Patient’s information

From June 2011 to June 2021, patients with port wine stain and Sturge-Weber syndrome who have already developed significant hyperplasia and obvious asymmetric deformities in maxillofacial region were selected. There were 36 patients with port wine stains, 16 males and 20 females. Age ranged from 8 to 65 years. Among them, 11 patients were with Sturge-Weber syndrome, 4 men and 7 women. This retrospective study was approved by the bioethics committee of the School and Hospital of Stomatology. All patients signed an informed consent form prior to treatment and agreed to use clinical data for academic communication and publication.

Medications

Bleomycin is produced by Pfizer Pharmaceutical Co., Ltd., 15,000 units/stick. Prepared with normal saline, the concentration of bleomycin is 500 units/ml. Triamcinolone acetonide is produced by Zhejiang Xianju Pharmaceutical Factory, 50 mg/5 ml. Prepared with normal saline, triamcinolone acetonide concentration is 5 mg/ml.

Therapeutic method

Inject the drug into lesions with a 5-ml syringe. The injection volume was determined from age and the lesion surface area. Within the safe dose range of the drug applicable in the corresponding age group, the drug dose is determined according to the size of the lesion. The dosage of bleomycin was 500 units/cm2, and the dosage of triamcinolone acetonide was 5 mg/cm2. Drug injection depth: From the dermis layer to the subcutaneous fat layer, the bone attachment site was injected into the periosteum. Drugs were injected in manner of radiation dispersion, the injection spacing was less than 0.5 cm, and the drugs were evenly distributed in the lesions as much as possible. The interval between treatments was determined according to the hyperplasia status and size of port wine stains. If the area of port wine stains was large and the hyperplasia was obvious, it was difficult to cover all lesions with one administration, and the interval between administrations was 2 weeks. If the area of port wine stains was small, a single injection treatment can cover all lesions, and the interval was 4 weeks. For large areas of port wine stains, it can be taken for 2–3 consecutive days to cover all the lesions; the next injection was given after 3–4 weeks. Generally, the interval between initial treatment was short, and the interval between later periods was long. When port wine stains appeared atrophied and thinned, the interval between administrations was 2–3 months. The interval between administrations was not fixed and depended on the condition after each treatment. The lesion was usually injected with the drug 10–15 times. The use of triamcinolone acetonide depended on lesion hyperplasia.

Clinical indication for discontinuation of treatment 1.

When the volume of the lesion became significantly smaller, the tissue became thinner, and the affected side was close to symmetrical with the unaffected side; the treatment was stopped.

2.

The surface of the lesion was wrinkled, and the erythema turned purple or subsided.

Evaluation index

After the treatment, efficacy evaluation was performed, and patients were regularly reviewed and followed up for 4–5 years. Observing items are listed as follows.

Change of histopathology

The histopathological changes of bright erythematous nevus before and after bleomycin treatment were compared.

Changes in the thickness of the lesion

B-scan ultrasonography examination was performed on the same lesion location before and after treatment to observe the change of lesion thickness. Three positions were taken for each lesion, and the location sites were recorded. The measured data was analyzed by t-test.

Changes in facial morphology

Before and after treatment, the lesion site and the face were photographed to observe the changes of lesion thickness, lesion surface color, and facial asymmetry. Patients without pulsed dye laser and photodynamic therapy were included in the observation of erythema change, and there were 7 patients. Patients who had undergone laser treatment were excluded. The plastic surgeon scored the lesions and facial morphology before and after the treatment. According to hyperplasia, facial asymmetry, and erythema, it was divided into five grades: (1) slight, (2) mild, (3) moderate, (4) sub-severe, and (5) severe. The obtained data was analyzed separately. Means and standard deviations were calculated. The Wilcoxon test (IBM SPSS Statistics 19 version) was used to evaluate the differences between the thickness, symmetry, and erythema before and after treatment. P-value < 0.05 was considered significant.

Recurrence of the lesion

Observe whether hypertrophy and erythema exacerbate again. The change was divided into three grades: (1) no recurrence; (2) mild recurrence, slight hyperplasia, and slight color change; and (3) recurrence, obvious hyperplasia, and erythema darkening. P-value < 0.05 was considered significant.

The patient’s self-evaluation

The change before and after treatment of the lesion was divided into three levels: (1) no change, (2) change, and (3) obvious change.

Patient satisfaction with the results of treatment

The patient’s satisfaction with the treatment results was divided into three levels: (1) dissatisfied, (2) satisfied, and (3) very satisfied.