Clinical trial NCT06985563: Exacerbation of Erythema Following PDT in Patients With Probable Capillary Malformation-Arteriovenous Malformation Syndrome: Retrospective Analysis of Ultrasound and Clinical Characteristics.
Official title: Exacerbation of Erythema Following PDT in Patients With Probable Capillary Malformation-Arteriovenous Malformation Syndrome: Retrospective Analysis of Ultrasound and Clinical Characteristics.
Study type: OBSERVATIONAL
Overall status: COMPLETED
Conditions: Probable Capillary Malformation-Arteriovenous Malformation Syndrome
Keywords: exacerbation of erythema following PDT
probable CM-AVM syndrome
Ultrasound
retrospective study
Lead sponsor: Second Affiliated Hospital of Xi'an Jiaotong University
Sponsor class: OTHER
First posted: 2025-05-22
Enrollment: 16
Sex: ALL
Minimum age (years): 1
Standard ages: CHILD
ADULT
OLDER_ADULT
Accepts healthy volunteers: no
Observational model: COHORT
Time perspective: RETROSPECTIVE
Sampling method: NON_PROBABILITY_SAMPLE
Study population: Patients experiencing exacerbation of clinical symptoms, such as darker coloration than before treatment, following at least one Hemoporfin-PDT session were recruited between 2017 and 2023 at the Department of Dermatology, Second Affiliated Hospital of Xi'an Jiaotong University.

Brief summary: 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: 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.

Eligibility criteria: 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.

Primary outcomes:
measure: ultrasound characteristics
timeFrame: The efficacy evaluation will be conducted 3 months after the completion of the 1st HMME-PDT treatment.
Secondary outcomes:
Other outcomes:
Locations:
city: Xi'an
country: China
facility: The Second Affiliated Hospital of Xi'an Jiaotong University
state: Shaanxi
Study officials:
affiliation: Second Affiliated Hospital of Xi'an Jiaotong University
name: Weihui Zeng
role: STUDY_CHAIR
References:
citation: 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.
citation: 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.
citation: 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.
citation: 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.