Topical rapamycin combined with pulsed dye laser in the treatment of capillary vascular malformations in Sturge-Weber syndrome: Phase II, randomized, double-blind, intraindividual placebo-controlled clinical trial

Marques, L.E. A.; Núñez‐Córdoba, Jorge M.; Aguado, Leyre; Pretel, Maider; Boixeda, P.; Nagore, Eduardo; Baselga, Eulàlia; Redondo, Pedro

doi:10.1016/j.jaad.2014.10.011

Cited by 93 publications

(84 citation statements)

References 31 publications

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“…In contrast, considerable revascularization of the PDL-only sites was observed. Furthermore, in a clinical study involving 23 subjects with Sturge-Weber syndrome and PWS, Marqués et al [120] reported that the combination of PDL and topical rapamycin led to a higher degree of blanching than PDL alone.…”

Section: Development Of New Treatment Approachesmentioning

confidence: 99%

The Role of Laser Speckle Imaging in Port-Wine Stain Research: Recent Advances and Opportunities

Choi

Tan

Jia

et al. 2016

IEEE J. Select. Topics Quantum Electron.

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Here, we review our current knowledge on the etiology and treatment of port-wine stain (PWS) birthmarks. Current treatment options have significant limitations in terms of efficacy. With the combination of 1) a suitable preclinical microvascular model, 2) laser speckle imaging (LSI) to evaluate blood-flow dynamics, and 3) a longitudinal experimental design, rapid preclinical assessment of new phototherapies can be translated from the lab to the clinic. The combination of photodynamic therapy (PDT) and pulsed-dye laser (PDL) irradiation achieves a synergistic effect that reduces the required radiant exposures of the individual phototherapies to achieve persistent vascular shutdown. PDL combined with anti-angiogenic agents is a promising strategy to achieve persistent vascular shutdown by preventing reformation and reperfusion of photocoagulated blood vessels. Integration of LSI into the clinical workflow may lead to surgical image guidance that maximizes acute photocoagulation, is expected to improve PWS therapeutic outcome. Continued integration of noninvasive optical imaging technologies and biochemical analysis collectively are expected to lead to more robust treatment strategies.

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Section: Development Of New Treatment Approachesmentioning

confidence: 99%

The Role of Laser Speckle Imaging in Port-Wine Stain Research: Recent Advances and Opportunities

Choi

Tan

Jia

et al. 2016

IEEE J. Select. Topics Quantum Electron.

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“…However, the extent to which these lesions may improve varies widely and complete resolution is infrequent. 12 Woo et al reported only 2% of patients with complete disappearance of lesion after a mean of 17 treatment sessions. 13 While PDL is very effective in producing initial lightening of PWS lesions, there is a subsequent revascularization after laser therapy that results in frequent treatment failure.…”

Section: Discussionmentioning

confidence: 99%

“…15,16 Resolution of PWS, as defined by persistent blanching of the lesion, is reported in less than 10%-20% of cases. 12,[17][18][19][20] Early intervention is indicated to prevent the development of hypertrophy and nodularity, which has been shown to increase treatment resistance. 12,21 Thickened PWS lesions at older age have been associated with increased therapeutic failure.…”

Section: Discussionmentioning

confidence: 99%

“…12,[17][18][19][20] Early intervention is indicated to prevent the development of hypertrophy and nodularity, which has been shown to increase treatment resistance. 12,21 Thickened PWS lesions at older age have been associated with increased therapeutic failure. Sclerotherapy, selective endovenous thermal ablation, surgical stripping, phlebectomy, and occasionally, deep vein reconstructions are other more invasive therapeutic A B C options for KTS which are recommended to be saved for refractory lesions.…”

Section: Discussionmentioning

confidence: 99%

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Successful Treatment of Unilateral Klippel-Trenaunay Syndrome With Pulsed-Dye Laser in a 2-Week Old Infant

2017

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Introduction: Klippel-Trenaunay syndrome (KTS) is a rare congenital mesodermal abnormality characterized by varicose veins, cutaneous capillary malformation, as well as bone and soft tissue hypertrophy. Case Report: A 2-week-old female infant presented to our clinic because of vascular nevus and progressive enlargement of her right extremities and trunk since birth. The patient was treated with 595-nm pulsed-dye laser (PDL). Her port-wine stain (PWS) disappeared completely after third PDL session and the soft tissue hypertrophy stopped. The patient experienced neither recurrence nor any change in size after 7 years of follow up. Conclusion: PDL can treat KTS completely with no reccurence if it is used in "early stage" of disease.

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“…Although most currently used drugs do not directly affect the GLU receptors, new-generation AMPA (α-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid) receptor antagonists, such as perampanel, can inhibit GLU-mediated excitation, and may be considered for SWS epilepsy treatment in the future as clinical experience with such drugs becomes available in pediatric populations (Biro et al, 2015; Rosenfeld et al, 2015). Another therapeutic option would involve inhibition of mammalian target of rapamycin (mTOR) complex 1 signaling that may not only reverse increases of glutamatergic neurotransmission (Talos et al, 2012) but could also be beneficial to treat the vascular malformation (Marques et al, 2015; Shirazi et al, 2007). …”

Section: Discussionmentioning

confidence: 99%

Imaging increased glutamate in children with Sturge–Weber syndrome: Association with epilepsy severity

Juhász

Xuan

et al. 2016

Epilepsy Research

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Background Sturge-Weber syndrome (SWS) is strongly associated with epilepsy. Brain tissue studies have suggested that epileptic activity in SWS is driven by glutamatergic synaptic activity. Here, we used proton magnetic resonance spectroscopic imaging (MRSI) to test if glutamate (GLU) concentrations are increased in the affected hemisphere and if such increases are associated with severity of epilepsy in children with SWS. We also studied the metabolic correlates of MRSI abnormalities, using glucose positron emission tomography (PET) imaging. Methods 3T MRI and glucose PET were performed in 10 children (age: 7–78 months) with unilateral SWS and a history of epilepsy. MRSI data were acquired from the affected (ipsilateral) and non-affected (contralateral) hemispheres. GLU, N-acetyl-aspartate (NAA) and creatine (Cr) were quantified in multiple voxels; GLU/Cr and NAA/Cr ratios were calculated and compared to seizure frequency as well as glucose PET findings. Results The highest GLU/Cr ratios were found in the affected hemisphere in all children except one with severe atrophy. The maximum ipsilateral/contralateral GLU/Cr ratios ranged between 1.0–2.5 (mean: 1.6). Mean ipsilateral/contralateral GLU/Cr ratios were highest in the youngest children and showed a strong positive correlation with clinical seizure frequency scores assessed at the time of the scan (r=0.88, p=0.001) and also at follow-up (up to 1 year, r=0.80, p=0.009). GLU increases in the affected hemisphere coincided with areas showing current or previous increases of glucose metabolism on PET in 5 children. NAA/Cr ratios showed no association with clinical seizure frequency. Conclusions Increased glutamate concentrations in the affected hemisphere, measured by MRSI, are common in young children with unilateral SWS and are associated with frequent seizures. The findings lend support to the role of excess glutamate in SWS-associated epilepsy.

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Topical rapamycin combined with pulsed dye laser in the treatment of capillary vascular malformations in Sturge-Weber syndrome: Phase II, randomized, double-blind, intraindividual placebo-controlled clinical trial

Cited by 93 publications

References 31 publications

The Role of Laser Speckle Imaging in Port-Wine Stain Research: Recent Advances and Opportunities

The Role of Laser Speckle Imaging in Port-Wine Stain Research: Recent Advances and Opportunities

Successful Treatment of Unilateral Klippel-Trenaunay Syndrome With Pulsed-Dye Laser in a 2-Week Old Infant

Imaging increased glutamate in children with Sturge–Weber syndrome: Association with epilepsy severity

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