J Korean Ophthalmol Soc.  2010 Nov;51(11):1513-1519. 10.3341/jkos.2010.51.11.1513.

A Case of Periorbital Infantile Capillary Hemangioma Treated With Propranolol

• Lee EK
• Choung HK
• Kim NJ
• Lee MJ
• Kwon BS
• Khwarg SI

• KMID: 2213988
• DOI: http://doi.org/10.3341/jkos.2010.51.11.1513

Abstract

PURPOSE
To report a case of periorbital infantile capillary hemangioma treated with propranolol.
CASE SUMMARY
A 6-month-old girl visited our clinic for a mass on the right upper eyelid, which had been present since birth. A dome-shaped, red-purple colored mass occupying almost the entire right upper eyelid was observed, and overlying branch-shaped telangiectases were also detected. The visual axis of the right eye was partially obscured, due to the ptotic eyelid, and her ability to fix and follow was poor in the right eye. The patient was diagnosed with infantile capillary hemagioma. Because amblyopia in her right eye was suspected, treatment with propranolol was initiated. After 2.5 months of propranolol treatment, the hemangioma decreased in size and volume dramatically, changed from red-purple to light red color, and softened. The ptosis of the right upper eyelid improved, as well as the ability of the right eye to fix and follow. No significant adverse effects (e.g. hypotension, bradycardia, hypoglycemia, bronchospasm, or elevated liver enzymes) were observed, except transient QTc prolongation during cotreatment with ibuprofen, due to an upper respiratory infection.
CONCLUSIONS
Propranolol should be considered as a treatment option for periorbital infantile capillary hemangioma. Further clinical studies are required to establish the optimal guidelines and long-term outcome.

Keyword

Hemangioma of infancy; Infantile capillary hemangioma; Periorbital hemangioma; Propranolol

MeSH Terms

Amblyopia
Axis, Cervical Vertebra
Bradycardia
Bronchial Spasm
Capillaries
Eye
Eyelids
Hemangioma
Hemangioma, Capillary
Humans
Hypoglycemia
Hypotension
Ibuprofen
Infant
Light
Liver
Parturition
Propranolol
Telangiectasis
Ibuprofen
Propranolol

Figure

• 

  Figure 1. Clinical photographs. (A&B) At the first visit, a large red-purple colored mass occupying almost entire right upper eyelid was noted. The visual axis of the right eye was partially obscured by the ptotic eyelid resulting from the mass. Relatively well circumscribed dome-shaped mass with rubbery consistency was located in the subcutaneous tissue of the right upper eyelid. Telangiectases and large superficial veins were also found. (C&D) After 1 week of propranolol treatment, the mass softened, and its color changed from red-purple to light red. (E&F) After 3 weeks of treatment, the hemangioma showed decrease in size and volume. The visual axis was no longer obscured. (G&H) After 11 weeks of treatment, the ptosis of the right upper eyelid improved, and symmetry of both eyelids' level was attained. The hemangioma flattened, softened and faded in color dramatically.

Reference

References

1. Haik BG, Karcioglu ZA, Gordon RA, et al. Capillary hemangioma (infantile periocular hemangioma). Surv Ophthalmol. 1994; 38:399–426.

2. Margileth AM, Museles M. Cutaneous hemangiomas in children. Diagnosis and conservative management. JAMA. 1965; 194:523–6.

3. Ceisler E, Blei F. Ophthalmic issues in hemangiomas of infancy. Lymphat Res Biol. 2003; 1:321–30.

4. Robb RM. Refractive errors associated with hemangiomas of the eyelids and orbit in infancy. Am J Ophthalmol. 1977; 83:52–8.

5. Sutula FC, Glover AT. Eyelid necrosis following intralesional corticosteroid injection for capillary hemangioma. Ophthalmic Surg. 1987; 18:103–5.

6. Cogen MS, Elsas FJ. Eyelid depigmentation following corticosteroid injection for infantile ocular adnexal hemangioma. J Pediatr Ophthalmol Strabismus. 1989; 26:35–8.

7. Droste PJ, Ellis FD, Sondhi N, et al. Linear subcutaneous fat atrophy after corticosteroid injection of periocular hemangiomas. Am J Ophthalmol. 1988; 105:65–9.

8. Shorr N, Seiff SR. Central retinal artery occlusion associated with periocular corticosteroid injection for juvenile hemangioma. Ophthalmic Surg. 1986; 17:229–31.

9. Zarem HA, Edgerton MT. Induces resolution of cavernous hemangiomas following prednisolone therapy. Plast Reconstr Surg. 1967; 39:76–83.

10. Bennett ML, Fleischer AB Jr, Chamlin SL, et al. Oral corticosteroid use is effective for cutaneous hemangiomas: an evi-dence-based evaluation. Arch Dermatol. 2001; 137:1208–13.

11. Ezekowitz RA, Mulliken JB, Folkman J. Interferon alfa-2a therapy for life-threatening hemangiomas of infancy. N Engl J Med. 1992; 326:1456–63.

12. Rampini E, Rampini P, Occella C, et al. Interferon alpha 2b for treatment of complex cutaneous haemangiomas of infancy: a reduced dosage schedule. Br J Dermatol. 2000; 142:189–91.

13. Frieden IJ, Haggstrom AN, Drolet BA, et al. Infantile hemangiomas: current knowledge, future directions: proceedings of a research workshop on infantile hemangiomas. Pediatr Dermatol. 2005; 22:383–406.

14. Gorst CM, Munnoch DA, Hancock K. Combined treatment of a proliferative periorbital hemangioma with a tunable dye laser and intralesional steroids to prevent deprivation amblyopia. J R Coll Surg Edinb. 2001; 46:234–6.

15. Landthaler M, Hohenleutner U, Abd El-Raheem T. Laser therapy of childhood haemangiomas. Br J Dermatol. 1995; 133:275–81.

16. Braun IF, Levy S, Hoffman JC Jr. The use of transarterial micro-embolization in the management of hemangiomas of the perioral region. J Oral Maxillofac Surg. 1985; 43:239–48.

17. Kim BJ, Kim YD. Surgical treatment of a capillary hemangioma with preoperative embolization. J Korean Ophthalmol Soc. 2002; 43:805–13.

18. Walker RS, Custer PL, Nerad JA. Surgical excision of periorbital capillary hemangiomas. Ophthalmology. 1994; 101:1333–40.

19. Aldave AJ, Shields CL, Shields JA. Surgical excision of selected amblyogenic periorbital capillary hemangiomas. Ophthalmic Surg Lasers. 1999; 30:754–7.

20. Leaute-Labreze C, Dumas de la Roque E, Hubiche T, et al. Propranolol for severe hemangiomas of infancy. N Engl J Med. 2008; 358:2649–51.

21. Fay A, Nguyen J, Jakobiec FA, et al. Propranolol for isolated orbital infantile hemangioma. Arch Ophthalmol. 2010; 128:256–8.

22. Taban M, Goldberg RA. Propranolol for orbital hemangioma. Ophthalmology. 2010; 117:195.

23. Manunza F, Syed S, Laguda B, et al. Propranolol for complicated infantile haemangiomas: a case series of 30 infants. Br J Dermatol. 2010; 162:466–8.

24. Zimmermann AP, Wiegand S, Werner JA, et al. Propranolol therapy for infantile haemangiomas: review of the literature. Int J Pediatr Otorhinolaryngol. 2010; 74:338–42.

25. Pope E, Krafchik BR, Macarthur C, et al. Oral versus highdose pulse corticosteroids for problematic infantile hemangiomas: a randomized, controlled trial. Pediatrics. 2007; 119:e1239–47.

26. Bartoshesky LE, Bull M, Feingold M. Corticosteroid treatment of cutaneous hemangiomas: how effective? A report on 24 children. Clin Pediatr (Phila). 1978; 17(625):629–38.

27. Weiss AH. Adrenal suppression after corticosteroid injection of periocular hemangiomas. Am J Ophthalmol. 1989; 107:518–22.

28. Glatt HJ, Putterman AM, Van Aalst JJ, et al. Adrenal suppression and growth retardation after injection of periocular capillary hemangioma with corticosteroids. Ophthalmic Surg. 1991; 22:95–7.

29. Sommers Smith SK, Smith DM. Beta blockade induces apoptosis in cultured capillary endothelial cells. In Vitro Cell Dev Biol Anim. 2002; 38:298–304.

30. Gottschling S, Schneider G, Meyer S, et al. Two infants with life-threatening diffuse neonatal hemangiomatosis treated with cyclophosphamide. Pediatr Blood Cancer. 2006; 46:239–42.

31. Lawley LP, Siegfried E, Todd JL. Propranolol treatment for hemangioma of infancy: risks and recommendations. Pediatr Dermatol. 2009; 26:610–4.

32. Sans V, Dumas de la Roque E, Berge J, et al. Propranolol for severe infantile hemangiomas: follow-up report. Pediatrics. 2009; 124:e423–31.

33. Farhangi V, Sansone RA. QTc prolongation due to propranolol overdose. Int J Psychiatry Med. 2003; 33:201–2.

34. Yao X, McIntyre MS, Lang DG, et al. Propranolol inhibits the human ether-a-go-go-related gene potassium channels. Eur J Pharmacol. 2005; 519:208–11.