Good response to pulsed dye laser in patients with capillary malformation‐arteriovenous malformation syndrome (CM‐AVM) (original) (raw)

Histological study on the treatment of vascular malformations resistant to pulsed dye laser

LASER THERAPY, 2013

Background and Aims: The pulsed dye laser (PDL) is recognized both as an effective treatment for vascular malformations and the as first treatment of choice for these lesions. However, PDL irradiation has poor efficacy in some patients, particularly the elderly. The present study histologically assessed such patients to try to elucidate the reason. Materials and Methods: A pulsed dye laser was used in 3 subjects in whom previous laser treatment was ineffective. Three-millimeter punch biopsies were obtained before laser treatment, 1 week and 3 months after the laser treatment. Each specimen was stained with toluidine blue and examined under light microscopy followed by electron microscopy with oolong tea extract (OTE) staining. Results: Microscopy revealed an increase in the vasculature at baseline and an increased number of dermal fibroblasts. One week post-irradiation, inflammatory cell infiltration was observed together with extensive interstitial perivascular edema. At 1 week and 3 months after laser irradiation, normal structures were observed for both blood vessels and capillary endothelial cells. Mild changes were noted in other interstitial features, but findings obtained 3 months after irradiation were almost similar to those before irradiation. Conclusions: The lower efficacy of PDL treatment in the elderly was possibly due to the markedly low amount of red blood cells in our subjects' blood vessels, a major chromophore for the PDL, was markedly low. It is possible that age-related denaturation of dermal matrix collagen plays some role in maintaining the vasculature in the interstitium with edema, and inflammatory cell infiltration could lead to the cellular release of some cytokines which favor reconstruction of the vasculature.

Targeted Diode Laser Therapy for Oral and Perioral Capillary-Venous Malformation in Pediatric Patients: A Prospective Study

Children

Background: This study describes the management protocol for capillary-venous malformations in pediatric patients and reports the epidemiology of diagnosed and treated cases at the Unit of Odontostomatology of the Aldo Moro University of Bari from 2014 to 2022. Methods: The authors classified the intraoral and perioral capillary-venous malformations by superficial diameter (<1 cm, 1–3 cm, >3 cm) and ultrasonographical depth extension (≤5 mm, >5 mm). All patients underwent pulsed-mode diode laser transmucosal photocoagulation (8–12 W/cm2); those with malformations that were wide (>3 cm) and deep (>5 mm) received intralesional photocoagulation, too (13 W/cm2). The children received general anesthesia based on their compliance and lesions’ extension. The follow-up lasted six months. Results: A total of 22 females and 14 males (age range 4–18 years) presented 63 capillary-venous malformations. Five patients with Sturge–Weber syndrome, seven with hereditary hemorrhagic tel...

Laser Treatment of Venous Malformations

Annals of Plastic Surgery, 2006

Venous malformations (VM) are developmental errors comprised of enlarged dysplastic blood vessels. Clinically they manifest as either a faint blue patch or a soft blue vascular mass. Treatment options include a sclerosing agent (such as alcohol or ethiblock), surgery or laser therapy. A review of the literature concerning VM and laser treatment yielded a few sporadic reports describing series of 3 to 46 cases. In this retrospective study we reviewed the files of the relevant cases. The data included the extent of the lesion, the different anatomical sites, age of patient and the extent of clearance of the lesion as the treatment's end point. This study includes 56 cases which makes it the largest series presented. The success rate of laser treatment was 92.8%. The immediate complication rate was very low (approximately 3.57%) including minimal scarring and deformity. No long term complications were noted. Our study concludes that treatment of VM is a difficult task due to the nature of the lesions. Laser treatment of these lesions enables one to obtain good results with a very low incidence of complications. Surgery and other treatment modalities are not always satisfactory, yield similar or less efficient results and have a higher complication rate. Laser treatment can play an important role in the treatment of VM and in fact may be the treatment of choice in some settings.

Histopathological hallmarks of cutaneous lesions of capillary malformation–arteriovenous malformation syndrome

Journal of the European Academy of Dermatology and Venereology, 2020

IMPORTANCE Capillary malformation-arteriovenous malformation (CM-AVM) syndrome is a recently described syndrome with distinctive cutaneous lesions. Very little is known about the histopathology of these lesions. OBJECTIVE The purpose of the study was to evaluate the histopathological characteristics of the pink macules of the CM-AVM syndrome and to investigate if these pink macules could be classified as capillary malformations or arteriovenous malformations based on their histopathological features. DESIGN-SETTINGS-PARTICIPANTS We conducted a retrospective multicenter study involving 8 hospitals in Spain. Fifteen biopsies from pink macules of the CM-AVM syndrome were analyzed, and compared with 5 biopsies of diverse capillary malformations and 3 stage I arteriovenous malformations. RESULTS Pink macules' biopsies of the CM-AVM syndrome showed similar features including a high vascular density encompassing capillaries and numerous thick walled arterioles mainly located in the superficial dermis, a predominance of elongated over round vessels, scarce or absent erythrocytes within the lumina and discrete perivascular inflammation. CMs were characterized by an increased number of capillary-type vessels mostly rounded and located in the upper dermis. AVMs were composed by highly increased numbers of vessels with a branching pattern involving the full thickness of the dermis, without erythrocytes within the lumina, Wilms tumour 1 protein was positive in the endothelial cells both in pink macules of the CM-AVM and in arteriovenous malformations.

Hypotrichosis associated with capillary malformation-arteriovenous malformation syndrome

The British journal of dermatology, 2015

Capillary malformation-arteriovenous malformation (CM-AVM) syndrome is associated with multifocal small CMs and a high risk for high-flow lesions. It is an autosomal dominant disorder, caused by RASA1 gene mutations. Recently, two novel clinical features have been identified: numerous small pale halos with central punctate red spots, and naevus anemicus. To identify the prevalence of the new clinical manifestations in our patients with CM-AVM. The secondary objective was to investigate the presence of other skin lesions. We retrospectively searched the picture database of our department for cases with a clinical diagnosis of CM-AVM, based on the identification of multiple cutaneous CMs and a negative history of epistaxis. We prospectively conducted a clinical and dermoscopic skin examination in all of these patients. Seven patients with multiple CMs were found, and only in one case was a cutaneous AVM present. Five patients had red punctate spots surrounded by pale halos on the uppe...

Capillary Malformation–Arteriovenous Malformation, a New Clinical and Genetic Disorder Caused by RASA1 Mutations

The American Journal of Human Genetics, 2003

Capillary malformation (CM), or "port-wine stain," is a common cutaneous vascular anomaly that initially appears as a red macular stain that darkens over years. CM also occurs in several combined vascular anomalies that exhibit hypertrophy, such as Sturge-Weber syndrome, Klippel-Trenaunay syndrome, and Parkes Weber syndrome. Occasional familial segregation of CM suggests that there is genetic susceptibility, underscored by the identification of a large locus, CMC1, on chromosome 5q. We used genetic fine mapping with polymorphic markers to reduce the size of the CMC1 locus. A positional candidate gene, RASA1, encoding p120-RasGAP, was screened for mutations in 17 families. Heterozygous inactivating RASA1 mutations were detected in six families manifesting atypical CMs that were multiple, small, round to oval in shape, and pinkish red in color. In addition to CM, either arteriovenous malformation, arteriovenous fistula, or Parkes Weber syndrome was documented in all the families with a mutation. We named this newly identified association caused by RASA1 mutations "CM-AVM," for capillary malformationarteriovenous malformation. The phenotypic variability can be explained by the involvement of p120-RasGAP in signaling for various growth factor receptors that control proliferation, migration, and survival of several cell types, including vascular endothelial cells. 1995). This invasion and the subsequent arterial differentiation is also guided by VEGF originating from sensory nerves in the dermis . Defective cutaneous vascular development manifests as malformed vessels that vary in size, location, blood flow, and clinical severity . Capillary malformation (CM), or "port-wine stain," (MIM 163000) is the most common vascular malformation, occurring in 0.3% of newborns (Jacobs and Walton 1976). CM is a flat, cutaneous, slow-flow lesion that is composed of dermal capillary-venular-like channels that are dilated and/or increased in number (Jacobs

Lasers in the Treatment of Vascular Anomalies

Current Otorhinolaryngology Reports, 2014

Lasers have become an integral part in the management of hemangiomas and vascular malformations of many types. Patient outcomes have improved with new discoveries in laser technology and technique. This comprehensive review provides the parameters by which lasers are employed for common vascular anomalies of the head and neck. Research is still needed to identify ideal laser wavelength and treatment intervals for the variety of vascular lesions encountered.

Venous malformations treated with dual wavelength 595 and 1064 nm laser system

Journal of the European Academy of Dermatology and Venereology, 2013

Background & objective Venous malformations (VM) represent a localized error in the embryological development of the venous branch of the circulation. The management of VM is complex and challenging. The aim of this study was to assess the efficacy and safety of combined sequential pulsed dye laser (PDL)-Nd:YAG laser in patients with cutaneous or mucosal VM.

Treatment of vascular malformation with the surgical diode laser: Clinical case

Revista Portuguesa de Estomatologia, Medicina Dentária e Cirurgia Maxilofacial

The objective of this study is to present a clinical case of a hemangioma of the lower lip successfully treated by diode laser. A vascular malformation occupying the entire red zone of the lower right lip, including the inner face, up to the midline, of a 12-year-old patient, was excised in two sessions with a diode laser (GaAl) of 980 nm with fiber tips of Ø 400 μm. Total re-epithelization of the wound was achieved in approximately 21 to 30 days. No postoperative pain occurred, and the functional and aesthetic results were good. The use of diode laser in oral vascular anomalies can be considered a good treatment option. (Rev Port

Comparative Evaluation of Efficacy and Safety of the Diode Laser (980 nm) and Sclerotherapy in the Treatment of Oral Vascular Malformations

International Journal of Vascular Medicine

Background. Vascular malformations are structural abnormalities which are formed by progressively enlarging aberrant and ecstatic vessels without endothelial cell proliferation and composed of the type of vessel involved, i.e., capillary, veins, and arteriovenous. Treatment of vascular malformations may involve many techniques like sclerotherapy, embolization, surgical resection, cryotherapy, laser treatment, or medical therapy. This observational prospective study is aimed at evaluating and comparing the effects and efficacy of diode laser and sclerotherapy in the treatment of oral vascular malformation. Materials and Methods. 40 patients presenting with oral vascular malformation were included in the present study. The patients were divided equally (20 in each) into two groups, i.e., the laser group and sclerotherapy group. Sclerotherapy was performed with 3% sodium tetradecyl sulfate while the laser group was treated with diode laser 980 nm with transmucosal thermophotocoagulatio...