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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0040" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">To the Editor&#58;</span></p><p id="par0005" class="elsevierStylePara elsevierViewall">Photodynamic therapy &#40;PDT&#41; is a light therapy that involves the production of reactive oxygen species and free radicals to induce oxidation in biological tissues&#46; The effect is achieved through the application of a photosensitizing agent&#44; which is then activated by exposure to wavelength-specific light&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The most commonly used photosensitizer is methyl aminolevulinate &#40;MAL&#41;&#46; PDT is primarily used in the treatment of basal cell carcinoma&#44; actinic keratosis&#44; and Bowen disease&#44; although it can also be used to treat certain non-neoplastic diseases&#46; The most common side effects are pain and local inflammation&#59; wheals are very rare&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We present the cases of 2 pediatric patients who developed urticarial reactions to PDT&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The first patient was an 11-year-old girl with nevoid basal cell carcinoma syndrome &#40;Gorlin syndrome&#41; who presented with the characteristic clinical manifestations&#44; including frontal bossing&#44; hypertelorism&#44; small ears&#44; syndactyly&#44; palmar pits&#44; and a large number of small&#44; pedunculated&#44; papular lesions on the neck&#44; axillas&#44; groin&#44; and popliteal fossae&#46; Histologic examination of these lesions revealed basal cell carcinoma&#46; The lesions had previously been treated with cryotherapy&#44; electrocoagulation&#44; and imiquimod&#46; PDT treatment was prescribed and administered on several lesions on the upper back and the neck according to the standard protocol&#58; MAL 160<span class="elsevierStyleHsp" style=""></span>mg&#47;g cream &#40;Metvix&#41; applied under occlusion for 3<span class="elsevierStyleHsp" style=""></span>hours followed by illumination using an Aktilite lamp at a dose of 37<span class="elsevierStyleHsp" style=""></span>J&#47;cm<span class="elsevierStyleSup">2</span>&#46; Three minutes after starting illumination&#44; semicircular wheals appeared in the area being treated&#44; and light exposure was stopped &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The lesions disappeared after a few hours without treatment and did not recur&#46; Some months later the patient underwent provocation testing with the MAL cream applied under occlusion for 3<span class="elsevierStyleHsp" style=""></span>hours followed by illumination&#46; Wheals appeared in the illuminated area where the cream had been applied&#44; but no lesions were observed in the area exposed to light without prior application of cream &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The wheals disappeared spontaneously and there was no recurrence&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second patient was a 4-year-old girl with no relevant past history or family history who presented with brownish&#44; erythematous&#44; maculopapular lesions in a blaschkoid distribution affecting the dorsum and anterior aspect of the left forearm&#44; thigh&#44; and left leg&#46; Porokeratosis was revealed on histology &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46; The lesions had not responded to treatment with topical corticosteroids or retinoids&#46; Treatment with PDT was administered using the standard protocol without pretreatment curettage&#46; After 2<span class="elsevierStyleHsp" style=""></span>minutes of light exposure&#44; large erythematous&#44; edematous lesions appeared in the area being treated and the procedure was stopped &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; The lesions disappeared after a few hours without treatment and did not recur&#46; Provocation testing was not carried out due to the young age of the patient&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">In both patients&#44; Giemsa staining of skin biopsies taken prior to PDT showed no significant increase in the number of mast cells&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Urticarial reactions to PDT using 5-aminolevulinic acid and MAL have been described in the literature&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> A 2008 study reported a 0&#46;9&#37; prevalence &#40;12&#47;1353 patients&#41; for severe itching and wheals within the first minute of illumination&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The patients most predisposed to reaction were those who had received more than 7 courses of treatment &#40;3&#46;8&#37; prevalence&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Those authors postulated that this type of urticaria may be mediated by immunoglobulin E with histamine release from mast cells in the dermis&#46; This pathogenesis is consistent with the recurrent nature of the reactions in subsequent treatments&#44; the satisfactory control of these reactions through the administration before treatment of an antihistamine such as cetirizine&#44; and the immediate appearance of urticaria in areas not previously treated with PDT&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> In our patients&#44; urticaria appeared during the first course of treatment&#46; In the first patient&#44; a subsequent provocation skin test confirmed that the reaction was produced by the combination of MAL and illumination&#44; and not by either one separately&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Of particular note was the especially extensive reaction in our patients&#46; We found no reports in the literature of nevoid basal cell carcinoma syndrome involving a similar reaction to PDT&#44; despite the increasing use of PDT in these patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> Similarly&#44; we found no reports of urticarial reactions in patients with porokeratosis treated with PDT&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> An additional associated factor could be the young age of these patients&#46; We found no cases in the literature of reaction to PDT in children&#44; but this treatment is rarely used in this age group&#46; It has been suggested that mast cells may play a role in this type of reaction and at the same time participate in the nonspecific immune response that results in the tissue damage caused indirectly by PDT&#46; We therefore postulated the possibility of high mast cell counts in the affected areas&#44; but this hypothesis was ruled out by the results of Giemsa staining&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary&#44; these 2 rare cases of urticaria during PDT in children&#44; a previously unreported reaction in this age group&#44; demonstrate the need for dermatologists to be aware of this reaction so that they will stop illumination promptly and treat the resulting lesions appropriately&#46;</p></span>"
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Case and Research Letters
Urticaria-Like Reaction Secondary to Photodynamic Therapy in 2 Pediatric Patients
Reacción urticariforme secundaria a terapia fotodinámica en 2 pacientes pediátricos
A. Miguéleza,
Corresponding author
anamig@telefonica.net

Corresponding author.
, A. Martín-Santiagob, A. Bauzáb, Y. Gilabertec
a Servicio de Dermatología, Hospital Universitario de la Princesa, Madrid, Spain
b Servicio de Dermatología, Hospital Universitario Son Espases, Palma de Mallorca, Spain
c Servicio de Dermatología, Hospital San Jorge, Huesca, Spain
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A&#44; Observe the cornoid lamella&#8212;a column of stacked parakeratotic cells extending through the stratum corneum characteristic of porokeratosis&#46; Hematoxylin-eosin&#44; original magnification &#215;100&#46; B&#44; Large erythematous&#44; edematous lesions on the left leg on the area where PDT treatment was applied&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0040" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">To the Editor&#58;</span></p><p id="par0005" class="elsevierStylePara elsevierViewall">Photodynamic therapy &#40;PDT&#41; is a light therapy that involves the production of reactive oxygen species and free radicals to induce oxidation in biological tissues&#46; The effect is achieved through the application of a photosensitizing agent&#44; which is then activated by exposure to wavelength-specific light&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The most commonly used photosensitizer is methyl aminolevulinate &#40;MAL&#41;&#46; PDT is primarily used in the treatment of basal cell carcinoma&#44; actinic keratosis&#44; and Bowen disease&#44; although it can also be used to treat certain non-neoplastic diseases&#46; The most common side effects are pain and local inflammation&#59; wheals are very rare&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We present the cases of 2 pediatric patients who developed urticarial reactions to PDT&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The first patient was an 11-year-old girl with nevoid basal cell carcinoma syndrome &#40;Gorlin syndrome&#41; who presented with the characteristic clinical manifestations&#44; including frontal bossing&#44; hypertelorism&#44; small ears&#44; syndactyly&#44; palmar pits&#44; and a large number of small&#44; pedunculated&#44; papular lesions on the neck&#44; axillas&#44; groin&#44; and popliteal fossae&#46; Histologic examination of these lesions revealed basal cell carcinoma&#46; The lesions had previously been treated with cryotherapy&#44; electrocoagulation&#44; and imiquimod&#46; PDT treatment was prescribed and administered on several lesions on the upper back and the neck according to the standard protocol&#58; MAL 160<span class="elsevierStyleHsp" style=""></span>mg&#47;g cream &#40;Metvix&#41; applied under occlusion for 3<span class="elsevierStyleHsp" style=""></span>hours followed by illumination using an Aktilite lamp at a dose of 37<span class="elsevierStyleHsp" style=""></span>J&#47;cm<span class="elsevierStyleSup">2</span>&#46; Three minutes after starting illumination&#44; semicircular wheals appeared in the area being treated&#44; and light exposure was stopped &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The lesions disappeared after a few hours without treatment and did not recur&#46; Some months later the patient underwent provocation testing with the MAL cream applied under occlusion for 3<span class="elsevierStyleHsp" style=""></span>hours followed by illumination&#46; Wheals appeared in the illuminated area where the cream had been applied&#44; but no lesions were observed in the area exposed to light without prior application of cream &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The wheals disappeared spontaneously and there was no recurrence&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second patient was a 4-year-old girl with no relevant past history or family history who presented with brownish&#44; erythematous&#44; maculopapular lesions in a blaschkoid distribution affecting the dorsum and anterior aspect of the left forearm&#44; thigh&#44; and left leg&#46; Porokeratosis was revealed on histology &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46; The lesions had not responded to treatment with topical corticosteroids or retinoids&#46; Treatment with PDT was administered using the standard protocol without pretreatment curettage&#46; After 2<span class="elsevierStyleHsp" style=""></span>minutes of light exposure&#44; large erythematous&#44; edematous lesions appeared in the area being treated and the procedure was stopped &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; The lesions disappeared after a few hours without treatment and did not recur&#46; Provocation testing was not carried out due to the young age of the patient&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">In both patients&#44; Giemsa staining of skin biopsies taken prior to PDT showed no significant increase in the number of mast cells&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Urticarial reactions to PDT using 5-aminolevulinic acid and MAL have been described in the literature&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> A 2008 study reported a 0&#46;9&#37; prevalence &#40;12&#47;1353 patients&#41; for severe itching and wheals within the first minute of illumination&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The patients most predisposed to reaction were those who had received more than 7 courses of treatment &#40;3&#46;8&#37; prevalence&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Those authors postulated that this type of urticaria may be mediated by immunoglobulin E with histamine release from mast cells in the dermis&#46; This pathogenesis is consistent with the recurrent nature of the reactions in subsequent treatments&#44; the satisfactory control of these reactions through the administration before treatment of an antihistamine such as cetirizine&#44; and the immediate appearance of urticaria in areas not previously treated with PDT&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> In our patients&#44; urticaria appeared during the first course of treatment&#46; In the first patient&#44; a subsequent provocation skin test confirmed that the reaction was produced by the combination of MAL and illumination&#44; and not by either one separately&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Of particular note was the especially extensive reaction in our patients&#46; We found no reports in the literature of nevoid basal cell carcinoma syndrome involving a similar reaction to PDT&#44; despite the increasing use of PDT in these patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> Similarly&#44; we found no reports of urticarial reactions in patients with porokeratosis treated with PDT&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> An additional associated factor could be the young age of these patients&#46; We found no cases in the literature of reaction to PDT in children&#44; but this treatment is rarely used in this age group&#46; It has been suggested that mast cells may play a role in this type of reaction and at the same time participate in the nonspecific immune response that results in the tissue damage caused indirectly by PDT&#46; We therefore postulated the possibility of high mast cell counts in the affected areas&#44; but this hypothesis was ruled out by the results of Giemsa staining&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary&#44; these 2 rare cases of urticaria during PDT in children&#44; a previously unreported reaction in this age group&#44; demonstrate the need for dermatologists to be aware of this reaction so that they will stop illumination promptly and treat the resulting lesions appropriately&#46;</p></span>"
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Article information
ISSN: 15782190
Original language: English
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