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Coumou A.D.,Academic Medical Center Amsterdam | Van Den Berg R.,Academic Medical Center Amsterdam | Bot J.C.,Academic Medical Center Amsterdam | Beetsma D.B.,Gelre Ziekenhuizen | Saeed P.,Orbital Center
Orbit | Year: 2014

A 37-year old male with a long history of a left orbital venous/lympathic malformation presented with ocular injection, increased proptosis and reduced left vision. Angiography demonstrated a carotid cavernous dural AV fistula combined with a concomitant venous/lymphatic malformation. After attempts at transvenous embolization, a direct uncomplicated transorbital puncture of the cavernous sinus via a lateral orbitotomy was performed with complete resolution of ocular symptoms. © 2014 Informa Healthcare USA, Inc. All rights reserved.


Slater O.,Great Ormond Street Hospital for Children NHS Foundation Trust | Balm A.J.M.,Netherlands Cancer Institute | Hartley B.,Great Ormond Street Hospital for Children NHS Foundation Trust | van den Brekel M.W.,Netherlands Cancer Institute | And 15 more authors.
European Journal of Cancer | Year: 2015

Background: Radiotherapy is a well-known cause of adverse events (AEs). To reduce AEs, an innovative local treatment was developed in Amsterdam: Ablative surgery, MOuld brachytherapy and surgical REconstruction (AMORE). Aims: (1) to determine the prevalence of AEs in HNRMS survivors and (2) to compare AEs between survivors treated with the international standard: external beam radiotherapy (EBRT-based: London) and survivors treated with AMORE if feasible, otherwise EBRT (AMORE-based: Amsterdam). Methods: All HNRMS survivors, treated in London or Amsterdam between January 1990 and December 2010 (n = 153), and alive ≥2. years post-treatment were eligible (n = 113). A predefined list of AEs was assessed in a multidisciplinary clinic and graded according to the Common Terminology Criteria for Adverse Events. Results: Eighty HNRMS survivors attended the clinic (median follow-up 10.5. years); 63% experienced ≥1 severe or disabling event, and 76% had ≥5 AEs (any grade). Survivors with EBRT-based treatment were, after adjustment for site, age at diagnosis, and follow-up duration, at increased risk to develop any grade 3/4 event or ≥5 AEs (any grade) compared with survivors with AMORE-based treatments (p = 0.032 and 0.01, respectively). Five year overall survival (source population) after EBRT-based treatment was 75.0%, after AMORE-based treatment 76.9%, p = 0.56. Conclusion: This study may serve as a baseline inventory and can be used in future studies for prospective assessments of AEs following the introduction of novel local treatment modalities. AMORE-based local treatment resulted in similar overall survival and a reduction of AEs secondary to local treatment. © 2015 Elsevier Ltd.


Purpose: To highlight the importance of Magnetic Resonance Imaging (MRI) and the use of propranolol as both a final diagnostic tool and adequate treatment for orbital Infantile Haemangiomas (IHs). Methods: A retrospective study was conducted using a case series of 5 infants diagnosed with orbital IH. All patients presented with progressive unilateral proptosis and were at high risk of developing amblyopia, some had combined swelling of the eyelid, impaired eye movements or exposure keratopathy. Propranolol was administered in an initial dose of 0.6mg/kg/day orally divided in three daily doses and increased in 4 days to 2.7mg/kg/day. MRI was performed in all children. Results: Striking MR characteristics of an IH lesion were seen in each of our 5 cases, including the presence of flow voids, high contrast enhancement, hypo-intense T1W signal, iso-to hyper intense T2W signal, and lobulated appearance. All patients showed a quick clinical response to treatment, resulting in significant reduction in tumour size within a range of 1-3 weeks and almost complete regression of the lesion at the end of the treatment schedule. Conclusions: Our study adds another 5 cases to the growing body of reports confirming the efficacy and safety-under controlled circumstances-of propranolol therapy in orbital IH management, in which we highlight the use of propranolol as both a final diagnostic tool and as an adequate treatment. © 2014 Informa Healthcare USA, Inc.


Clement S.C.,University Utrecht | Slater O.,Great Ormond Street Hospital | Chisholm J.C.,Royal Marsden Hospital | Abela C.,Craniofacial Unit | And 13 more authors.
European Journal of Cancer | Year: 2016

Purpose Head and neck rhabdomyosarcoma (HNRMS) survivors are at increased risk of developing pituitary dysfunction as an adverse event of radiotherapy. Our aim was to investigate the frequency and risk factors for pituitary dysfunction in these survivors. Secondly, we aimed to compare the prevalence of pituitary dysfunction between survivors treated with external beam radiation therapy (EBRT) and survivors treated with the ablative surgery, moulage technique after loading brachytherapy, and surgical reconstruction (AMORE) procedure. Methods Eighty HNRMS survivors treated in London (EBRT based) and Amsterdam (AMORE based: AMORE if feasible, otherwise EBRT) in the period 1990-2010 and alive ≥2 years post-treatment were evaluated. Survivors were evaluated in multidisciplinary late-effects clinics, with measurement of linear growth, determination of thyroid function, and growth hormone parameters. Additional data, such as baseline characteristics, anthropometrics, pubertal stage, and the results of additional laboratory investigations, were retrieved from patient charts. Results Pituitary dysfunction was diagnosed in 24 in 80 (30%) survivors, after a median follow-up time of 11 years. Median time to develop pituitary dysfunction after HNRMS diagnosis was 3.0 years. Risk factors were EBRT-based therapy (odds ratio [OR] 2.06; 95% confidence interval [CI] 1.79-2.46), parameningeal tumour site (OR 1.83; 95% CI 1.60-2.17) and embryonal RMS histology (OR 1.49; 95% CI 1.19-1.90). Conclusions Radiotherapy used for the treatment of HNRMS confers a significant risk of the development of pituitary dysfunction. AMORE-based treatment in children with HNRMS resulted in less pituitary dysfunction than treatment with conventional EBRT. Our findings underscore the importance of routine early endocrine follow-up in this specific population. © 2015 Elsevier Ltd. All rights reserved.


Slater O.,Great Ormond Street Hospital for Children NHS Foundation Trust | Balm A.J.M.,Netherlands Cancer Institute | Hartley B.,Great Ormond Street Hospital for Children NHS Foundation Trust | Van Den Brekel M.W.,Netherlands Cancer Institute | And 15 more authors.
European Journal of Cancer | Year: 2015

Background: Radiotherapy is a well-known cause of adverse events (AEs). To reduce AEs, an innovative local treatment was developed in Amsterdam: Ablative surgery, MOuld brachytherapy and surgical REconstruction (AMORE). Aims: (1) to determine the prevalence of AEs in HNRMS survivors and (2) to compare AEs between survivors treated with the international standard: external beam radiotherapy (EBRT-based: London) and survivors treated with AMORE if feasible, otherwise EBRT (AMORE-based: Amsterdam). Methods: All HNRMS survivors, treated in London or Amsterdam between January 1990 and December 2010 (n = 153), and alive ≥2 years post-treatment were eligible (n = 113). A predefined list of AEs was assessed in a multidisciplinary clinic and graded according to the Common Terminology Criteria for Adverse Events. Results: Eighty HNRMS survivors attended the clinic (median follow-up 10.5 years); 63% experienced ≥1 severe or disabling event, and 76% had ≥5 AEs (any grade). Survivors with EBRT-based treatment were, after adjustment for site, age at diagnosis, and follow-up duration, at increased risk to develop any grade 3/4 event or P5 AEs (any grade) compared with survivors with AMORE-based treatments (p = 0.032 and 0.01, respectively). Five year overall survival (source population) after EBRT-based treatment was 75.0%, after AMORE-based treatment 76.9%, p = 0.56. Conclusion: This study may serve as a baseline inventory and can be used in future studies for prospective assessments of AEs following the introduction of novel local treatment modalities. AMORE-based local treatment resulted in similar overall survival and a reduction of AEs secondary to local treatment. © 2015 Elsevier Ltd. All rights reserved.

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