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Hydroa vacciniforme is a rare, idiopathic photodermatosis with an onset in childhood and characterized by acute vesiculation, crusting and scarring following sun exposure. Familial cases are extremely rare with only one previous report. We report a brother and sister, both of whom have developed hydroa vacciniforme.
Hydroa vacciniforme is a troublesome and scarring photosensitivity disorder for which treatment is unsatisfactory. Dietary fish oil rich in omega-3 polyunsaturated fatty acids reportedly increases the resistance to ultraviolet-induced erythema and rash provocation in polymorphic light eruption. We report for the first time the response of hydroa vacciniforme to dietary fish oil. Three Caucasian boys with the condition were placed on MaxEPA, five capsules daily. Phototesting was performed at baseline and after 3 months supplementation. At baseline, low erythemal thresholds were seen to monochromated UVA at 350 and 370 nm in all three boys, while one also had a low threshold to 320 nm (UVA) and another showed a low threshold to 300 nm (UVB). Broad-band UVA provocation challenge produced typical skin lesions in all the subjects. Following fish oil, all the boys showed reduced erythemal sensitivity to UVA and one also showed reduced sensitivity to UVB. Provocation challenge revealed a reduced response in all three children. Clinically, these changes were accompanied by pronounced improvement in one child, mild improvement in the second child, but no improvement in the third. The third boy subsequently showed good clinical response to azathioprine.
Hydroa vacciniforme is a rare chronic photodermatosis with onset in childhood. It is characterized by recurrent vesicles healing with scars. We report a typical case of a young, 8-year-old boy showing favorable evolution under treatment with chloroquine and a topical sunscreen.
Hydroa vacciniforme vesicles can be reproduced experimentally with repetitive UVA irradiations, but this photo-reproduction is not constant. The aim of this study was to search for the factors that influence photo-reproduction. To reproduce hydroa vacciniforme lesions six patients underwent repetitive UVA and polychromatic irradiations on the back. In four patients out of six, UVA irradiation with high doses induced papulo-vesicular lesions. Photo-induced lesions were very close to those induced by sunlight. Photo-reproduction failed when phototesting was done after or shortly before remission. Therefore, the absence of photo-reproduction appears to be a good prognostic factor.
Patients with hydroa vacciniforme (HV)-like eruptions and malignant potential have been reported from Asia and Mexico, and those patients frequently had an associated latent Epstein-Barr virus (EBV) infection. In order to elucidate the association of latent EBV infection with HV, we studied six children with typical manifestations of HV by detection of EBV genes and EBV-related RNAs in biopsy specimens from cutaneous lesions. Cutaneous lesions of all six children with typical HV contained EBV-encoded small nuclear RNA (EBER)+ cells in 3-10% of the dermal infiltrates, whereas no Bam HI-H, l-fragment (BHLF) mRNA, or transcripts encoding EA-D antigen, were detected. No EBER + cells were detected in other inflammatory or benign lymphoproliferative skin disorders tested. Polymerase chain reaction amplification confirmed the presence of EBV DNA sequences in five of six biopsy specimens from the patients. Latent EBV infection is associated with the development of cutaneous lesions of HV.
Hydroa vacciniforme is a rare, chronic photodermatosis with onset in childhood that is characterized by recurrent vesiculation, necrosis, and vacciniform scarring limited to sun-exposed skin. We report a case of nonvesicular hydroa vacciniforme in which only extensive crusting associated with hypertrophic scarring on sun-exposed skin was present. Multiple exposures to UVA, however, reproduced lesions with the typical clinical and histologic features of hydroa vacciniforme. Results of blood, urine, and fecal porphyrin studies were normal.
Hydroa vacciniforme is a rare photosensitivity disorder with onset in childhood. The distinctive lesion is a vesicle which heals with scarring. We report a case of hydroa vacciniforme in which an abnormal minimal erythema dose to wavelengths of 322 to 370 nm within the ultraviolet A (UVA) range was demonstrated. Vesicles could be induced only with multiple exposures to UVA. Increased tolerance to UVA erythema was induced by multiple UVB exposures, although tanning was poor. Of note were the presence of several halo nevi and a history of the loss of ability to tan. The clinical features and appropriate laboratory evaluation of hydroa vacciniforme are reviewed.
Two patients with hydroa vacciniforme, a rare photodermatosis of unknown etiology, demonstrated distinctive scarring and vesiculobullous skin lesions on light-exposed body areas. Results of blood and urine porphyrin studies were normal, and no systemic abnormalities were noted. A small bullous lesion was produced in normal skin in case 1 with 15 times the minimal erythema dose of ultraviolet energy. The conditions of both patients improved while they were taking beta carotene orally.
Hydroa vacciniforme is a rare, chronic photodermatosis with onset in childhood. Multiple exposures to UV-A reproduced the symptoms and the vesicular and scarring lesions typical of the sun-induced disease in our patient. Treatment with hydroxychloroquine sulfate, but not indomethacin, reduced the photosensitivity both to sunlight and to artificial UV-A light. Serum complement levels were low while the disease was active and returned to normal after treatment of the patient with hydroxychloroquine.
Hydroa vacciniform is a rare recurrent photosensitive skin condition in which deep-seated vesicles appear on sun-exposed areas and heal leaving a depressed scar. Two cases are reported, with particular emphasis on phototesting investigations. Results of blood and urine porphyrin studies were normal and no systemic abnormalities were noted. Phototesting may be viewed as an important diagnostic aid, since the induction of lesions, clinically identical to hydroa vacciniform can occur following sequential exposure to UV-A, as was the case in one of our patients.
Hydroa vacciniforme Bazin is a rare photodermatosis characterized by bullous skin lesions and distinctive scarring. Early diagnosis is important to provide disfiguring scarring at the light exposed areas of the skin. The onset of the disease occurs predominantly in early childhood and involutes spontaneously at puberty. The case of a 5 years old girl is reported and differentiation from other photodermatoses is discussed.
Hydroa vacciniforme is a rare disorder manifested in early childhood by recurrent photoinduced vesicles that heal with scarring. We report a case in which repetitive exposures to artificial ultraviolet light in the A range reproduced the clinical findings induced by natural sunlight. Phototesting may be viewed as an important diagnostic aid, as the induction of lesions clinically identical to hydroa vacciniforme can provide a reliable criterion for the diagnosis.
Ten patients with hydroa vacciniforme are reported. The condition appears to be a distinct clinical and histological entity. Haematological, biochemical, immunological, bacteriological and viral investigations were negative. Three patients demonstrated low minimal erythema doses (MEDs) following monochromatic ultraviolet (UVA) irradiation of back skin; such UVA sensitivity may be a feature of hydroa vacciniforme. Treatment was unsatisfactory, although spontaneous improvement in the condition tended to occur and regular application of sunscreens with high protection factors against both UVA and UVB was helpful.
Hydroa vacciniforme is a rare, chronic, photosensitive disorder manifested in childhood by recurrent vesicles that heal with scarring. Reproduction of vesicles with repetitive UVA phototesting may be an important diagnostic aid. Recurrent eruption resulted in severe scarring of the face and a flexion contracture of the finger in our patient. The clinical features and laboratory evaluation of hydroa vacciniforme are reviewed.
The authors described three cases of Bazin's hydroa vacciniforme. Study of the history of the disease enables present nosological conceptions to be better understood. Diagnosis of hydroa vacciniforme is essentially clinical : appearance of the rash, periodicity and spontaneous improvement after puberty are characteristic. Hydroa vacciniforme has to be distinguished from porphyrias and polymorphous light sensitive eruptions. The etiology of this photodermatosis of child is unknown, but associated disturbances in tryptophan metabolism in some cases seem to be linked to a vitamin B6 deficiency.
A 7-year-old Thai boy had several episodes of hydroa vacciniforme which were accompanied by an anterior uveitis with corneal clouding and stellate keratic precipitates. Wearing sunglasses prevented additional eye symptoms despite recurrence of the skin lesions. Repetitive UVA phototesting reproduced the typical skin lesions with fever and malaise. No reproduction of skin lesions was revealed by repetitive UVB phototesting. One should be aware of eye involvement in hydroa vacciniforme, and those who experience the eye involvement should be advised to wear protective sunglasses.
OBJECTIVE: To describe a severe type of hydroa vacciniforme (HV) in order to provide basis for treatment and prevention of this disease. METHODS: Nine cases of HV were collected in our outpatient department, and clinical and laboratory examinations were performed. RESULTS: All the cases had erythemas, papules, bullae, erosions or ulcers on exposed sites, with recurrences, gradually resulting in deformation. Five patients suffered from hand deformation manifesting rigidity, flexor tetanus, or malposition of the first, second and third interphalangeal joints of hands. Two of the above mentioned patients manifested partial bone absorption of fingers. Five patients showed partial defect of auricle. Two patients showed saddle nose, and partial absorption of nasal cartilage. One patient displayed cicatricial contracture of lower lip, with incisor extrusion. Four patients showed opacity of cornea. Through laboratory examinations, porphyrinopathies were excluded. Phototests showed increased sensitivity to ultraviolet-A radiation (UVA). CONCLUSIONS: Based on the clinical features and laboratory examinations, these patients were diagnosed as having severe HV. It is believed that this condition belongs to a disease spectrum induced by ultraviolet light. Our analysis suggests that for children with severe type of HV, preventive measures and therapeutic agents should be taken as soon as possible in order to avoid deterioration of the disease and malformations.
Three cases of "hydroa vacciniforme" are reported. The clinicopathological findings are emphasized. In two cases the patients presented keloids on the blister scars and in one of them there was association with focal epilepsia. The occular complications in the "hydroa vacciniforme" are uncommon but severe. The studies of cellular photobiology ("in vitro" irradiation of lymphocytes with U.V. rays) showed an imapirement of the ADN dark1repair, as demonstrated by other authors.