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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 11  |  Issue : 1  |  Page : 50-53

Propranolol therapy for the management of oral lobular capillary hemangioma in a 2-year-old preschooler: A case report and review


Department of Pedodontics and Preventive Dentistry, University College of Medical Sciences, Guru Teg Bahadur Hospital, Delhi, India

Date of Submission17-Dec-2019
Date of Acceptance23-Jan-2020
Date of Web Publication11-Mar-2020

Correspondence Address:
Dr. Puja Sabherwal
Department of Pedodontics and Preventive Dentistry, University College of Medical Sciences, Guru Teg Bahadur Hospital, Delhi
India
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DOI: 10.4103/srmjrds.srmjrds_91_19

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  Abstract 

Lobular capillary hemangioma (LCH), a histological variant of pyogenic granuloma (PG) is an inflammatory hyperplasia commonly seen on oral mucosa as a reaction to local irritants, trauma or hormonal influences. The term “PG” is a misnomer as it is neither pus filled nor representative of a granuloma histologically. It is an inflammatory lesion composed of proliferating vascular, fibrous, and inflammatory tissue. The benign tumor has varied clinical and histopathological presentations and occurs frequently on the gingiva in the oral cavity. The article presents a case report where 2-week propranolol therapy was used to manage oral LCH (with a rare site of presentation) in a 2-year-old preschooler with a 9-month follow-up showing no recurrence followed by review of literature.

Keywords: Literature review, lobular capillary hemangioma, preschooler, propanolol therapy


How to cite this article:
Kalra N, Tyagi R, Khatri A, Singh HV, Sabherwal P, Sharma M. Propranolol therapy for the management of oral lobular capillary hemangioma in a 2-year-old preschooler: A case report and review. SRM J Res Dent Sci 2020;11:50-3

How to cite this URL:
Kalra N, Tyagi R, Khatri A, Singh HV, Sabherwal P, Sharma M. Propranolol therapy for the management of oral lobular capillary hemangioma in a 2-year-old preschooler: A case report and review. SRM J Res Dent Sci [serial online] 2020 [cited 2020 Jun 3];11:50-3. Available from: http://www.srmjrds.in/text.asp?2020/11/1/50/280381


  Introduction Top


Lobular capillary hemangioma (LCH) also known as pyogenic granuloma (PG) is a benign, vascular lesion primarily due to reactive inflammatory response due to local irritants. It is a hyperplastic, soft tissue benign tumor caused by a vast variety of factors, including local irritants, traumatic injuries, and hormonal factors.[1] The most common predilection is among teenagers and young adults, with a male: female ratio of 2:16 showing marked female predilection. LCH of the oral cavity involves the gingiva in 70-75% cases. It may also occur on the buccal mucosa, tongue, lips, palate, and other lining mucosa of the oral cavity and can vary in size from few millimeters to several centimeters.[2]

Several treatment options include a conservative management regime which includes the removal of all possible irritants, medical management, surgical excision, curettage, use of cautery and lasers.

The paper presents a case of a 2-year-old preschooler treated using 1% topical propranolol therapy showing regression of the lesion after 2 weeks with no recurrence on a follow-up of 9 months.


  Case Report Top


A 2-year-old boy reported to the Department of Pedodontics and Preventive Dentistry at UCMS and GTB Hospital, Delhi, with the chief complaint of a growth present behind the left back tooth region since 4 months. It was pea-sized to begin with and increased to an ovoid size mass approximately 2.5 cm × 1.5 cm in dimension. Episodes of bleeding were present, and the tendency of the child to chew and pull at the mass was reported by the parents particularly when alone. On observation, a reddish-pink lobulated mass with evident ulceration of the overlying mucosa was seen [Figure 1]. An unusually sharp lingual cusp was present with respect to erupting 74. No systemic illness or family history was reported.
Figure 1: (a) Extraoral view. (b) Intraoral view. (c) Intraoral view post recurrence of the lesion

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Oral hygiene instructions were demonstrated and parental consent was obtained. Coronoplasty was done for 74 along with localized oral prophylaxis for 74 regions. Routine hemogram tests were normal. Incisional biopsy was performed under local anesthesia, and the specimen was sent for histopathological investigation in 10% formalin as the transport medium. The histopathological examination confirmed the diagnosis of LCH revealing parakeratinized epithelium with superficial ulceration with evident lobules of small-sized vessels and inflammatory cell infiltrate of neutrophils and lymphocytes in the connective tissue mucosa [Figure 2].
Figure 2: Lobules of small-sized vessels with superficial ulceration consistent with lobular capillary hemangioma (pyogenic granuloma) (H and E, ×200)

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On further perusal of literature, the topical application of propranolol was seen as a successful measure leading to the regression of PGs on skin and mucosa [Flow Chart 1] and [Flow Chart 2]. Therefore, after due medical clearance, the child was treated with 1% topical propranolol therapy for oral LCH and recalled after 2 weeks.



At the next appointment after 2 weeks, the mass was found to regress. Propranolol application and adjunctive removal of local irritant factors, i.e., the sharp cusp of 74 lead to resolution of the lesion [Figure 1].

After a follow-up of 9 months, the child remained asymptomatic with no recurrence.


  Discussion Top


LCH, a histological variant of “PG,” is an oral disease which appears in the mouth as an overgrowth of tissue due to traumatic injury, repeated irritation, or hormonal influences[3] and it seen commonly in children and adolescents. Termed as “Granuloma telangietacticum” by Gomes et al., PG has two histological types - LCH and non-LCH. LCH usually comprises of proliferating blood vessels in lobules and aggregates, with no edema and capillary dilatation. NLCH type comprises of vascular core which resembles granulation tissue with fibrous tissue and blood vessels. Oral PGs are mainly of LCH type.[4]

The etiological factors may be poor hygiene, infection, trauma such as cheek biting and overhanging restorations.[5] BRAF c.1799T>A, an endothelial cell mutation has been identified as a driving mutation in the pathogenesis of PG.[6] Alpha herpes simplex viridae 1 is linked to its etiology which may reflect its high occurrence in children.

The name “PG” is considered a misnomer since the condition neither involves the presence of pus and nor does it represent a granuloma on histological examination. It is a reactive inflammatory process consisting of proliferating vascular channels, proliferating immature fibroblastic connective tissue, and scattered inflammatory cells in the connective tissue region.[3] The summary of clinical features is shown in [Table 1].[7],[8],[9]
Table 1: Summary of clinical features of lobular capillary hemangioma

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If left untreated, LCHs may undergo fibrous maturation and interfere with esthetics or function. There may be episodes of recurrent bleeding due to trauma.

The differential diagnosis includes hemangioma, vascular malformations, peripheral giant cell granuloma, metastatic cancer, and hyperplastic gingival inflammation which were ruled out by clinical and histopathological examination.[10]

The use of beta-blockers for the management of hemangiomas stemmed from a research in 2008 at the Bordeaux Children's Hospital in France, where children with extensive infantile hemangiomas[10] who were receiving concomitant treatment with propranolol (for obstructive hypertrophic cardiomyopathy) showed regression of the hemangioma on the initiation of propranolol.[11]

For children, topical beta-blocker therapy has become a preferred modality for pediatric PG on skin and mucosa of the body because of minimal adverse effects and good efficacy [Figure 1] and [Figure 2].

Topical Timolol for 12–24 weeks has been used successfully. Other modalities which are the US FDA approved include 1% Propranolol ointment as was in our case. Topical imiquimod 5% cream and ingenol mebutate 0.015% cream are other upcoming modalities which are yet to be approved by the FDA and could be promising modalities in the years to come.[12] The treatment, whether topical or oral, warrants timely monitoring. The systemic absorption may be found after timolol eye drops/topical use of propranolol; thus, patients should be monitored for hypotension, bradycardia, bronchoconstriction, and hypoglycemia.[13] No significant redness, irritation, or scaling of the treated site was reported.[14]

Intralesional injections of 3%–4% sodium tetradecyl sulfate, a sclerosant has proven excellent results[15] however may be problematic due to the cooperation of infants and children.

Alternative treatment options include surgical removal, curettage, cauterization, and lasers.

Surgical excision is indicated in case of excessive bleeding, trauma, and cosmetic and functional distress. Excision for lesions on gingiva is done up to the mucoperiosteum with proper scaling and root planning to alleviate all possible irritants.[16]

Cryotherapy, silver nitrate, and soft-tissue lasers are other modalities of treatment. Cryotherapy or silver nitrate therapy may be effective for very small lesions.[12] Therapy using a diode laser at wavelengths of 808–980 nm is preferred as it has advantages such as hemostasis, minimal swelling, scarring, and patient comfort.[17]

Follow-up visits are required only if the lesion recurs. In case of recurrence, the lesion may be managed with any alternate, as discussed above.


  Conclusion Top


LCH is a localized, benign exophytic growth commonly present in the oral cavity in children, adolescents, and pregnant females. The management may be based on a planned and programmed approach which includes detailed case history and diagnosis confirmation and choosing the treatment modality suitable for the patient and lesion. Early diagnosis and initiation of treatment along with proper oral hygiene and a strict follow-up care would minimize the recurrence.

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.
Ximenes M, Triches TC, Cardoso M, Bolan M. Pyogenic granuloma on the tongue: A pediatric case report. Gen Dent 2013;61:27-9.  Back to cited text no. 1
    
2.
Mahajan A, Singhal P, Poonam. Lobular capillary hemangioma in a child: A case report and literature review. Open Access Sci Rep. 2012;1:265.  Back to cited text no. 2
    
3.
Venugopal S, Shobha KS, Netravathi TD. Pyogenic granuloma – A case report. J Dent Sci Res 2010;1:80-5.  Back to cited text no. 3
    
4.
Gomes SR, Shakir QJ, Thaker PV, Tavadia JK. Pyogenic granuloma of the gingiva: A misnomer? A case report and review of literature. J Indian Soc Periodontol 2013;17:514-9.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Verma PK, Srivastava R, Baranwal HC, Chaturvedi TP, Gautam A, Singh A. “Pyogenic granuloma – Hyperplastic lesion of the gingiva: Case reports”. Open Dent J 2012;6:153-6.  Back to cited text no. 5
    
6.
Groesser L, Peterhof E, Evert M, Landthaler M, Berneburg M, Hafner C. BRAF and RAS mutations in sporadic and secondary pyogenic granuloma. J Invest Dermatol 2016;136:481-6.  Back to cited text no. 6
    
7.
Amirchaghmaghi M, Falaki F, Mohtasham N, Mozafari PM. Extragingival pyogenic granuloma: A case report. Cases J 2008;1:371.  Back to cited text no. 7
    
8.
Nevile BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: W.B. Saunders; 2004. p. 437-95.  Back to cited text no. 8
    
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de Giorgi V, Sestini S, Nardini P, Carli P. A 42-year-old man with a rapidly growing lesion of the soft palate. CMAJ 2005;173:367.  Back to cited text no. 9
    
10.
Ganesan A, Kumar NG, Azariah E, Asokan GS. Oral pyogenic granuloma: A case report and a comprehensive review. SRM J Res Dent Sci 2015;6:250-6.  Back to cited text no. 10
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11.
Léauté-Labrèze C, Dumas de la Roque E, Hubiche T, Boralevi F, Thambo JB, Taïeb A. Propranolol for severe hemangiomas of infancy. N Engl J Med 2008;358:2649-51.  Back to cited text no. 11
    
12.
Wine Lee L, Goff KL, Lam JM, Low DW, Yan AC, Castelo-Soccio L. Treatment of pediatric pyogenic granulomas using β-adrenergic receptor antagonists. Pediatr Dermatol 2014;31:203-7.  Back to cited text no. 12
    
13.
de Graaf M, Breur JM, Raphaël MF, Vos M, Breugem CC, Pasmans SG. Adverse effects of propranolol when used in the treatment of hemangiomas: A case series of 28 infants. J Am Acad Dermatol 2011;65:320-7.  Back to cited text no. 13
    
14.
Mashiah J, Hadj-Rabia S, Slodownik D, Harel A, Sprecher E, Kutz A. Effectiveness of topical propranolol 4% gel in the treatment of pyogenic granuloma in children. J Dermatol 2019;46:245-8.  Back to cited text no. 14
    
15.
Khaitan T, Sinha R, Sarkar S, Kabiraj A, Ramani D, Sharma M. Conservative approach in the management of oral pyogenic granuloma by sclerotherapy. J Indian Acad Oral Med Radiol 2018;30:46-51.  Back to cited text no. 15
  [Full text]  
16.
Nevile BW, Damm DD, Allen CM, Bouquot JE. In: Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: W.B. Saunders; 2004. p. 437-95.  Back to cited text no. 16
    
17.
González S, Vibhagool C, Falo LD Jr., Momtaz KT, Grevelink J, González E. Treatment of pyogenic granulomas with the 585 nm pulsed dye laser. J Am Acad Dermatol 1996;35:428-31.  Back to cited text no. 17
    


    Figures

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    Tables

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