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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 8  |  Issue : 1  |  Page : 37-40

Oral pyogenic granuloma in a port-wine stain: A rare co-occurrence


Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental Sciences, NITTE University, Deralakatte, Mangalore, Karnataka, India

Date of Web Publication30-Mar-2017

Correspondence Address:
Medhini Madi
Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental Sciences, NITTE University, Deralakatte, Mangalore - 575 018, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/srmjrds.srmjrds_51_16

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  Abstract 

Oral pyogenic granuloma is an inflammatory hyperplasia seen in the oral cavity occurring in response to stimuli such as local irritants and hormonal factors. Port-wine stain is a congenital capillary malformation which manifests on the mucosa or skin. Pyogenic granulomas in the oral cavity have rarely been reported to be present in congenital capillary malformations such as port-wine stains. Such a co-occurrence of oral pyogenic granuloma and a port-wine satin has been reported in this case report.

Keywords: Capillary malformation, port-wine stain, pyogenic granuloma


How to cite this article:
Madi M, Babu SG, Achalli S, Castelino R. Oral pyogenic granuloma in a port-wine stain: A rare co-occurrence. SRM J Res Dent Sci 2017;8:37-40

How to cite this URL:
Madi M, Babu SG, Achalli S, Castelino R. Oral pyogenic granuloma in a port-wine stain: A rare co-occurrence. SRM J Res Dent Sci [serial online] 2017 [cited 2022 Sep 25];8:37-40. Available from: https://www.srmjrds.in/text.asp?2017/8/1/37/203483


  Introduction Top


Pyogenic granuloma is a nonneoplastic mass of excess granulation tissue. It arises in reaction to stimuli such as traumatic injury, local irritants, or hormonal factors.[1],[2] Hullihen's description in the year 1844 was possibly the first pyogenic granuloma described in English literature. Hartzell in the year 1904 introduced the term “Pyogenic granuloma” or “Granuloma pyogenicum.”[3],[4]

Congenital low-flow vascular malformations are seen in approximately 0.3–0.5% of newborn infants. These are port-wine stains.[5] Head and neck region is the most common site of occurrence for such lesions.[6] Above 40% of all the cases of port-wine stain are limited to the cutaneous distribution of the trigeminal nerve. Typically, port-wine stain will thicken and darken over time. Nodularity and hypertrophy usually develop in 65% of these cases by the fifth decade of life.[7] The risk of spontaneous bleeding and possibility of formation of a pyogenic granuloma increases as hypertrophy develops.[8]

Here, we report a rare co-occurrence of oral pyogenic granuloma on a port-wine stain.


  Case Report Top


A 33-year-old female reported to the Department of Oral Medicine and Radiology with a chief complaint of pain in the upper right back region of the mouth for 2–3 years. The patient noticed a small growth near the upper right back teeth 3 years back. Initially, the growth was small which grew to reach the present size. Dull intermittent pain was present in the region of the growth which increases at the time of brushing the teeth. There was bleeding on brushing. There was no history of discharge or ulcerations on the growth. Detailed medical history revealed that the patient had reddish discoloration of the skin of the face on the right side since birth. It was lighter in shade when the patient was younger in age and has reached the present shade over the years. The patient also gave a history of inability and discomfort to sleep facing the right side.

On extraoral examination, reddish discoloration was seen involving the right side of the face extending from the midline anteriorly, extending up to the right ear posteriorly, superiorly extending above the right eyebrows, and inferiorly extending up to the line joining the angle of the mouth on the right side till the right ear lobe [Figure 1].
Figure 1: Clinical photograph showing port-wine stain on the right side of the face

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Intraorally, the reddish discoloration involves the right buccal mucosa and right side of the soft palate. Intraoral examination revealed a lobulated, sessile growth measuring approximately 1.5 cm × 1.5 cm in dimension, pink, with no surface discharge, bleeding or ulceration in the region of 14, 15. The growth was soft to firm in consistency and tender on palpation. Pus discharge was seen at the region of the apex of 14 [Figure 2]. Blanching was seen on diascopy [Figure 3].
Figure 2: Clinical photograph showing port-wine stain on the right buccal mucosa, right side of the soft palate, and oral pyogenic granuloma in the region of 14, 15

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Figure 3: Photograph showing blanching on diascopy test

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A provisional diagnosis of port-wine stain on the right side of the face, right buccal mucosa, and soft palate was arrived at. To rule out the possibility of its association with syndromes such as Sturge–Weber–Dimitri and Klippel–Trenaunay syndromes, the patient was radiologically evaluated. Considering the clinical features of the intraoral growth arising from the port-wine stain, a provisional diagnosis of a pyogenic granuloma was rendered. Peripheral giant cell granuloma, peripheral ossifying fibroma, traumatic fibroma, and hemangioma were considered as differential diagnosis. Histopathological evaluation was done to arrive at a confirmed diagnosis.

Panoramic radiograph showed normal condylar and coronoid process grossly decayed teeth with respect to 18, 15, 14, 27, 38, 48, decayed teeth with respect to 21, 22, root stump with respect to 47, impacted tooth with respect to 28, and missing 37. It also revealed hazy radiolucency at the apex of 14, suggestive of periapical abscess [Figure 4]. Lateral cephalogram and posteroanterior view of the skull showed no abnormalities [Figure 5]a and [Figure 5]b. Panoramic radiograph, lateral cephalogram, and a posteroanterior view of the skull were done to rule out intracranial extension that eliminated possibilities of a syndromic association.
Figure 4: Panoramic radiograph

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Figure 5: (a and b) Lateral cephalogram and posteroanterior view of the skull

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The growth was excised using soft-tissue diode laser and sent for histopathological examination. The diode laser provided an optimum combination of clean removal of the tissue and hemostasis. Histopathological examination of the H and E stained slides shows epithelium and connective tissue. Epithelium is parakeratinized, stratified squamous type showing hyperplasia in few areas and also shows break in the epithelium in one area. Connective tissue shows fibroblasts, numerous blood vessels of varying size with proliferating endothelial cells, and mixed inflammatory cells in the form of plasma cells, lymphocytes, and neutrophils. The features were suggestive of pyogenic granuloma [Figure 6]a and [Figure 6]b. She was advised to re-visit after 5 days but failed to keep up any further appointment.
Figure 6: (a) Photomicrograph with ×10 magnification suggestive of pyogenic granuloma. (b) Photomicrograph with ×40 magnification suggestive of pyogenic granuloma

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  Discussion Top


Cutaneous pyogenic granulomas arising in port-wine stains have been described in literature. Sheehan and Lesher Jr. conducted a literature search. Their findings revealed that 21 cases including their own case report of cutaneous pyogenic granulomas arising in port-wine stains have been documented.[8] A case of an oral pyogenic granuloma occurring within the gingiva involved in a port-wine stain is a rare co-occurrence.

A port-wine stain is a macular telangiectatic patch. The lesion is existent at birth and remains throughout life. They represent hamartomatous capillary malformations. They leave deep red hue on the skin or the mucosa. These are considered as low-flow vascular malformations. They can occur on any part of the body. Most commonly, they affect the face in the distribution of the trigeminal nerve. To begin with, the lesions are pale, pink patches. They mature with time and change into a violaceous color. They may even remain static or lighten in color. Vascular ectasia may even convert the patch-like lesion into a nodular growth.[9],[10]

Pyogenic granuloma is a common tumor-like growth of the oral cavity or skin. It is considered to be nonneoplastic in nature. Pyogenic granuloma is a smooth, lobulated, and exophytic lesion. It manifests itself as small, red, erythematous papules having either a pedunculated or a sessile base. The size of a pyogenic granuloma varies from being just a few millimeters to several centimeters. In rare occasions, the size exceeds 2.5 cm reaching its maximum size within weeks or months and remains in the same size for an indefinite period of time. Clinically, the lesion on few occasions develops slowly, sometimes rapidly and asymptomatic. Depending on the age of the lesion, the color changes from pink to red to purple. The surface is characteristically ulcerated and friable.[11] The younger lesions are highly vascular. Considerable bleeding may be seen on minor trauma. The older lesions are more collagenized and hence appear pink. The hormonal influences of progesterone and estrogen stimulate the growth to grow faster. This may be the reason behind the high incidence of pyogenic granuloma in women, particularly in pregnant women and those consuming oral contraceptive drugs.[12]

Expression of angiogenic factors is increased and granuloma cell apoptosis is decreased due to estrogen and progesterone. Increased blood flow to the region of the pyogenic granuloma due to the enlarged capillaries of the port-wine stain may have predisposed the patient to develop pyogenic granuloma.[13]

Various treatment options have been suggested for pyogenic granuloma. They include curettage, cryotherapy, corticosteroid injection, sclerotherapy, laser ablation, and surgical excision. Sclerotherapy with sodium tetradecyl sulfate is a new treatment approach that has been described in literature. Pyogenic granulomas associated with port-wine stains often recur. Therefore, complete surgical excision is the generally favored treatment option.[1] Our patient underwent the removal of pyogenic granuloma by diode laser, and the perioperative, intraoperative, and postoperative period was uneventful.


  Conclusion Top


Port-wine stain remains permanent in the lifetime of the patient. It can cause major cosmetic and esthetic problems. Although various treatment modalities are available, complete clearance is very rare. Early recognition of possible syndromic association is of great help for the patient to lead a healthy and relatively normal life. Recognition and immediate treatment of possible lesions associated with this stain is also of utmost importance.

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.
Jafarzadeh H, Sanatkhani M, Mohtasham N. Oral pyogenic granuloma: A review. J Oral Sci 2006;48:167-75.  Back to cited text no. 1
    
2.
Reddy NR, Kumar PM, Selvi T, Nalini HE. Management of recurrent post-partum pregnancy tumor with localized chronic periodontitis. Int J Prev Med 2014;5:643-7.  Back to cited text no. 2
    
3.
Hullihen SP. Case of aneurysm by anastomosis of the superior maxillae. Am J Dent Sc 1844;4:160-2.  Back to cited text no. 3
    
4.
Hartzell MB. Granuloma pyogenicum. J Cutan Dis Syph 1904;22:520-5.  Back to cited text no. 4
    
5.
Alper JC, Holmes LB. The incidence and significance of birthmarks in a cohort of 4, 641 newborns. Pediatr Dermatol 1983;1:58-68.  Back to cited text no. 5
    
6.
Orten SS, Waner M, Flock S, Roberson PK, Kincannon J. Port-wine stains. An assessment of 5 years of treatment. Arch Otolaryngol Head Neck Surg 1996;122:1174-9.  Back to cited text no. 6
    
7.
Geronemus RG, Ashinoff R. The medical necessity of evaluation and treatment of port-wine stains. J Dermatol Surg Oncol 1991;17:76-9.  Back to cited text no. 7
    
8.
Sheehan DJ, Lesher JL Jr., Pyogenic granuloma arising within a port-wine stain. Cutis 2004;73:175-80.  Back to cited text no. 8
    
9.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral & Maxillofacial Pathology. 2nd ed. Philadelphia: WB Saunders; 2002. p. 467-8.  Back to cited text no. 9
    
10.
Klapman MH, Yao JF. Thickening and nodules in port-wine stains. J Am Acad Dermatol 2001;44:300-2.  Back to cited text no. 10
    
11.
Epivatianos A, Antoniades D, Zaraboukas T, Zairi E, Poulopoulos A, Kiziridou A, et al. Pyogenic granuloma of the oral cavity: Comparative study of its clinicopathological and immunohistochemical features. Pathol Int 2005;55:391-7.  Back to cited text no. 11
    
12.
Ojanotko-Harri AO, Harri MP, Hurttia HM, Sewón LA. Altered tissue metabolism of progesterone in pregnancy gingivitis and granuloma. J Clin Periodontol 1991;18:262-6.  Back to cited text no. 12
    
13.
Yuan K, Wing LY, Lin MT. Pathogenetic roles of angiogenic factors in pyogenic granulomas in pregnancy are modulated by female sex hormones. J Periodontol 2002;73:701-8.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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Abstract
Introduction
Case Report
Discussion
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