Photodiagnosis and Photodynamic Therapy (2010) 7, 201—203

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LETTER TO THE EDITOR

The effective management of a leg hemangiomphotodynamic therapy

Introduction

Vascular lesions are thought to represent some of the mostcommon tumours of infancy and have been traditionallyclassified by Virchow into two categories: hemangiomasand lymphangiomas. However, in 1996, the InternationalSociety for the Study of Vascular Anomalies adopted anew classification based on that described by Mulliken andGlowacki [1]. Vascular lesions now include hemangiomas,with endothelial proliferation and vascular malformations,which have normal endothelial turnover. Hemangiomas aremore common in females and 80% occur in the head andneck. They typically have a rapid proliferative period, fol-lowed by a slow regressive phase. This is in contrast tovascular malformations which continue to increase in size,due to the progressive ectasia of the involved vessels,which may be affected by infection, trauma or hormonalchanges. Treatment is indicated when the lesions becomesymptomatic by causing functional disturbances or forcosmesis [1,2].

There are a range of therapies used in the management ofvascular lesions depending upon the character of the lesion.Superficial capillary malformations may be treated withlaser coagulation. Sclerotherapy, with ethanol or sodiumtertradecyl sulphate may be used to treat venous malfor-mations. High-flow malformations may be embolized andthen surgically resected. Lymphatic malformations oftenneed surgical de-bulking or removal. Radiotherapy andchemotherapy have also been described in the treatmentof hemangiomas [2].

Photodynamic therapy (PDT) is a developing technology,used in the treatment of advanced tumours and vascularmalformations. The main application for PDT is to targettumours of the head and neck, gastrointestinal tract, pul-monary malignancies and skin pathologies. The effects ofPDT are a result of a cold photochemical process, via theinteraction between a photosensitiser, oxygen and light. PDT

may be provided under local, regional or general anaesthesiaand it may be delivered by surface illumination or interstitialapplication, via inserted optical fibers. The photosensitiser,administered whether locally or systemically, is selectivelytaken up and retained by tissues with a high vascular flow

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1572-1000/$ — see front matter © 2010 Elsevier B.V. All rights reserved.doi:10.1016/j.pdpdt.2010.04.001

ing ultrasound-guided interstitial

ate. Activation by light of a specific wavelength results inhe production of oxygen free radicals or intracellular oxy-en. This leads to apoptosis or direct cell death, as a resultf intracellular oxygenation and vascular shutdown [3—5]Fig. 1).

We present a case report that describes the success-ul application of ultrasound-guided interstitial PDT for aatient with an extensive hemangioma of the leg.

ase report

26-year-old Caucasian male was referred through hisrthopaedic surgeon to the UCLH Head and Neck Center,ondon with a 15-year history of severe pain in his rightower leg. The pain was exacerbated by movement, pre-ented the patient from playing sports as a teenager andrequently disturbed his sleep. It was managed with anal-esics and anti-inflammatory medications.

At the age of 24, because the pain continued to be poorlyontrolled, a surgical biopsy was performed to excludealignant change. The histopathological review suggestedhemangioma with fatty metaplasia and no malignant fea-

ures. A magnetic resonance scan revealed that the lesionxtended from the posterior compartment, to the lateralnd anterior compartments of his right calf. The lesionas not amenable to conventional treatment. Due to its

arge size, surgical resection carried a significant risk to theessels and neurological structures close to the lesion. Anngiogram illustrated the presence of large number of feed-ng vessels, deeming the lesion unsuitable for embolization.

On examination the patient walked with a slight short legait. The right limb was 1—2 cm shorter than the left limb.e had a notable swelling over the anterior lateral aspect ofhe proximal half of his right tibia and a swelling in the pos-erior aspect of his calf, which was tender to touch. He wasble to hold his leg straight and neurovascular examinationf the leg was unremarkable. The right leg was moderatelyasted in comparison to the left. He had had no devel-

pmental problems, no significant family history and wastherwise fit and well.

Following discussion at a multi-disciplinary team meet-ng, it was decided that the most suitable option was to

202 Letter to the Editor

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Figure 1 Principal

reat the lesion with ultrasound-guided interstitial photo-ynamic therapy (US-iPDT). Meta-tetra-hydroxyphenyl chlo-ine (mTHPC) was administered at a dose of (0.15 mg/kg)ntravenously into the mid-cubital vein 96 hours prior toreatment.

Photodynamic therapy was performed under loco-egional anaesthesia, via a popliteal block and localnfiltration with 0.05% Bupivacaine. Two 18 Gauge 70 mmong spinal needles were inserted under US guidance into thepper anterior and posterior portion of the vascular malfor-

ation. The area of treatment was scanned by ultrasound

EMP 1100 with high resolution) prior to needle insertion, tonsure accurate identification of the target volume. Greatare was taken to ensure that the needles were insertedarallel to each other, with 1 cm distance in between.

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igure 2 MRI Image (left) showing extensive hemangioma; while MRhis was associated with peri-hemangioma inflammation and hence

otodynamic therapy.

‘Laser light delivery fibers’ with ‘polished tips’ and aore diameter of 400 �m were introduced into the 2 spinaleedles. A four-channel 652 nm diode laser was used for illu-ination. Diode light was used for illumination and 20 J/cm2

as delivered per station to the target tissue.Postoperatively, gradual re-exposure to light was carried

ut at an incremental rate of 100 lx per day. The patientas advised about the need to avoid direct exposure to sun

ight for up to 2 weeks after injection of the photosensitisernd he was given light exposure guidelines. The patient was

iven intravenous metronidazole and dexamethasone for 3ays and analgesia as required. The patient was dischargedrom hospital care 2 days postoperatively.

A 6-week post-PDT MRI with contrast showed a significanteduction in the size of the hemangioma (Fig. 2). The patient

I with contrast image (right) showing major resolution post-PDT,the haziness in the image.

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Letter to the Editor

had increased function of his right leg. He was able to walkfor long distances, run for shorter distances and play sportswithout pain in his leg. He continues to be asymptomatic1-year post-PDT.

The only adverse complication was that 4 weeks postop-eratively the patient sustained a skin burn of his left arm fol-lowing inadvertent sun exposure. This healed following thetopical application of 1% hydrocortisone cream for 5 days.

Discussion

Conventional management of hemangiomas may include oneor a combination of embolization, surgery ± reconstruction,sclerotherapy, alcohol ablation, chemotherapy and radio-therapy. Where lesions are large, surgery can have markedadverse effect on form and function and due to difficulty indelineating these lesions, recurrence can be high. In the casedescribed surgery would risk the loss of the limb functionand the lesion was not amenable to embolization. The sideeffects of chemotherapy are well known and radiotherapycarries the risk of inducing new tumours [2—5].

PDT for vascular lesions has been used primarily for themanagement of age-related macular degeneration. It hasbeen described as effective in the treatment of port-winestain birthmarks, alone and in combination with pulsed dyelaser therapy [4]. There are relatively few studies in theliterature describing the use of PDT for deep seated vascu-lar lesions, especially outside of the head and neck region.However, this case report shows that PDT may have a rolein the management of such pathologies.

The effectiveness of PDT is dependant on the dosimetricprofile. Ideally, one would quantify the distribution of thelight fluence rate, the optical properties, the drug concen-tration and tissue oxygenation for PDT. Zhu et al. helped todevelop a system for real time recording of these values,using dosimetry and motorised probes, to develop a feed-back system. They suggested this data should be recordedin future studies involving PDT [6].

The other advantage of PDT is that it can be repeatedwithout cumulative toxicity. The success of PDT is depen-dent on upon the depth of necrosis being greater that the

depth of the individual malformation [5]. Thus, interstitialPDT represents is ideal, in that it allows more effectivetreatment of the target tissue volume.

This case report also highlights the advantage of ultra-sound, in combination with PDT. The use of ultrasound also

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eans that it is easier to ensure that the needles arenserted parallel to each other and are be directed awayrom vital structures, such as nerves and blood vessels.xpertise is required in the guidance of needle inser-ion with ultrasound, to aid the assessment of the exacthree-dimensional location of the needles, seen on a twoimensional image [5].

eferences

1] Mulliken JB, Glowacki J. Hemangiomas and vascular malforma-tions in infants and children: a classification based on endothe-lial characteristics. Plast Reconstr Surg 1982;69:412—22.

2] Betz C, Jager HR, Brookes JAS, Richards R, Leunig A, Hopper C.Interstitial photodynamic therapy for a symptom-targeted treat-ment of complex vascular malformations in the head and neckregion. Laser Surg Med 2007;39:571—82.

3] Beck DO, Gosain AK. The presentation and management ofhemangiomas. Plast Reconstr Surg 2009;123:181—91.

4] Yuan KH, Li Q, Yu WL, Zeng D, Zhang C, Zheng H. Comparisonof photodynamic therapy and pulsed dye laser in patients withport wine stain birthmarks: a retrospective analysis. PhotodiagPhotodyn Therapy 2008;5:50—7.

5] Jerjes W, Upile T, Hamdoon Z, et al. Ultrasound-guided photody-namic therapy for deep seated pathologies: prospective study.Laser Surg Med 2009;41(October):612—21.

6] Zhu TC, Finlay JC, Hahn SM. Determination of the distri-bution of light, optcal properties, durg concentration andtissue oxygentation in-vivo in human prostrate during motexafinlutetium-mediated photodynamic therapy. J Photochem Photo-biol B 2005;79:231—41.

Priya Shah BDS, MFDS ∗

UCLH Head and Neck Centre, 1st Floor East Wing,250 Euston Road, London NW1 2PG, United Kingdom

Waseem JerjesUCLH Head and Neck Centre, London, United Kingdom

Tahwinder UpileUniversity College London Medical School, London,

United Kingdom

Colin HopperUCLH Head and Neck Centre, London, United Kingdom

∗ Corresponding author.E-mail address: shah priya@hotmail.com (P. Shah)

Available online 28 April 2010