Occupational respiratory allergy in peach crop workers.

BACKGROUND: Occupational respiratory diseases in workers of peach tree crops have been reported punctually and have been associated with sensitization to proteins present in both pollen and leaf tree. We report the study of 37 workers with respiratory symptoms related to occupational exposure to peach trees. METHODS: Prick tests and specific IgE determinations were performed with extracts from leaves and branches of peach tree. Immunodetection in leaf extract was realized by sodium dodecyl sulfate-polyacrylamide gel electrophoresis SDS-PAGE-immunoblotting with patient sera and rabbit serum anti-Pru p 3. Immunodetection inhibition was performed with rPru p 3 and pollen profilins. The clinical relevance of sensitization was demonstrated by specific bronchial challenge test (SBCT) with peach leaf extract. RESULTS: Most patients suffered symptoms when peach trees had leaves, specifically during thinning and harvesting fruit (rhinoconjunctivitis: 100% and asthma: 67.5%). Sensitization to leaf extract was demonstrated in 86% of patients. IgE-immunoblotting with peach leaf extract revealed in six patient sera a pair of bands of 10 and 16 kDa, and in nine a 16-kDa band. Those bands could be two isoforms of peach leaf lipid transfer proteins( LTP), so the recognition frequency of some LTP isoform by our patient sera was 42%. 33% of the sera recognized a doubled band of about 14.5 kDa and this recognition was inhibited by nPho d 2. The SBCT with peach leaf extract was positive in the asthmatic sensitized patients tested. CONCLUSIONS: Sensitization to peach leaves was the cause of occupational respiratory symptoms in our patients. Some patient sera revealed IgE-binding proteins matching LTP and/or profilin.

Renal amyloidosis in a patient with X-linked agammaglobulinemia (Bruton's disease) and bronchiectasis.

PURPOSE: We present a patient with Bruton's disease and bronchiectasis who developed renal AA amyloidosis. CASE REPORT: A 38 year-old man was diagnosed with X-linked agammaglobulinemia (Bruton's disease) when he was 3 years old, and he has been treated with parenteral immunoglobulin since then. Eighteen years later, he was diagnosed with central pulmonary bronchiectasis by computerized tomography (CT). In 2008, he gradually developed anemia, edema of lower limbs, and loss of weight. METHOD AND RESULTS: Laboratory studies revealed deterioration of renal function, normocytic normochromic anemia and nephrotic range proteinuria. Hepatitis B and C and HIV serology were negative. Ultrasound and CT of abdomen were normal. A renal biopsy revealed deposits with positive PAS and Congo red staining in glomeruli, interstitium, and vessel's walls. Immunohistochemistry showed positive staining of the A amyloid. Direct immunofluorescence was positive with thioflavin and showed focal and glomerular mesangial IgG deposits, suggesting renal AA amyloidosis. For 2 years the patient conducted pharmacological treatment and follow-up for the Nephrology department with poor prognosis and progression of renal function impairment. In January 2011 he began dialysis treatment with improvement, and he is currently on the waiting list for renal transplantation. CONCLUSION: We present a patient with Bruton's disease and bronchiectasis who developed renal AA amyloidosis a finding rarely reported.

Recurrent angioedema due to lysozyme allergy.

A 54-year-old woman suffered an episode of dyspnea and edema affecting her eyelids, tongue, and lips a few minutes after intake of Lizipaina (bacitracin, papain, and lysozyme). She was treated with intravenous drugs and her symptoms improved within 2 hours. She had experienced 3 to 4 bouts of similar symptoms related to the ingestion of cured cheeses or raw egg. Specific serum immunoglobulin (Ig) E against lysozyme was present at a concentration of 0.45 kU/L, and no specific IgE was found against egg white and yolk, ovalbumin, or ovomucoid. Skin prick tests were positive with commercial extracts of egg white and lysozyme but doubtful with yolk, ovalbumin, and ovomucoid. Prick-to-prick tests with raw egg white and yolk gave positive results, but negative results were obtained with cooked egg white and yolk and 5 brands of cheese (3 of them containing lysozyme and the other 2 without lysozyme). Controlled oral administration of papain, bacitracin, and cheeses without lysozyme was well tolerated. We suggest that the presence of lysozyme in a pharmaceutical preparation, cured cheese, and raw egg was responsible for the symptoms suffered by our patient, probably through an IgE-mediated mechanism.

Cutaneous reactions due to diltiazem and cross reactivity with other calcium channel blockers.

BACKGROUND: The spectrum of cutaneous eruptions in association with calcium channel blockers is extensive, varying from exanthemas to severe adverse events. Reactions due to diltiazem occur more frequently than with other calcium channel blockers. Patch testing has been used as confirmatory testing in patients with extensive cutaneous reactions. Cross-reactivity among these drugs have not been established. MATERIAL: We present 3 patients: 1) A 54-year-old man developed a generalized erythema-multiforme-like reaction followed by erythrodermia and exfoliative dermatitis 6-7 days after starting on diltiazem. The drug was stopped and remission was obtained with emollients and systemic corticosteroids and antihistamines within 12 days. 2) A 80-year-old woman experienced a pruritic exanthematous eruption on her trunk which evolved to generalized erythrodermia and superficial desquamation. This reaction appeared 10 days after taking diltiazem, and gradually improved in 10-12 days after discontinuation of this drug. 3) A 79-year-old man presented with erythema and pruritus initially on the back, and then affecting thorax, extremities and face. He had started treatment with diltiazem three days before. Diltiazem was stopped and steroid and antihistamine therapy was given. His skin condition improved, but 3 days later the patient received verapamil with worsening of previous situation. He recovered within 7 days. METHODS AND RESULTS: Two to six months after the reaction, we carried out epicutaneous tests with calcium channel blockers from different groups. Diltiazem proved positive (at 48 and 96 hours) in the three patients; nifedipine was also positive in patient 2, and verapamil in patient 3. Controlled administration of verapamil was well tolerated in patient 2 after the reaction, and the patient 1 has taken nifedipine without problems. CONCLUSIONS: 1) We report 3 cases of cutaneous reactions due to diltiazem. 2) Epicutaneous tests have been useful for diagnosis. 3) As one of patients had positive patch tests to diltiazem and nifedipine, and other one with diltiazem and verapamil, more studies are needed to demonstrate cross reactions among calcium channel blockers.

Cutaneous reactions due to diltiazem and cross reactivity with other calcium channel blockers

Background: The spectrum of cutaneous eruptions in association with calcium channel blockers is extensive, varying from exanthemas to severe adverse events. Reactions due to diltiazem occur more frequently than with other calcium channel blockers. Patch testing has been used as confirmatory testing in patients with extensive cutaneous reactions. Cross-reactivity among these drugs have not been established. Material: We present 3 patients: 1) A 54-year-old man developed a generalized eythema-multiforme-like reaction followed by erythrodermia and exfoliative dermatitis 6-7 days after starting on diltiazem. The drug was stopped and remission was obtained with emollients and systemic corticosteroids and antihistamines within 12 days. 2) A 80-year-old woman experienced a pruritic exanthematous eruption on her trunk which evolved to generalized erythrodermia and superficial desquamation. This reaction appeared 10 days after taking diltiazem, and gradually improved in 10-12 days after discontinuation of this drug.3) A 79-year-old man presented with erythema and pruritus initially on the back, and then affecting thorax, extremities and face. He had started treatment with diltiazem three days before. Diltiazem was stopped and steroid and antihistamine therapy was given. His skin condition improved, but 3 days later the patient received verapamil with worsening of previous situation. He recovered within 7 days. Methods and results: Two to six months after the reaction, we carried out epicutaneous tests with calcium channel blockers from different groups. Diltiazem proved positive (at 48 and 96 hours) in the three patients; nifedipine was also positive in patient 2, and verapamil in patient 3. Controlled administration of verapamil was well tolerated in patient 2 after the reaction, and the patient 1 has taken nifedipine without problems. Conclusions: 1) We report 3 cases of cutaneous reactions due to diltiazem. 2) Epicutaneous tests have been useful for diagnosis. 3) As one of patients had positive patch tests to diltiazem and nifedipine, and other one with diltiazem and verapamil, more studies are needed to demonstrate cross reactions among calcium channel blockers

Introducción: El espectro de las reacciones cutáneas producidas por los antagonistas del calcio es extenso, abarcando desde exantemas hasta reacciones graves. El diltiazem es el implicado con más frecuencia. Para el diagnóstico, las pruebas epicutáneas son útiles en las reacciones cutáneas extensas. La reactividad cruzada entre estos fármacos no ha sido establecida. Material: se presentan tres pacientes: 1) Varón de 54 años, que desarrolló una reacción eritema multiforme-like que evolucionó a eritrodermia y dermatitis exfoliativa, 6-7 días después de iniciar tratamiento con diltiazem. Mejoró en 12 días tras suspender el fármaco y recibir tratamiento con emolientes y corticoides y antihistamínicos sistémicos. 2) Mujer de 80 años que sufrió una erupción exantemática en el tronco, con evolución a eritrodermia y descamación superficial, 10 días después de comenzar a tomar diltiazem. Mejoró a los 10-12 días de interrumpirlo. 3) Varón de 79 años que presentó al 3er. día de tratamiento con diltiazem eritema y prurito, al principio en la espalda y luego en tronco, extremidades y cara. Mejoró al suspenderlo, pero volvió a empeorar al tomar verapamilo. Métodos y resultados: Se realizaron pruebas epicutáneas con diltiazem y otros antagonistas del calcio 2-6 meses después de la reacción. Diltiazem fue positivo en los 3 pacientes (a las 48 y 96 h); nifedipino fue también positivo en el paciente 2 y verapamilo en el paciente 3. Verapamilo fue administrado de forma controlada al paciente 2 con buena tolerancia y el paciente 1 ha tomado nifedipino sin problemas. Conclusiones: 1) Comunicamos 3 casos de reacciones cutáneas producidas por diltiazem. 2) Las pruebas epicutáneas han sido útiles para el diagnóstico. 3) En uno de los pacientes las pruebas epicutáneas fueron positivas para diltiazem y nifedipino y en otro paciente la positividad fue al diltiazem y verapamilo, por lo que son necesarios más estudios para demostrar la reactividad cruzada entre los antagonistas del calcio

Tolerance and short-term effect of a cluster schedule with pollen-extracts quantified in mass-units.

We performed a prospective, multicenter study to assess the tolerance and possible short-term effects of allergen vaccines administered according to a cluster schedule in the months immediately preceding the onset of the pollen season. The study was carried out in eight centers and included 191 patients (children and adults) with allergic respiratory disease due to sensitization to olive tree and/or grass pollen. Of these, 34 patients acted as controls and the remaining patients received immunotherapy administered in the initiation phase according to a cluster schedule of eight doses injected on four visits. After 3 months of treatment, significant differences were found between the two groups in medication consumption (antihistamines in drops and oral formulations: p = 0.045 and p = 0.001, respectively; short-acting beta2-agonist treatments: p = 0.004) and respiratory symptoms (wheezing and coughing: p = 0.035 and 0.014, respectively). The cytokine profile (interleukin [IL]-4, 5, 10 and 2, interferon [IFN-gamma], and tumor necrosis factor [TNF-alpha]) was determined before the start of treatment and at the end of follow-up (4-5 months). Levels of IL-4, 5 and 10 (Th2 profile) decreased while those of IL-2, IFN-gamma, and TNF-alpha (Th1 profile) decreased. These differences were more marked in the active group than in the control group but were not statistically significant. No severe adverse effects were recorded. This study shows that the schedule tested had an acceptable tolerance profile and produced significant changes in symptom and medication scores after a few months of treatment. A double-blind, placebo-controlled study is needed to confirm these results.

Tolerance and short-term effect of a cluster schedule with pollen-extracts quantified in mass-units

We performed a prospective, multicenter study to assess the tolerance and possible short-term effects of allergen vaccines administered according to a cluster schedule in the months immediately preceding the onset of the pollen season. The study was carried out in eight centers and included 191 patients (children and adults) with allergic respiratory disease due to sensitization to olive tree and/or grass pollen. Of these, 34 patients acted as controls and the remaining patients received immunotherapy administered in the initiation phase according to a cluster schedule of eight doses injected on four visits. After 3 months of treatment, significant differences were found between the two groups in medication consumption (antihistamines in drops and oral formulations: p = 0.045 and p = 0.001, respectively; short-acting β2-agonist treatments: p = 0.004) and respiratory symptoms (wheezing and coughing: p = 0.035 and 0.014, respectively). The cytokine profile (interleukin [IL]-4, 5, 10 and 2, interferon [IFN-γ], and tumor necrosis factor [TNF-α]) was determined before the start of treatment and at the end of follow-up (4-5 months). Levels of IL-4, 5 and 10 (Th2 profile) decreased while those of IL-2, IFN-γ, and TNF-α (Th1 profile) decreased. These differences were more marked in the active group than in the control group but were not statistically significant. No severe adverse effects were recorded. This study shows that the schedule tested had an acceptable tolerance profile and produced significant changes in symptom and medication scores after a few months of treatment. A double-blind, placebo-controlled study is needed to confirm these results (AU)

Se ha llevado a cabo un estudio prospectivo y multicéntrico con el objetivo de valorar la tolerancia y posible efecto a corto plazo de las vacunas alergénicas administradas bajo pauta cluster en los meses inmediatamente anteriores al inicio de la estación polínica. El estudio se realizó en 8 centros, incluyéndose un total de 191 pacientes (niños y adultos) con enfermedad alérgica respiratoria por sensibilización a polen de olivo y/o gramíneas. De ellos, 34 actuaron como controles y a los pacientes restantes se les administró inmunoterapia bajo una pauta cluster, en la fase de iniciación, de 8 dosis administradas en 4 visitas. Tras 3 meses de tratamiento, se registraron diferencias significativas entre ambos grupos en el consumo de medicación (antihistamínicos en colirio y orales -p = 0,045 y p = 0,001 respectivamente- y ß2 de corta duración -p = 0,004-) así como en síntomas pulmonares (sibilancias y tos -p = 0,035 y 0,014 respectivamente-). Por otro lado, se determinó el perfil de citocinas (IL-4, 5, 10 y 2, IFN-gamma y TNF-a) de forma previa al inicio del tratamiento y al finalizar el seguimiento (4-5 meses). Se observaron descensos en los niveles de IL-4, 5 y 10 (perfil TH2) y aumento en los valores de IL-2, IFN-gamma y TNF-a (perfil TH1), más marcados en el grupo activo que en el control, sin alcanzar significación estadística. No se registraron efectos adversos severos. Por tanto, podemos observar que la pauta ensayada mostró un adecuado perfil de tolerancia, y tras pocos meses de tratamiento se registraron cambios significativos en la puntuación de síntomas y medicación, siendo necesaria la realización de un estudio con un diseño doble ciego frente a placebo para confirmar los resultados obtenidos (AU)

Cheilitis granulomatosa of Melkersson-Rosenthal syndrome: treatment with intralesional corticosteroid injections.

BACKGROUND: Melkersson-Rosenthal syndrome may manifest as the classical triad (orofacial edema, facial nerve palsy and stable lingua plicata) but monosymptomatic manifestations or combinations of typical symptoms are not infrequent. The available therapeutic options provide only limited success or temporary benefit. CASE REPORT: A 20-year-old man presented with a 7-month history of recurrent episodes of swelling of the upper lip without pain, burning or local pruritus. No causative factors, such as food, drugs or latex, or physical, chemical or emotional conditions could be identified. The patient had been treated with oral antihistamines and corticosteroids with no clinical improvement. Physical examination showed firm edema without fovea, limited to the central area of the upper lip without epidermal changes or symptoms on palpation. The patient had a previous history of facial palsy 6 years previously and recurrent episodes of herpes simplex labialis. Skin prick tests with inhalant aeroallergens, food, latex and Anisakis allergens were negative. Laboratory investigation revealed normal complete blood count, erythrocyte sedimentation rate, thyroid hormones, biochemistry, complement components (C3, C4 and C1-esterase inhibitor) and CH50, rheumatoid factor, antinuclear antibodies, immune complexes, protein electrophoresis and immunoglobulins. Thorax and paranasal sinus radiographs were clear. Biopsy of the involved area of the lip showed edema with lymphocytic and plasma cell infiltration and mononuclear perivascular infiltrates without granulomas, suggesting initial granulomatous cheilitis. Because the patient showed lack of response and/or poor tolerance to prior treatments (deflazacort, clofazimine and metronidazole), intralesional triamcinolone injections were administered with satisfactory response from the first session. CONCLUSIONS: Response to available treatments for Melkersson-Rosenthal syndrome is highly variable. In the present case, intralesional triamcinolone injections were effective.

Cheilitis granulomatosa of Melkersson-Rosenthal syndrome: Treatment with intralesional corticosteroid injections

Background: Melkersson-Rosenthal syndrome may manifest as the classical triad (orofacial edema, facial nerve palsy and stable lingua plicata) but monosymptomatic manifestations or combinations of typical symptoms are not infrequent. The available therapeutic options provide only limited success or temporary benefit. Case report: A 20-year-old man presented with a 7-month history of recurrent episodes of swelling of the upper lip without pain, burning or local pruritus. No causative factors, such as food, drugs or latex, or physical, chemical or emotional conditions could be identified. The patient had been treated with oral antihistamines and corticosteroids with no clinical improvement. Physical examination showed firm edema without fovea, limited to the central area of the upper lip without epidermal changes or symptoms on palpation. The patient had a previous history of facial palsy 6 years previously and recurrent episodes of herpes simplex labialis. Skin prick tests with inhalant aeroallergens, food, latex and Anisakis allergens were negative. Laboratory investigation revealed normal complete blood count, erythrocyte sedimentation rate, thyroid hormones, biochemistry, complement components (C3, C4 and C1-esterase inhibitor) and CH50, rheumatoid factor, antinuclear antibodies, immune complexes, protein electrophoresis and immunoglobulins. Thorax and paranasal sinus radiographs were clear. Biopsy of the involved area of the lip showed edema with lymphocytic and plasma cell infiltration and mononuclear perivascular infiltrates without granulomas, suggesting initial granulomatous cheilitis. Because the patient showed lack of response and/or poor tolerance to prior treatments (deflazacort, clofazimine and metronidazole), intralesional triamcinolone injections were administered with satisfactory response from the first session. Conclusions: Response to available treatments for Melkersson-Rosenthal syndrome is highly variable. In the present case, intralesional triamcinolone injections were effective (AU)

Introducción: El síndrome de Melkersson-Rosenthal puede presentarse como la clásica tríada (angioedema, parálisis facial y lengua plicata), pero no es infrecuente que aparezcan formas monosintomáticas y combinaciones de los síntomas típicos. Las opciones terapéuticas disponibles ofrecen una mejoría parcial o transitoria.Caso clínico: Paciente varón de 20 años de edad que refiere un aumento de tamaño del labio superior de 7 meses de evolución, sin dolor, quemazón ni prurito local. No lo relaciona con la ingesta de fármacos, alimentos, factores físicos, emocionales ni manipulación de látex. El cuadro tiene periodos de exacerbaciones y mejoría. Ha realizado tratamiento con antihistamínicos y corticoides orales, persistiendo siempre un edema residual. A la exploración se apreciaba edema sin fóvea, firme, elástico, fundamentalmente en la zona central del labio superior, sin cambios epidérmicos y asintomático a la palpación.Como antecedentes, refería un episodio de parálisis facial 6 años antes y herpes labial recurrente.Los tests cutáneos mediante prick-test con neumoalergenos inhalantes, alimentos y extractos de látex y Anisakis fueron negativos. No se detectaron alteraciones en el hemograma, VSG, hormonas tiroideas, bioquímica, C3, C4, CH50, C1 inhibidor, actividad del C1 inhibidor, serología reumática, ANA, inmunocomplejos circulantes, proteinograma e inmunoglobulinas. El estudio radiológico de tórax y senos paranasales fue normal. El estudio histológico de la muestra obtenida del labio superior mostraba un infiltrado perivascular de linfocitos y células plasmáticas compatible con queilitis granulomatosa en fase inicial.Ante la falta de respuesta y/o mala tolerancia de los tratamientos previos (deflazacort, clofazimina y metronidazol) se optó por realizar infiltraciones locales con triamcinolona acetónido, con respuesta satisfactoria desde la primera sesión.Conclusiones: La respuesta de los síntomas del síndrome de Melkersson-Rosenthal a las medidas terapéuticas disponibles es muy variable. En nuestro caso, la inyección intralesional de corticoides ha resultado eficaz (AU)

Erythrodermia to pseudoephedrine in a patient with contact allergy to phenylephrine.

BACKGROUND: Phenylephrine and pseudoephedrine are sympathomimetic drugs belonging to the phenylamine family. Adverse cutaneous effects associated with these drugs have been reported but, in view of their frequent use, appear to be rare. The very close chemical structures of these drugs could explain potential cross-reactions among them but the results reported in the literature are controversial. CASE REPORT: An 18-year-old woman developed blepharoconjunctivitis after application of phenylephrine and tropicamide eye drops. Four years after this reaction, she took 1 tablet of Narine (pseudoephedrine and loratadine) and 3-4 hours later developed a generalized erythrodermic reaction. Cutaneous biopsy revealed hydropic changes in the basal layer and, in the dermis, moderate edema with slight perivascular lymphocyte and eosinophil infiltrates. Patch tests with European standard series, commercial eye drops, tropicamide, phenylephrine, pseudoephedrine and other sympathomimetic agents were applied to the patient's back. After 47 and 96 hours, only the patches with pseudoephedrine and phenylephrine were positive. CONCLUSIONS: We believe that our patient has presented two different reactions with different clinical outcome and histopathology, which are unlikely to be due to cross-reactivity between the drugs involved. We have found no similar coincidences reported int the literature.

Erythrodermia to pseudoephedrine in a patient with contact allergy to phenylephrine

Background: Phenylephrine and pseudoephedrine are sympathomimetic drugs belonging to the phenylamine family. Adverse cutaneous effects associated with these drugs have been reported but, in view of their frequent use, appear to be rare. The very close chemical structures of these drugs could explain potential cross-reactions among them but the results reported in the literature are controversial. Case report: An 18-year-old woman developed blepharoconjunctivitis after application of phenylephrine and tropicamide eye drops. Four years after this reaction, she took 1 tablet of Narine® (pseudoephedrine and loratadine) and 3-4 hours later developed a generalized erythrodermic reaction. Cutaneous biopsy revealed hydropic changes in the basal layer and, in the dermis, moderate edema with slight perivascular lymphocyte and eosinophil infiltrates. Patch tests with European standard series, commercial eye drops, tropicamide, phenylephrine, pseudoephedrine and other sympathomimetic agents were applied to the patient's back. After 47 and 96 hours, only the patches with pseudoephedrine and phenylephrine were positive. Conclusions: We believe that our patient has presented two different reactions with different clinical outcome and histopathology, which are unlikely to be due to cross-reactivity between the drugs involved. We have found no similar coincidences reported int the literature (AU)

Antecedentes: La fenilefrina y la pseudoefedrina son fármacos simpaticomiméticos que pertenecen a la familia fenilamina. Teniendo en cuenta el amplio uso de estos fármacos, las reacciones adversas cutáneas producidas por ellos no parecen frecuentes. La similitud química de estas sustancias podría explicar la existencia de reacciones cruzadas entre ellas, pero los resultados de la literatura sobre este tema son controvertidos. Caso clínico: Mujer de 18 años de edad, remitida a nuestras consultas por haber presentado a las 3-4 horas de tomar un comprimido de Narine (loratadina y pseudoefedrina) una reacción eritrodérmica generalizada. Cuatro años antes había presentado una bléfaroconjuntivitis tras la aplicación simultánea de 2 colirios, Oculos Fenilefrina 10 per cent y Colircusi Tropicamida . Las pruebas epicutáneas con la batería estándar del GEIDC, los colirios comerciales y con preparaciones de tropicamida, fenilefrina, pseudoefedrina y otros agentes simpaticomiméticos sólo fueron positivas con fenilefrina y pseudoefedrina a las 48 y 96 horas. La biopsia cutánea de la reacción eritrodérmica mostraba cambios hidrópicos en la membrana basal y, en la dermis, moderado edema con ligero infiltrado perivascular de linfocitos y eosinófilos. Conclusiones: Se trata de 2 reacciones diferentes tanto clínica como histopatológicamente, probablemente no explicables por la posible reactividad cruzada entre los fármacos implicados. No hemos encontrado coincidencias similares en la literatura (AU)