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Profiles and Prognosis of Severe Infantile Atopic Dermatitis with Food Allergy
Abstracts S239
J ALLERGY CLIN IMMUNOL VOLUME 117, NUMBER 2
Induction of a Pathergy-Like Reaction Following Prick Skin Testing T. L. Johnson, II1, P. McCleskey2, M. Rathkopf1, J. Meffert2, L. L. Hagan1; 1Allergy/Immunology, Wilford Hall Medical Center, Lack land AFB, TX, 2Dermatology, Wilford Hall Medical Center, Lackland AFB, TX. RATIONALE: A 46-year-old female who received 6 months of immunotherapy for perennial and seasonal allergic rhinitis 5 years prior to presentation returned for consideration of restarting immunotherapy secondary to uncontrolled rhinoconjunctivitis. The patient had no other chronic illnesses and lacked symptoms suggestive for Behcet’s disease. Past history was negative for local or systemic reactions to previous skin testing and immunotherapy injections. METHODS: Prick skin testing was performed utilizing the Quintest® device to an aeroallergen panel consisting of 53 antigens to assess for the development of new sensitizations. RESULTS: The patient had positive results to multiple trees, weeds, molds, grasses, dust mite and cat. Five days after the testing procedure the patient called and stated that she developed pustules and pruritus at many of the skin test sites. The pustules were first noted on the day following testing. Upon examination the patient had round erythematous patches with central hyperpigmentation along with erosions and crusts. Lesions were present at 37 of 40 sites that had a 1+ or greater reaction (including histamine control). Out of 15 test sites (including negative control) that lacked reactivity at the time of initial reading; 2 had the development of the above lesions. Histopathologic examination revealed confluent epidermal necrosis, extensive interface vacuolar changes, and focal subepidermal vesicle formation. Dermal changes included a perivascular lymphocytic inflammatory infiltrate with endothelial swelling and few eosinophils or neutrophils. CONCLUSIONS: This report may represent a previously uncharacterized pathergy-like reaction induced by skin testing in a patient that to date lacks symptoms of known pathergy associated illnesses.
924
Profiles and Prognosis of Severe Infantile Atopic Dermatitis with Food Allergy M. Iguchi1, M. Ebisawa2, H. Tachimoto2; 1Jikei University school of Medicine, Tokyo, JAPAN, 218-1 Sakuradai, Department of pediatrics, National Organization Sagamihra National Hospital, Sagamihara city, Kanagawa, JAPAN. RATIONALE: The aim of this study was to clarify the profiles and prognosis of severe infantile atopic dermatitis (AD) (n=67) with food allergy (FA) against more than 2 food antigens. METHODS: All subjects were admitted to the Sagamihara National Hospital to control AD symptoms and to receive diagnosis of FA from 1997 to 2003. We analyzed their patients’ profiles and the prognosis of atopic condition for 5 years. RESULTS: 97% of their chronic eczema started by 4 months old. The number of food antigens determined by food provocation was decreased by 1.1 antigens (from 4.6 to 3.5) compared with the number of sensitized food antigens proved by specific IgE or SPT. Hen’s egg was the most common, followed by cow’s milk, wheat, and soybean. The number of food antigens had become less and less, finally it reached 1.4 items at the age of five. The release of egg elimination at the age of five was still only 30%. In addition the age when the release rate of cow’s milk elimination reached 50% was at 5 y, however, those for wheat and soybean were at 2 y. Although most of the subjects used steroid ointment regularly at the time of discharge, the percentage of “regular use” at 5 years old decreased by 10%. Incidence of bronchial asthma from the subjects at the age of four reached 53%. CONCLUSIONS: Their prognosis was relatively fair except for the prolonged egg and cow’s milk FA and the high incidence of BA from the subjects. Funding: Ministry of Health, Labor, and Welfare
925
Randomized, Placebo-Controlled Trial of Lactobacillus rhamnosus GG as Treatment of Mild to Moderate Atopic Dermatitis in Infancy C. Grüber1, M. Wendt2, S. Lau1, M. Kulig1, U. Wahn1, T. Werfel3, B. Niggemann1; 1Dept. of Pediatric Pneumology/Immunology, Charité Universitätsmedizin Berlin, Berlin, GERMANY, 2Department of Dermatology and Allergology, Hanover Medical University, Hanover, GERMANY, 3(2) Department of Dermatology and Allergology, Hanover Medical University, Hanover, GERMANY. RATIONALE: The increase of atopic dermatitis prevalence has been associated with a decrease of “healthy” microbials such as lactobacilli in the intestinal flora. Some studies suggested that supplementation of food with lactobacilli may improve atopic dermatitis in children. This study was designed to investigate the therapeutic effect of Lactobacillus rhamnosus GG (LGG) in infants suffering from atopic dermatitis. METHODS: Infants aged 3-12 months suffering from mild to moderate atopic dermatitis (SCORAD index of 15-40) without current antiinflammatory treatment were randomised to receive LGG (5x109 CFU BID) or placebo as a food supplement for 12 weeks. SCORAD index and use of hydrocortisone 1% ointment as rescue medication (2 points per application) were recorded at 4, 8, and 12 weeks of treatment and combined as symptom load (SL). RESULTS: 54 infants (LGG group, mean± SD SCORAD index 24.6± 8.8) and 48 infants (placebo group, SCORAD index 23.6± 7.8) were randomised and completed the treatment period (intention to treat analysis). SL generally improved over time at 4 weeks (LGG vs placebo, 23.8± 12.4 vs 20.6± 9.9), 8 weeks (22.5± 14.6 vs 17.9± 13.1), and 12 weeks (19.6± 15.4 vs 15.1± 12.1), without statistical significant group differences. No significant group differences were found for use of rescue medication (0.8g± 45.0g vs 3.5g ± 29.8g), increase in median total serum IgE (0.17± 0.30kU/L vs 0.26± 0.45kU/L), and new developed allergic sensitization against hen’s egg or cow’s milk (18.8% vs 10.0%). CONCLUSIONS: This randomised controlled trial showed no therapeutic effect of LGG on mild to moderate atopic dermatitis in infancy. Funding: Infectopharm Arzneimittel, Heppenheim, Germany
926
MONDAY
An Autoimmune Blistering Skin Disorder: Paraneoplastic Pemphigus (PNP) In Association With A Breast Neoplasm P. Buddiga; Baz Allergy, Asthma & Sinus Center, Fresno, CA. RATIONALE: Paraneoplastic pemphigus (PNP) is a potentially rare complication usually associated with a neoplasm and is characterized by intractable ulcerative stomatitis, mucosal surface erosions and polymorphic cutaneous eruptions. METHODS/CASE: A 50-year-old African-American female with invasive breast carcinoma and supraclavicular lymph node metastasis was treated with a chemotherapeutic regimen of Cyclophosphamide and Doxorubicin. During her multiple cycle course she developed severe oral mucositis leading to the discontinuation of chemotherapy. The oral lesions progressed and she developed a diffuse polymorphic cutaneous eruption represented by erythematous papules, bullae and erosions that responded to oral corticosteroid treatment with Prednisone 60 mg/day for 2 weeks and tapered down to 20 mg everyday. Oral mucosal and skin lesion biopsies were performed and interpreted as consistent with PNP. RESULTS: Surgical Pathology Skin and Anterior Palate Biopsy-Direct Immunofluorescence IgG - 4+ linear intraepidermal intercellular deposit, complete epidermis IgA, IgM, C1q - negative C3D - 4+ linear epidermal basement membrane deposit, 3+ linear intraepidermal intercellular deposit , bottom one-fourth of epidermis. Breast biopsy- Invasive mammary carcinoma, poorly differentiated with focal mucinous features. CONCLUSIONS: Despite initial short-term success of aggressive immunosuppressant therapy for controlling the polymorphic cutaneous eruptions of PNP, the relative poor prognosis and high mortality rate in those with malignant tumors (10 % - 2 year survival) suggests failure of inducing a remission, despite persistent use of various immunomodulating agents. This demonstrates the need for further studies to elucidate the disease pathogenesis by which neoplasia induces autoimmunity so that this may facilitate the arrival of an immunotherapeutic efficacious intervention.
923
J ALLERGY CLIN IMMUNOL VOLUME 117, NUMBER 2
Induction of a Pathergy-Like Reaction Following Prick Skin Testing T. L. Johnson, II1, P. McCleskey2, M. Rathkopf1, J. Meffert2, L. L. Hagan1; 1Allergy/Immunology, Wilford Hall Medical Center, Lack land AFB, TX, 2Dermatology, Wilford Hall Medical Center, Lackland AFB, TX. RATIONALE: A 46-year-old female who received 6 months of immunotherapy for perennial and seasonal allergic rhinitis 5 years prior to presentation returned for consideration of restarting immunotherapy secondary to uncontrolled rhinoconjunctivitis. The patient had no other chronic illnesses and lacked symptoms suggestive for Behcet’s disease. Past history was negative for local or systemic reactions to previous skin testing and immunotherapy injections. METHODS: Prick skin testing was performed utilizing the Quintest® device to an aeroallergen panel consisting of 53 antigens to assess for the development of new sensitizations. RESULTS: The patient had positive results to multiple trees, weeds, molds, grasses, dust mite and cat. Five days after the testing procedure the patient called and stated that she developed pustules and pruritus at many of the skin test sites. The pustules were first noted on the day following testing. Upon examination the patient had round erythematous patches with central hyperpigmentation along with erosions and crusts. Lesions were present at 37 of 40 sites that had a 1+ or greater reaction (including histamine control). Out of 15 test sites (including negative control) that lacked reactivity at the time of initial reading; 2 had the development of the above lesions. Histopathologic examination revealed confluent epidermal necrosis, extensive interface vacuolar changes, and focal subepidermal vesicle formation. Dermal changes included a perivascular lymphocytic inflammatory infiltrate with endothelial swelling and few eosinophils or neutrophils. CONCLUSIONS: This report may represent a previously uncharacterized pathergy-like reaction induced by skin testing in a patient that to date lacks symptoms of known pathergy associated illnesses.
924
Profiles and Prognosis of Severe Infantile Atopic Dermatitis with Food Allergy M. Iguchi1, M. Ebisawa2, H. Tachimoto2; 1Jikei University school of Medicine, Tokyo, JAPAN, 218-1 Sakuradai, Department of pediatrics, National Organization Sagamihra National Hospital, Sagamihara city, Kanagawa, JAPAN. RATIONALE: The aim of this study was to clarify the profiles and prognosis of severe infantile atopic dermatitis (AD) (n=67) with food allergy (FA) against more than 2 food antigens. METHODS: All subjects were admitted to the Sagamihara National Hospital to control AD symptoms and to receive diagnosis of FA from 1997 to 2003. We analyzed their patients’ profiles and the prognosis of atopic condition for 5 years. RESULTS: 97% of their chronic eczema started by 4 months old. The number of food antigens determined by food provocation was decreased by 1.1 antigens (from 4.6 to 3.5) compared with the number of sensitized food antigens proved by specific IgE or SPT. Hen’s egg was the most common, followed by cow’s milk, wheat, and soybean. The number of food antigens had become less and less, finally it reached 1.4 items at the age of five. The release of egg elimination at the age of five was still only 30%. In addition the age when the release rate of cow’s milk elimination reached 50% was at 5 y, however, those for wheat and soybean were at 2 y. Although most of the subjects used steroid ointment regularly at the time of discharge, the percentage of “regular use” at 5 years old decreased by 10%. Incidence of bronchial asthma from the subjects at the age of four reached 53%. CONCLUSIONS: Their prognosis was relatively fair except for the prolonged egg and cow’s milk FA and the high incidence of BA from the subjects. Funding: Ministry of Health, Labor, and Welfare
925
Randomized, Placebo-Controlled Trial of Lactobacillus rhamnosus GG as Treatment of Mild to Moderate Atopic Dermatitis in Infancy C. Grüber1, M. Wendt2, S. Lau1, M. Kulig1, U. Wahn1, T. Werfel3, B. Niggemann1; 1Dept. of Pediatric Pneumology/Immunology, Charité Universitätsmedizin Berlin, Berlin, GERMANY, 2Department of Dermatology and Allergology, Hanover Medical University, Hanover, GERMANY, 3(2) Department of Dermatology and Allergology, Hanover Medical University, Hanover, GERMANY. RATIONALE: The increase of atopic dermatitis prevalence has been associated with a decrease of “healthy” microbials such as lactobacilli in the intestinal flora. Some studies suggested that supplementation of food with lactobacilli may improve atopic dermatitis in children. This study was designed to investigate the therapeutic effect of Lactobacillus rhamnosus GG (LGG) in infants suffering from atopic dermatitis. METHODS: Infants aged 3-12 months suffering from mild to moderate atopic dermatitis (SCORAD index of 15-40) without current antiinflammatory treatment were randomised to receive LGG (5x109 CFU BID) or placebo as a food supplement for 12 weeks. SCORAD index and use of hydrocortisone 1% ointment as rescue medication (2 points per application) were recorded at 4, 8, and 12 weeks of treatment and combined as symptom load (SL). RESULTS: 54 infants (LGG group, mean± SD SCORAD index 24.6± 8.8) and 48 infants (placebo group, SCORAD index 23.6± 7.8) were randomised and completed the treatment period (intention to treat analysis). SL generally improved over time at 4 weeks (LGG vs placebo, 23.8± 12.4 vs 20.6± 9.9), 8 weeks (22.5± 14.6 vs 17.9± 13.1), and 12 weeks (19.6± 15.4 vs 15.1± 12.1), without statistical significant group differences. No significant group differences were found for use of rescue medication (0.8g± 45.0g vs 3.5g ± 29.8g), increase in median total serum IgE (0.17± 0.30kU/L vs 0.26± 0.45kU/L), and new developed allergic sensitization against hen’s egg or cow’s milk (18.8% vs 10.0%). CONCLUSIONS: This randomised controlled trial showed no therapeutic effect of LGG on mild to moderate atopic dermatitis in infancy. Funding: Infectopharm Arzneimittel, Heppenheim, Germany
926
MONDAY
An Autoimmune Blistering Skin Disorder: Paraneoplastic Pemphigus (PNP) In Association With A Breast Neoplasm P. Buddiga; Baz Allergy, Asthma & Sinus Center, Fresno, CA. RATIONALE: Paraneoplastic pemphigus (PNP) is a potentially rare complication usually associated with a neoplasm and is characterized by intractable ulcerative stomatitis, mucosal surface erosions and polymorphic cutaneous eruptions. METHODS/CASE: A 50-year-old African-American female with invasive breast carcinoma and supraclavicular lymph node metastasis was treated with a chemotherapeutic regimen of Cyclophosphamide and Doxorubicin. During her multiple cycle course she developed severe oral mucositis leading to the discontinuation of chemotherapy. The oral lesions progressed and she developed a diffuse polymorphic cutaneous eruption represented by erythematous papules, bullae and erosions that responded to oral corticosteroid treatment with Prednisone 60 mg/day for 2 weeks and tapered down to 20 mg everyday. Oral mucosal and skin lesion biopsies were performed and interpreted as consistent with PNP. RESULTS: Surgical Pathology Skin and Anterior Palate Biopsy-Direct Immunofluorescence IgG - 4+ linear intraepidermal intercellular deposit, complete epidermis IgA, IgM, C1q - negative C3D - 4+ linear epidermal basement membrane deposit, 3+ linear intraepidermal intercellular deposit , bottom one-fourth of epidermis. Breast biopsy- Invasive mammary carcinoma, poorly differentiated with focal mucinous features. CONCLUSIONS: Despite initial short-term success of aggressive immunosuppressant therapy for controlling the polymorphic cutaneous eruptions of PNP, the relative poor prognosis and high mortality rate in those with malignant tumors (10 % - 2 year survival) suggests failure of inducing a remission, despite persistent use of various immunomodulating agents. This demonstrates the need for further studies to elucidate the disease pathogenesis by which neoplasia induces autoimmunity so that this may facilitate the arrival of an immunotherapeutic efficacious intervention.
923