in consistency.

The provisional diagnosis of haemangioma was considered. Other pathologic entities that were included in the differential diagnoses were malignancies, trauma, and/or infection (bacterial, viral, and fungal), enlargement due to drugs.

MRI scan with angiography of face and neck was performed using following protocol: 1. Axial and coronal STIR sequences, 2. Post contrast axial T1 FATSAT sequences, 3. Post contrast dynamic MR angiography [Figure 6] and [Figure 7].
Figure 6: Short inversion time inversion recovery (STIR) images of MRI scan with angiography of head and neck showing show ill-defined lobulated hyperdense areas involving entire lower lip, left cheek, masticatory muscles, parotid/submandibular glands, left lateral wall of oropharynx, floor of mouth and anterior 2/3rd of tongue predominantly on the left side. Small calcification is noted within lower lip

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Figure 7: Short inversion time inversion recovery (STIR) images of MRI scan with angiography of head and neck showing minimal thinning/cortical erosion of anterior part of mandibular body

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The short inversion time inversion recovery (STIR) images show ill defined, lobulated and hyperdense areas involving entire lower lip, left cheek, masticatory muscles, parotid/subamandibular glands, left lateral wall of oropharynx, floor of mouth, and anterior 2/3rd of tongue predominantly on the left side. Small calcification is noted within lower lip [Figure 6], Minimal thinning/cortical erosion of anterior part of mandibular body is noted [Figure 7].

The Time Resolved Imaging of Contrast Kinetics (TRICKS) shows few arterial feeders from bilateral facial arteries (left > right) are seen supplying the lesion, small veins are seen draining lesion into bilateral retromandibular and internal jugular veins (left > right) [Figure 8].
Figure 8: Time Resolved Imaging of Contrast Kinetics shows few arterial feeders from bilateral facial arteries (left > right) are seen supplying the lesion, small veins are seen draining lesion into bilateral retromandibular and internal jugular veins (left > right)

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The skull Antero Posterior (AP) shows relatively elongated hypertrophied mandible [Figure 9].
Figure 9: The skull Antero Posterior (AP) view showing relatively elongated hypertrophied mandible

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Final diagnosis was confirmed as Bilateral Capillary Hemangioma of lower lip and face.


   Management Plan Top


Patient has been advised reconstructive surgery.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Baker LL, Dillon WP, Hieshima GB, Dowd CF, Frieden IJ. Hemangiomas and vascular malformations of the head and neck: MR characterization. AJNR 1993;14:307-14.  Back to cited text no. 1
    
2.
Mattassi R, Loose DA, Vaghi M. Hemangiomas and Vascular Malformations. An Atlas of Diagnosis and Treatment. Springer; 2009. p. 17-20.  Back to cited text no. 2
    
3.
Buetow P, Kransdorf M, Moser R, Jelinek J, Berrey B. Radiologic appearance of intramuscular hemangioma with emphasis on MR imaging. Am J Roentgenol 1990;154:563-7.  Back to cited text no. 3
    
4.
Werner J, Dünne A, Folz B, Rochels R, Bien S, Ramaswamy A, et al. Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformations of the head and neck. Eur Arch Otorhinolaryngol 2001;258:141-9.  Back to cited text no. 4
    
5.
Muliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: A classification based on endothelial characteristics. Plastic Reconstruct Surg 1982;69:412-20.  Back to cited text no. 5
    
6.
Bhat V, Bhat V, Salins P. Imaging spectrum of hemangioma and vascular malformations of the head and neck in children and adolescents. J Clin Imaging Sci 2014;4:31.  Back to cited text no. 6
    
7.
Nandaprasad S, Sharada P, Vidya M, Karkera B, Hemanth M, Kaje C. Hemangioma-A review. Internet J Hematol 2009;6.  Back to cited text no. 7
    
8.
Sethuraman V, Kaliamoorthy S, Mathivanan S, Imran A. Venous vascular malformation of lip – A case report. Ann Dent Spec 2014;2:112-4.  Back to cited text no. 8
    

Top
Correspondence Address:
Dr. Ujjwal A Ujala
Department of Oral Medicine, Diagnosis and Radiology, Vananchal Dental College and Hospital, Farathiya, Gharwa, Jharkhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_574_18

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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    Abstract
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   Case Report
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