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Middle East African Journal of Ophthalmology Middle East African Journal of Ophthalmology
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Year : 2011  |  Volume : 18  |  Issue : 4  |  Page : 303-304  

Serendipity, the humble case report and modern health science challenges

Division of Ophthalmology, Faculty of Health Sciences, University of Stellenbosch, South Africa

Date of Web Publication23-Nov-2011

Correspondence Address:
David Meyer
Division of Ophthalmology, Faculty of Health Sciences, University of Stellenbosch
South Africa
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0974-9233.90132

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How to cite this article:
Meyer D. Serendipity, the humble case report and modern health science challenges. Middle East Afr J Ophthalmol 2011;18:303-4

How to cite this URL:
Meyer D. Serendipity, the humble case report and modern health science challenges. Middle East Afr J Ophthalmol [serial online] 2011 [cited 2022 Aug 16];18:303-4. Available from: http://www.meajo.org/text.asp?2011/18/4/303/90132

Any expecting parent would be devastated when their dear little infant is born with an unsightly periocular capillary hemangioma. These tumors often present at birth or soon thereafter may be small and of no clinical significance. However, when they are large and unsightly, both parents and physicians are motivated to intervene therapeutically. Given their predilection for the upper lid, the risk of the mass precluding normal visual development is high. Sensory deprivation, induced astigmatism and anisometropia contribute to the risk of developing amblyopia which is as high as 64%. [1] All clinicians who treat these tumors are keenly aware of the inadequacies of conventional treatment modalities and for many years a need has existed for simpler and more effective therapies which have, until recently, evaded us.

It was therefore with great excitement and expectation that the landmark paper by Lιautι-Labrθze et al, was received by clinicians when it first appeared in 2008 describing propranolol for the treatment of hemangiomas. [2] Propranolol was serendipitously found to induce early involution in capillary hemangiomas and this was reported in a prestigious journal in the format of a "humble" letter to the editor (a form of scientific communication belittled by many modern editors of international medical journals). The authors admitted that the regulators of hemangioma growth were still poorly understood and that exactly how propranolol, a non-selective b-adrenergic antagonist, induced such remarkable regression in capillary hemangiomas was uncertain but they nevertheless reported their convictions. They suggested vasoconstriction and induction of apoptosis in capillary endothelial cells as possible mechanisms. Since that communication several others, mostly in the form of editorials and letters, have appeared on the subject and mostly in nonophthalmic literature. [3],[4],[5]

In their report "Oral Propranolol for the Treatment of Periorbital Infantile Hemangioma: A Preliminary Report from Oman" the authors present four more cases of periorbital infantile hemangiomas successfully treated with systemic propranolol and then highlight some of the treatment challenges still awaiting clinicians. Reports such as these assist in accumulating evidence for the safety and efficacy of novel therapeutic modalities in relatively rare conditions such as periorbital infantile hemangiomas.

The issues, among others, which need further scientific reflection and deliberation include the exact posology, the efficacy of topical propranolol therapy vs. systemic treatment, [6] the effect of other nonselective b-blockers on these tumors and the optimal duration of treatment. Clearly this new application of an old drug should also stimulate us to think of and assess other molecules for use to the same end.

One such molecule is bleomycin. Bleomycin was first isolated in 1966 by Umezawa from a soil fungus, Streptomyces verticillus. The main mechanism of action of bleomycin is DNA cleavage via oxidative damage caused by free radicals which form when its metal binding core is oxidized.[7] Bleomycin also induces apoptosis in rapidly growing cells and has a sclerosing effect on vascular endothelium which makes it useful in the proliferating phase of vascular neoplasms. [8] Additional mechanisms include blocking the cell cycle at G2, degrading cellular RNA and the induction of tumor necrosis factor. Many investigators (mostly nonophthalmic) have reported the successful use of intralesional bleomycin injection (IBI) in treating hemangiomas [7],[8] and lymphangiomas in various anatomic locations. Basal cell carcinoma, Kaposi sarcoma, keratoacanthoma and skin metastases of malignant melanoma have also responded well to IBI. [7] One conservative estimate indicates that 56.2% of lesions treated with IBI will undergo 70-100% regression although other reports indicate a significantly higher success rate. [9]

We have used both intralesional bleomycin as well as systemic propranolol with extremely satisfactory results in extensive or refractive cases of periocular capillary hemangiomas as well as lymphangiomas. Convincing editors to publish such reports have, however, met with less satisfactory results.

May serendipity and the humble, but well-selected case report continue to make meaningful contributions to health science.

   References Top

1.Boyd MJ, Collin JRO. Capillary hemangiomas: An approach to their management. Br J Ophthalmol 1991;75:298-300.  Back to cited text no. 1
2.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. 2
3.Buckmiller LM. Propranolol treatment for infantile hemangiomas. Curr Opin Otolaryngol Head Neck Surg 2009;17:458-9.  Back to cited text no. 3
4.Holmes WJ, Mishra A, Gorst C, Se-Hwang L. Propranolol as first-line treatment for infantile hemangiomas. Plast Reconstr Surg 2010;1:420-1.  Back to cited text no. 4
5.Fay A, Nguyen J, Jakobiec FA, Meyer-Junghaenel L, Waner M. Propranolol for isolated orbital infantile hemangioma. Arch Ophthalmol 2010;128:256-8.  Back to cited text no. 5
6.Guo S, Ni N. Topical treatment for capillary hemangioma of the eyelid using b-blocker solution. Arch Ophthalmol 2010;128:255-6.  Back to cited text no. 6
7.Saitta P, Krishnamurthy K, Brown LH. Bleomycin in dermatology. Dermatol Surg 2008;34:1299-313.  Back to cited text no. 7
8.Pienaar C, Graham R, Geldenhuys S, Hudson DA. Intralesional bleomycin for the treatment of hemangiomas. Plast Reconstr Surg 2006;117:221-6.  Back to cited text no. 8
9.Mathur NN, Rana I, Bothra R, Dhawan R, Kathuria G, Pradhan T. Bleomycin sclerotherapy in congenital lymphatic and vascular malformations of head and neck. Int J Pediatr Otorhinolaryngol 2005;69:75-80.  Back to cited text no. 9


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