Minimally disseminated disease and outcome in overt orbital retinoblastoma

Aschero, Rosario; Torbidoni, V.; Sampor, Claudia; Laurent, Viviana Eunice; Zugbi, Santiago; Winter, Ursula; Lubieniecki, Fabiana; Alonso, Daniel F.; Schaiquevich, Paula; Chantada, Guillermo

doi:10.1002/pbc.27662

Cited by 4 publications

(3 citation statements)

References 15 publications

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“… 5 , 6 , 12 In low- and middle-income countries, mortality rates of these patients may be as high as 50 to 90% although some promising results were published for patients with extraocular disease limited to the orbit and/or preauricular nodes. 1 , 2 , 5 , 7 , 8 , 26 – 28 On the other hand, metastatic patients with orbital disease usually present with a poor clinical status and rapid deterioration making it essential to provide treatments that exert a fast response and prevent local relapse. Thus intensive multimodal treatments are used showing important benefits compared to historical cohorts in terms of overall survival.…”

Section: Discussionmentioning

confidence: 99%

Pharmacokinetics of Orbital Topotecan After Ophthalmic Artery Chemosurgery and Intravenous Infusion in the Swine Model

Requejo,

Opezzo,

Vater

et al. 2023

Invest. Ophthalmol. Vis. Sci.

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Purpose Surgery, multiagent systemic chemotherapy, and radiation are used for patients with orbital retinoblastoma but are associated with unacceptable short- and long-term toxicity (including death). We studied orbital and systemic exposure of topotecan in the swine model after ophthalmic artery chemosurgery (OAC) and intravenous (IV) delivery. Methods Landrace pigs (n = 3) underwent 30-minute OAC of topotecan (4 mg), and samples were serially obtained from the femoral artery and from a microdialysis probe inserted into the lateral rectus muscle sheath of the infused eye as a surrogate of the orbital irrigation. Animals were recovered, and, after a wash-out period, plasma and microdialysate samples from the contralateral eye were collected after a 30-minute IV infusion of topotecan (4 mg). Samples were quantified by high-performance liquid chromatography, and population pharmacokinetic analysis was conducted using MonolixSuite. Results After OAC, median topotecan exposure in the orbit was 5624 ng × h/mL (range 3922–12531) compared to 23 ng × h/mL (range 18–75) after IV infusion. Thus, topotecan exposure in the orbit was 218-fold (range 75–540) higher after OAC than after IV infusion despite comparable systemic exposure (AUCpl) between routes (AUC pl, OAC : 141 ng × h/mL [127–191] versus AUC pl, IV : 139 ng × h/mL [126–186]). OAC was more selective to target the orbit because the median (range) orbital-to-plasma exposure ratio was 44 (28–65) after OAC compared to 0.18 (0.13–0.40) after IV infusion. Conclusions OAC of topotecan resulted in higher orbital exposure than after IV infusion and was a more selective route for local drug delivery. Patients with orbital retinoblastoma may benefit from a multimodal treatment strategy including OAC therapy.

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Section: Discussionmentioning

confidence: 99%

Pharmacokinetics of Orbital Topotecan After Ophthalmic Artery Chemosurgery and Intravenous Infusion in the Swine Model

Requejo,

Opezzo,

Vater

et al. 2023

Invest. Ophthalmol. Vis. Sci.

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“…Some potential molecular biomarkers for disseminated retinoblastoma were described, like the detection of cone‐rod homeobox (CRX) mRNA or GD2 protein expression and the detection of somatic pathogenic RB1 variant in blood, bone marrow aspirate or cerebral spinal fluid 37–39 . Some of these biomarkers correlate with metastatic relapse in high‐risk patients, but have not been evaluated in a prospective study or in low‐risk patients 40,41 …”

Section: Discussionmentioning

confidence: 99%

Adjuvant therapy of histopathological risk factors of retinoblastoma in Europe: A survey by the European Retinoblastoma Group (EURbG)

Dittner-Moormann

Reschke

Abbink

et al. 2021

Pediatric Blood & Cancer

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Introduction Advanced intraocular retinoblastoma can be cured by enucleation, but spread of retinoblastoma cells beyond the natural limits of the eye is related to a high mortality. Adjuvant therapy after enucleation has been shown to prevent metastasis in children with risk factors for extraocular retinoblastoma. However, histological criteria and adjuvant treatment regimens vary and there is no unifying consensus on the optimal choice of treatment. Method Data on guidelines for adjuvant treatment in European retinoblastoma referral centres were collected in an online survey among all members of the European Retinoblastoma Group (EURbG) network. Extended information was gathered via personal email communication. Results Data were collected from 26 centres in 17 countries. Guidelines for adjuvant treatment were in place at 92.3% of retinoblastoma centres. There was a consensus on indication for and intensity of adjuvant treatment among more than 80% of all centres. The majority of centres use no adjuvant treatment for isolated focal choroidal invasion or prelaminar optic nerve invasion. Patients with massive choroidal invasion or postlaminar optic nerve invasion receive adjuvant chemotherapy, while microscopic invasion of the resection margin of the optic nerve or extension through the sclera are treated with combined chemo‐ and radiotherapy. Conclusion Indications and adjuvant treatment regimens in European retinoblastoma referral centres are similar but not uniform. Further biomarkers in addition to histopathological risk factors could improve treatment stratification. The high consensus in European centres is an excellent foundation for a common European study with prospective validation of new biomarkers.

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“…In an age when retinoblastoma patients are increasingly treated conservatively, clinical decision-making relies more and more on non-invasive biomarkers, such as reverse transcriptase-polymerase chain reaction for GD2 synthase mRNA or cell-free tumor DNA from the CSF to detect minimal residual disease (13,(18)(19)(20)(21). Detection of metastatic risk factors is often based on radiological imaging, but risk stratification leaves room for improvement (22)(23)(24).…”

Section: Discussionmentioning

confidence: 99%

Full-width postlaminar optic nerve tumor invasion of retinoblastoma as risk-factor for leptomeningeal spread of retinoblastoma. A case report and review of the literature

Jong

Valk

Jansen

et al. 2020

Ophthalmic Genetics

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Minimally disseminated disease and outcome in overt orbital retinoblastoma

Cited by 4 publications

References 15 publications

Pharmacokinetics of Orbital Topotecan After Ophthalmic Artery Chemosurgery and Intravenous Infusion in the Swine Model

Pharmacokinetics of Orbital Topotecan After Ophthalmic Artery Chemosurgery and Intravenous Infusion in the Swine Model

Adjuvant therapy of histopathological risk factors of retinoblastoma in Europe: A survey by the European Retinoblastoma Group (EURbG)

Full-width postlaminar optic nerve tumor invasion of retinoblastoma as risk-factor for leptomeningeal spread of retinoblastoma. A case report and review of the literature

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