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Sclerotherapy Treatment of Orbital Lymphatic Malformations: a Large Single-Centre Experience Alex M Barnacle,1 Maria Theodorou,2 Sarah J Maling,2 Yassir Abou-Rayyah2

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Sclerotherapy Treatment of Orbital Lymphatic Malformations: a Large Single-Centre Experience Alex M Barnacle,1 Maria Theodorou,2 Sarah J Maling,2 Yassir Abou-Rayyah2 Downloaded from http://bjo.bmj.com/ on February 18, 2016 - Published by group.bmj.com Clinical science Sclerotherapy treatment of orbital lymphatic malformations: a large single-centre experience Alex M Barnacle,1 Maria Theodorou,2 Sarah J Maling,2 Yassir Abou-Rayyah2 – 1Department of Radiology, ABSTRACT sclerosing agents and techniques.8 14 Results are Great Ormond Street Hospital Background Percutaneous sclerotherapy is an promising in terms of reduction in lesion size and for Children, London, UK 2 alternative to surgery for the treatment of orbital improvement in proptosis. Detailed VA outcomes Department of 14 Ophthalmology, Great Ormond lymphatic malformations (LMs). We present a large series have only been documented in one series. We Street Hospital for Children, of patients undergoing sclerotherapy for macrocystic LMs report the largest single-centre experience of sclero- London, UK with detailed visual acuity (VA) outcome data. therapy in patients with orbital LMs including Methods Data were collected prospectively in all detailed VA outcome data. Correspondence to Dr Alex M Barnacle, patients with macrocystic orbital LMs undergoing Department of Radiology, sclerotherapy. Sclerotherapy was performed under MATERIALS AND METHODS Great Ormond Street Hospital general anaesthesia, instilling sodium tetradecyl sulfate A retrospective analysis was undertaken of all for Children, London under imaging control. Procedures were repeated at WC1N 3JH, UK; patients at our institution undergoing sclerotherapy [email protected] 2-week to 6-week intervals, depending on clinical for macrocystic orbital LMs. IRB/Ethics Committee response. Patients underwent ophthalmological ruled that approval was not required for this study. Received 19 January 2015 assessment, ultrasound and/or MRI before and after All patients had a history of proptosis. A unilateral Revised 29 May 2015 treatment. Primary outcome measure: change in maximal orbital LM was confirmed with ultrasound (US) Accepted 30 May 2015 radiological diameter of the LM. Secondary outcome Published Online First and MRI. Only those patients with macrocystic 17 June 2015 measure: change in VA after treatment. LMs were included in this study. Patients with Results 29 patients underwent 71 procedures (1–8 microcystic orbital LMs were excluded from the procedures per patient) over 6.7 years. Mean study as alternative primary sclerosing regimes were age=7.31 years. 11 patients (37.9%) had undergone considered for these lesions. previous treatment, including excision biopsy, drainage The primary outcome of this study was defined and decompression. All patients presented with proptosis as radiological change in maximal diameter of the and/or pseudoptosis. 23 patients (79.3%) had decreased LM, assessed with US±MRI before and after treat- VA at presentation. Average follow-up was 21.8 months ment. A preoperative MRI was performed in all (range 3–75 months). All patients achieved a reduction cases; post-treatment MRIs were performed only in maximal lesion diameter of ≥50%, with complete when clinically indicated. All US were performed radiological resolution in 51.7% (n=15). VA improved in by a single radiologist (AMB). The imaging proto- 18/23 patients (78.2%). Average logMAR before col involved US performed before treatment, treatment=0.43 (SD ±0.47); average after between treatments, 6 weeks after final treatment treatment=0.25 (SD ±0.32); p<0.01. There was one and then at six monthly intervals for 2 years. complication (1.4%): one patient required a lateral Radiological response to treatment was defined as a canthotomy for an intralesional haematoma 2 h after change in maximal lesion diameter: resolved=no sclerotherapy. evidence of residual malformation in the orbit; Conclusions Sclerotherapy is a safe and highly good response=>75% reduction in maximal lesion effective treatment for orbital LMs with excellent VA diameter; partial response=50–75% reduction in outcomes. It should be considered as the first-line maximal lesion diameter; poor response=<50% treatment for this condition. reduction in maximal lesion diameter; and no response=no improvement in maximal lesion diameter. INTRODUCTION The secondary outcome was defined as change in Lymphatic malformations (LMs) or lymphangiomas VA following treatment. All patients underwent of the orbit are benign vascular malformations, preoperative ophthalmological assessment and were often diagnosed in childhood. Complications reassessed 1 day after treatment and again at include intralesional haemorrhage, cellulitis, ambly- 6 weeks. Visual assessment was conducted by the opia and impaired visual acuity (VA). Historically, orthoptic team; all patients had age-appropriate the management of orbital LMs has involved visual testing (Cardiff cards, Kay pictures or complex, aggressive surgical intervention with vari- crowded logMAR). A paired Student’s t test was able results and high recurrence rates.12 used to compare VA before and after sclerotherapy Percutaneous intralesional sclerotherapy is now treatment in patients in whom VAwas measurable. well established as a first-line treatment for LMs elsewhere in the body using a variety of sclerosing Sclerotherapy technique agents including sodium tetradecyl sulfate (STS), All sclerotherapy procedures were carried out ethanol, OK-432 (Picibanil), bleomycin and doxy- under general anaesthesia by an interventional radi- To cite: Barnacle AM, – Theodorou M, Maling SJ, cycline.3 7 Small series of up to 13 patients have ologist (AMB) in a biplane angiography suite. All et al. Br J Ophthalmol reported the use of percutaneous sclerotherapy in patients received a single dose of intravenous dexa- – 2016;100:204 208. the management of orbital LMs with a variety of methasone (0.25 mg/kg) on induction. Antibiotics 204 Barnacle AM, et al. Br J Ophthalmol 2016;100:204–208. doi:10.1136/bjophthalmol-2015-306657 Downloaded from http://bjo.bmj.com/ on February 18, 2016 - Published by group.bmj.com Clinical science were only given if active infection was suspected (n=2). The with <75% reduction in lesion size on US and in conjunction affected orbit was scanned using a high-resolution linear 5– with VA findings. STS was used as the sclerosant in all cases, at a 13 MHz US probe. The malformation was accessed percutan- strength of 1.5–3%. The average volume of sclerosant used was eously with a 22 gauge two-part needle through an upper lid 1.12 mL (range 0.22–3.8 mL). medial or lateral approach, depending on the position of the The average length of clinical follow-up was 21.8 months malformation. Needle placement was controlled with real-time (range 3–75 months, median 22 months). The procedural com- US throughout the procedure. Where possible, cyst contents plication rate was 1.4% (1/71 procedures). The first patient in were aspirated. Contrast medium (Omnipaque 240, GE the series (age 1.6 years) experienced an acute retrobulbar Healthcare, Oslo) was then instilled in small volumes (<1 mL) haemorrhage 2 h after the second sclerotherapy treatment for into the cyst during digital subtraction angiography (DSA) using an extensive LM. 3.8 mL of STS 3% was instilled during the biplane imaging. This allowed the operator to exclude commu- procedure, and a standard procedural technique was used. The nication between the cyst and the cavernous sinus, ophthalmic haemorrhage was managed with acetazolamide and an uncom- artery or other structures. Sclerosant was only injected if no plicated lateral canthotomy. She underwent two subsequent communication was seen. STS sclerosant was then instilled in uncomplicated sclerotherapy procedures, with complete reso- small aliquots into the largest cyst(s) under US control. The lution of the malformation (follow-up 75 months) and some sclerosant was left to dwell in the cyst and not aspirated. improvement in VA (no interest in Cardiff cards pre-treatment; Postoperative orbital bandaging was not used. Patients returned logMAR 0.87 post-treatment). to an inpatient ward with oral morphine prescribed on an ‘as The primary outcome measure was radiological change in size required’ basis and were closely monitored for acute proptosis of the lesion US±MRI after treatment. Radiological improve- caused by an intralesional bleed or severe swelling. All patients ment was assessed in all patients on US±MRI. All patients underwent ophthalmology assessment the following day prior achieved a reduction in lesion size of ≥50%. Fifteen patients to discharge. Routine follow-up took place at 6 weeks unless the (51.7%) demonstrated complete radiological resolution of their need for a repeat sclerotherapy procedure was predicted due to malformation, and 11 patients (37.9%) showed a good response. a large volume of cysts, in which case sclerotherapy was In many patients in whom the malformation was no longer repeated at 2–6 weeks. detected, there was residual heterogeneity of the fat in the poster- ior orbit, taken to reflect the presence of previous inflammation/ disease in the orbit. Examples of radiological appearances before RESULTS and after treatment are shown in figures 2–4. Twenty-nine patients were referred for treatment between The secondary outcome measure was change in VA after treat- October 2007 and September 2014. All had macrocystic orbital ment. Twenty-three patients (79.3%) had decreased VA in the fi LMs, with associated microcystic disease of the face in ve. affected eye at presentation. Of those 23 patients, VA improved None of the patients had microcystic disease affecting the orbit in 18 patients (78.2%), remained stable in 3 (13%), remained fi itself, and none of the lesions had a signi cant venous compo- unrecordable in 1 and worsened in 1 (logMAR 0.52 pre- nent. Mean age at the time of treatment was 7.31 years (range treatment, 0.70 post-treatment). Statistical analysis of change in – 0.3 21.6 years, median 5.8 years). The commonest mode of VA could not include two patients that had no interest in fi presentation was pain and/or proptosis ( gure 1). Twenty-three Cardiff cards at presentation or the patient who was NPL at patients (79.3%) had decreased VA in the affected eye at presen- presentation and in whom understandably this did not improve.
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