Exacerbation of Erythema Following PDT in Patients With Probable Capillary Malformation-Arteriovenous Malformation Syndrome: Retrospective Analysis of Ultrasound and Clinical Characteristics.

NCT ID: NCT06985563

Last Updated: 2025-05-22

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

COMPLETED

Total Enrollment

16 participants

Study Classification

OBSERVATIONAL

Study Start Date

2024-03-01

Study Completion Date

2024-06-01

Brief Summary

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This study aims to reduce the misdiagnosis rate of PWSs and improve the diagnosis of CM-AVM using ultrasound. Furthermore, we seek to provide more evidence-based recommendations for the treatment of patients with probable CM-AVM syndrome, particularly those presenting with erythema.

Detailed Description

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Background: Exacerbation of erythema following photodynamic therapy (PDT) in patients with probable capillary malformation-arteriovenous malformation (CM-AVM) syndrome, may be attributed to misdiagnosis as port-wine stains (PWSs).

Objective: To identify the ultrasound features of patients with exacerbation of erythema following PDT for a definite diagnosis.

Methods: A retrospective single-center study was conducted on PWS patients with exacerbation of erythema following PDT. The clinical and ultrasonic features were reviewed and analysed. One low-frequency and two high-frequency ultrasound (HFUS) devices were used to examine the lesions and the contralateral normal skin.

Results: The clinical characteristics of 16 patients were consistent with those of high-flow vascular stains (HFVS). Ultrasound findings revealed skin and hypodermis thickening, increased blood flow signals with an arteriovenous malformation (AVM) waveform and deeper, larger blood vessels in the dermis and hypodermis of pink macules (PMs) compared to normal skin. In the absence of genetic diagnosis, PWS patients with exacerbation of erythema following PDT were diagnosed as having probable CM-AVM syndrome.

Limitations: Retrospective single-center design and small sample size. Conclusion: Patients with PWSs are advised to undergo low-frequency ultrasound examinations to avoid misdiagnosis. HFUS can serve as a complementary method to promptly identify underlying AVMs beneath the PMs. We do not recommend PDT treatment for patients with probable CM-AVM syndrome.

Conditions

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Probable Capillary Malformation-Arteriovenous Malformation Syndrome

Study Design

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Observational Model Type

COHORT

Study Time Perspective

RETROSPECTIVE

Study Groups

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Exacerbation of erythema following PDT group

Patients experiencing exacerbation of clinical symptoms, such as darker coloration than before treatment, following at least one Hemoporfin-PDT session were recruited.

HMME-PDT

Intervention Type COMBINATION_PRODUCT

Prior to treatment, patients or their guardians provided written informed consent. Then patients were then prepared for the procedure by fully exposing the treatment area and shielding the surrounding normal skin with a black cloth. Hemoporfin (Shanghai Fudan-Zhangjiang Bio-Pharmaceutical) was administrated intravenously at a dosage of 5.0 mg/kg. Subsequently, patients underwent light radiation at 532 nm (Wuhan Yage Optic and Electronic Technique, Wuhan, China) for 18 to 25 minutes, with a power density randing form 75 to 110 mW/cm2.

Ultrasound examinations

Intervention Type DEVICE

Ultrasound examinations were performed using three types of devices: a Paragon XHD equipped with a L38-22 probe operating at a frequency of 20-30 MHz (Kolo Medical Co., Ltd, Suzhou, China), a DUB Skin Scanner with a 22 MHz probe (Taberna Pro Medicum, German), or a Mindray Resona 7 featuring an L14-5WU linear probe set to a frequency range of 11-12 MHz frequency (Mindray Bio-Medical Electronics Co., Ltd, Shenzhen, China).

Interventions

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HMME-PDT

Prior to treatment, patients or their guardians provided written informed consent. Then patients were then prepared for the procedure by fully exposing the treatment area and shielding the surrounding normal skin with a black cloth. Hemoporfin (Shanghai Fudan-Zhangjiang Bio-Pharmaceutical) was administrated intravenously at a dosage of 5.0 mg/kg. Subsequently, patients underwent light radiation at 532 nm (Wuhan Yage Optic and Electronic Technique, Wuhan, China) for 18 to 25 minutes, with a power density randing form 75 to 110 mW/cm2.

Intervention Type COMBINATION_PRODUCT

Ultrasound examinations

Ultrasound examinations were performed using three types of devices: a Paragon XHD equipped with a L38-22 probe operating at a frequency of 20-30 MHz (Kolo Medical Co., Ltd, Suzhou, China), a DUB Skin Scanner with a 22 MHz probe (Taberna Pro Medicum, German), or a Mindray Resona 7 featuring an L14-5WU linear probe set to a frequency range of 11-12 MHz frequency (Mindray Bio-Medical Electronics Co., Ltd, Shenzhen, China).

Intervention Type DEVICE

Eligibility Criteria

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Inclusion Criteria

* Patients who were greater than or equal to 1 year old at the time of their first HMME-PDT treatment.

* Patients who experiencing exacerbation of clinical symptoms, such as darker coloration than before treatment.

* Patients who have undergone one or more HMME-PDT treatments. ④ Patients with complete basic information and treatment-related information.
Minimum Eligible Age

1 Year

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Second Affiliated Hospital of Xi'an Jiaotong University

OTHER

Sponsor Role lead

Responsible Party

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Weihui Zeng

Professor

Responsibility Role PRINCIPAL_INVESTIGATOR

Principal Investigators

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Weihui Zeng

Role: STUDY_CHAIR

Second Affiliated Hospital of Xi'an Jiaotong University

Locations

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The Second Affiliated Hospital of Xi'an Jiaotong University

Xi'an, Shaanxi, China

Site Status

Countries

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China

References

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Rodriguez Bandera AI, Feito Rodriguez M, Chiloeches Fernandez C, Stewart N, Valdivielso-Ramos M. Role of colour-Doppler high-frequency ultrasonography in capillary malformation-arteriovenous malformation syndrome: a case series. Australas J Dermatol. 2020 Nov;61(4):349-352. doi: 10.1111/ajd.13311. Epub 2020 May 3.

Reference Type BACKGROUND
PMID: 32363619 (View on PubMed)

Cen Q, Sun Y, Zeng X, Liu Y, Liu F, Chen H, Lin X, Cai R. Unilateral and segmental distribution of facial erythema: is it a real port-wine stain? Hereditas. 2020 Jul 7;157(1):27. doi: 10.1186/s41065-020-00143-z.

Reference Type BACKGROUND
PMID: 32635943 (View on PubMed)

Valdivielso-Ramos M, Torrelo A, Martin-Santiago A, Hernandez-Nunez A, Azana JM, Campos M, Berenguer B, Garnacho G, Moreno R, Colmenero I. Histopathological hallmarks of cutaneous lesions of capillary malformation-arteriovenous malformation syndrome. J Eur Acad Dermatol Venereol. 2020 Oct;34(10):2428-2435. doi: 10.1111/jdv.16326. Epub 2020 May 19.

Reference Type BACKGROUND
PMID: 32124491 (View on PubMed)

Liu J, Zhou J, Hu D, Cui L, Li Y, Ye D, Wu T, Mi B, Geng S, Zeng W. Retrospective analysis of Hemoporfin-mediated photodynamic therapy in the treatment of naive port-wine stains. Photodiagnosis Photodyn Ther. 2022 Sep;39:103003. doi: 10.1016/j.pdpdt.2022.103003. Epub 2022 Jul 13.

Reference Type BACKGROUND
PMID: 35840007 (View on PubMed)

Other Identifiers

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2024213

Identifier Type: -

Identifier Source: org_study_id

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