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   Table of Contents      
BRIEF COMMUNICATION
Year : 2017  |  Volume : 65  |  Issue : 11  |  Page : 1221-1223

Congenital capillary hemangioma arising from palpebral conjunctiva of a neonate


1 Department of Ophthalmology, Coimbatore Medical College Hospital, Coimbatore, Tamil Nadu, India
2 Department of Paediatric Surgery, Coimbatore Medical College Hospital, Coimbatore, Tamil Nadu, India

Date of Submission23-Jun-2017
Date of Acceptance25-Sep-2017
Date of Web Publication13-Nov-2017

Correspondence Address:
S Padmanaban
Department of Ophthalmology, Coimbatore Medical College Hospital, Coimbatore - 641 018, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_487_17

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  Abstract 

Capillary hemangioma is the most common benign vascular eyelid tumor in childhood. The periocular lesion appears within the first few weeks after birth and usually has superficial or deep components. Primary conjunctival capillary hemangiomas are rarely reported. We present the case of a 2-day-old child with a pedunculated capillary hemangioma arising from superior palpebral conjunctiva. A complete surgical excision was performed under general anesthesia, and the child was asymptomatic at follow-up of two months.

Keywords: Congenital capillary hemangioma, conjunctival capillary hemangioma, management, pedunculated capillary hemangioma


How to cite this article:
Padmanaban S, Sumathi P, Kandoth P, Dharmendra R P. Congenital capillary hemangioma arising from palpebral conjunctiva of a neonate. Indian J Ophthalmol 2017;65:1221-3

How to cite this URL:
Padmanaban S, Sumathi P, Kandoth P, Dharmendra R P. Congenital capillary hemangioma arising from palpebral conjunctiva of a neonate. Indian J Ophthalmol [serial online] 2017 [cited 2019 Dec 6];65:1221-3. Available from: http://www.ijo.in/text.asp?2017/65/11/1221/218063

Capillary hemangioma is the most common benign vascular eyelid tumor in childhood.[1] It is present in 1%–4% of all births[2] and is more common in premature infants and often following chorionic villus sampling.[3] It is usually a cutaneous, subcutaneous, or deep orbital lesion and commonly presents a few weeks after birth. The usual clinical course of infantile cutaneous or subcutaneous hemangiomas includes an initial engorgement (age 6–12 months) followed by regression (age 1–7 years). It usually regresses spontaneously and hence is kept under close observation. Active intervention is performed only if the lesion is very extensive and causes amblyopia, mechanical ptosis, exposure keratopathy, or optic neuropathy. Treatment modalities for cutaneous or subcutaneous hemangiomas include topical[4] and oral beta blockers,[5],[6],[7],[8] local and oral steroids,[9],[10] surgical excision, immunotherapy,[11] laser photocoagulation with pulsed dye laser,[3] and embolization.[12] Although conjunctiva may be involved as a part of capillary hemangioma of the lid, isolated conjunctival lesion is rare and its congenital presence is rarer. In a series of 1463 cases of conjunctival tumors studied, only 10 were capillary hemangiomas, forming 16% of conjunctival vascular tumors, only 1% of all conjunctival tumors.[13] Usually, hemangiomas of conjunctiva are small and require only observation. Chang and Estes, in a recent paper, report usage of topical timolol in the management of small bulbar conjunctival hemangioma.[14] Oral propranolol was also recently reported to be in use in the treatment of a case of bilateral sessile infantile hemangioma of posterior lamella of upper lid by Syed Ali Raza Rizvi et al., with clearing of visual axis within 12 weeks of treatment.

We report a case of capillary hemangioma in a newborn, which could be treated completely by surgical excision due to its unique pedunculated morphology. Such a case of congenital pedunculated conjunctival capillary hemangioma has not been reported so far to the best of our knowledge.


  Case Report Top


A 2-day-old female baby presented to our pediatric surgery department with a red mass arising from left upper eyelid since birth [Figure 1]. In the antenatal history elicited, mother was found to be a primipara who had regular antenatal checkups, with no history of any invasive interventions, and the baby was born out of full-term normal vaginal delivery with no perinatal complications.
Figures 1 and 2: (1) Baby with left eye upper lid pedunculated swelling arising from palpebral conjunctiva (2) Upon lifting and resting the swelling on upper lid, thin broad pedicle appreciated

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On examination, the right eye appeared to be normal. Examination of the left eye showed a red, pedunculated, highly vascular, nontender mass of 2.5 cm × 1.5 cm arising from the palpebral conjunctiva of upper lid. The mass had a flattened stalk with terminal lobulations, at the tip of which, there was dried up mixture of blood and discharge. On upper eyelid eversion, the base of peduncle was found to be at 4 mm from lid margin. The lesion appeared to cause mechanical ptosis and covered the visual axis. The eyelid opening appeared inadequate [Figure 2]. There was minor bleeding from the surface of the swelling on manipulation. The rest of the conjunctiva was normal except for mild congestion. The cornea of the left eye was clear. There was no evidence of similar cutaneous lesions on the rest of the body.

A provisional diagnosis of capillary hemangioma was made. There was also the possibility of pyogenic granuloma and rare possibility of rhabdomyosarcoma. Complete hemogram of the baby showed normal hemoglobin (17.3 g%), white blood cell (11,000/μL), and platelet (2.5 lakhs/μL) levels. Ultrasonogram (USG) showed high-internal reflectivity with irregular acoustic structures suggestive of capillary hemangioma. USG of cranium and abdomen were done to rule out internal hemangiomas but were found to be normal.

Taking into consideration the pedunculated nature, unsightly appearance, possibility of developing amblyopia due to the central location and also to obtain a histopathological diagnosis (to rule out any other tumor), surgical removal was finalized as the treatment. On postnatal day 6, the baby was prepared for general anesthesia. Under anesthesia, detailed examination of the conjunctiva of both eyes was done to rule out deeper lesions and bilaterality. Excision of the mass was done with bipolar electrocautery device by a team consisting of ophthalmologist and pediatric surgeon. It was cut close to tarsal plate, and adequate hemostasis was obtained [Figure 3].
Figures 3 and 4: (3) On table image of baby after excision of lesion (4) Baby on first postoperative day

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On the 1st postoperative day, the eye was quiet with adequate eyelid opening, clear cornea, and normal fundus [Figure 4]. On follow-up visits, there was no conjunctival scarring and cornea appeared clear. Multiple sections studied showed a cellular neoplasm composed of plump endothelial cells with well-canalized and poorly canalized vessels, confirming the diagnosis of cellular infantile hemangioma [Figure 5] and [Figure 6]. The baby was followed up for 2 months postsurgery and showed no deformities and had adequate lid movements.
Figures 5 and 6: (5) HPE-Multiple sections studied showed a cellular neoplasm composed of plump endothelial cells with well-canalized and poorly canalized vessels, confirming the diagnosis of cellular infantile hemangioma (6) Multiple sections studied showed a cellular neoplasm composed of plump endothelial cells with well-canalized and poorly canalized vessels, confirming the diagnosis of cellular infantile hemangioma

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  Discussion Top


Capillary hemangioma is common in eyelids but uncommon in the conjunctiva. These are benign vascular tumors which consist of endothelial cell proliferation. It usually presents a few weeks after birth, sometimes at birth, grows for several months, and later regresses spontaneously over years. These occur singly or as part of syndromes (PHACES). Capillary hemangiomas of conjunctiva are rare, and mostly, the lesions are small, demanding only close watch. Usually, cutaneous and orbital hemangiomas are treated with oral steroids or propranolol. Recently, cases of effective management of small conjunctival hemangiomas with topical timolol have been reported. The current literature also shows management of sessile palpebral conjunctival capillary hemangiomas with oral beta blockers. In our case, the baby was brought to us with a red, lobulated mass with stalk present since birth and arising exclusively from the upper eyelid conjunctiva. In order to prevent amblyopia, the lesion was surgically excised, which was possible due to its pedunculated nature.


  Conclusion Top


Conjunctival capillary hemangioma of pedunculated nature has not been reported in literature so far to the best of our knowledge.

Acknowledgment

We would like to thank Dr. G Rajamani, Professor and Head of the Department, Department of Paediatric surgery, Coimbatore Medical College Hospital, Coimbatore, for the technical aid.

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.
Albert DM, Jakobiec FA. Principles and Practice of Opthalmology. 2nd ed., Vol. 5. Philadelphia: W. B. Saunders; 2000. p. 4436.  Back to cited text no. 1
    
2.
Sethuraman G, Yenamandra VK, Gupta V. Management of infantile hemangiomas: Current trends. J Cutan Aesthet Surg 2014;7:75-85.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Drolet BA, Esterly NB, Frieden IJ. Hemangiomas in children. N Engl J Med 1999;341:173-81.  Back to cited text no. 3
[PUBMED]    
4.
Chambers CB, Katowitz WR, Katowitz JA, Binenbaum G. A controlled study of topical 0.25% timolol maleate gel for the treatment of cutaneous infantile capillary hemangiomas. Ophthal Plast Reconstr Surg 2012;28:103-6.  Back to cited text no. 4
[PUBMED]    
5.
Vassallo P, Forte R, Di Mezza A, Magli A. Treatment of infantile capillary hemangioma of the eyelid with systemic propranolol. Am J Ophthalmol 2013;155:165-7000.  Back to cited text no. 5
[PUBMED]    
6.
Thoumazet F, Léauté-Labrèze C, Colin J, Mortemousque B. Efficacy of systemic propranolol for severe infantile haemangioma of the orbit and eyelid: A case study of eight patients. Br J Ophthalmol 2012;96:370-4.  Back to cited text no. 6
    
7.
Missoi TG, Lueder GT, Gilbertson K, Bayliss SJ. Oral propranolol for treatment of periocular infantile hemangiomas. Arch Ophthalmol 2011;129:899-903.  Back to cited text no. 7
[PUBMED]    
8.
Brad B. Kanski's Clinical Ophthalmology. 8th ed. China: Elsevier; 2016. p. 101.  Back to cited text no. 8
    
9.
Schwartz SR, Kodsi SR, Blei F, Ceisler E, Steele M, Furlan L, et al. Treatment of capillary hemangiomas causing refractive and occlusional amblyopia. J AAPOS 2007;11:577-83.  Back to cited text no. 9
    
10.
Coats DK, O'Neil JW, D'Elia VJ, Brady-McCreery KM, Paysse EA. SubTenon's infusion of steroids for treatment of orbital hemangiomas. Ophthalmology 2003;110:1255-9.  Back to cited text no. 10
    
11.
Wilson MW, Hoehn ME, Haik BG, Rieman M, Reiss U. Low-dose cyclophosphamide and interferon alfa 2a for the treatment of capillary hemangioma of the orbit. Ophthalmology 2007;114:1007-11.  Back to cited text no. 11
[PUBMED]    
12.
Ooi KG, Wenderoth JD, Francis IC, Wilcsek GA. Selective embolization and resection of a large noninvoluting congenital hemangioma of the lower eyelid. Ophthal Plast Reconstr Surg 2009;25:111-4.  Back to cited text no. 12
[PUBMED]    
13.
Shields JA, Shields CL. Eyelid, Conjunctival and Ocular Tumors: An Atlas and Textbook. 2nd ed. Philadelphia, PA: Lippincott Williams and Wilkins; 2008. p. 250-445.  Back to cited text no. 13
    
14.
Chang TC, Estes R. Beta blocker treatment of infantile conjunctival hemangiomas – Observations from 2 cases. J AAPOS 2014;18:80-2.  Back to cited text no. 14
[PUBMED]    


    Figures

  [Figure 1], [Figure 3], [Figure 5]



 

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