Abstract
Treatment of Eosinophilic Gastroenteritis With Interferon li,JI Alpha-2b Richard E Crockett, Dennis K Ledford University of South Florida, Tampa, FL Interferon alpha (IFN~) has been used to successfully treat a variety of eosinophilic diseases including hypereosinophilic syndrome, eosinophilic leukemia and eosinophilic pustular folliculitis. This is the first published case report of successful treatment of eosinophilic gastroenteritis with IFNt~. The patient developed severe, sub-acute epigastric pain and persistent vomiting at age 15. The pain was worsened by eating any foods and was not relieved by ranitidine 300 mg twice daily or multiple over-the-counter antacid medications. Endoscopy revealed esophagitis, gastritis and ulceration of the pyloric channel and duodenum. A biopsy of the ulcer showed nonspecific inflammation, but subsequent biopsies one month later showed dense eosinophilic mucosal and submucosal infiltration of the esophagus, stomach and duodenum. Colonoscopy and terminal ileoscopy were normal. Ulcer biopsies and serum antibodies were negative for H. pylori. The patient was not taking NSAIDS. Gastrin levels were normal. Three stool samples showed no ova or parasites. The absolute eosinophil count ranged from 600-1200 cells/ktL and total IgE was 1012 IU/mL. In vitro specific IgE was positive to wheat, egg, milk and fish. Echocardiogram and bone marrow biopsy were normal. Intermittent abdominal pain persisted despite medical management with omeprazole, prednisone and elimination diet, and surgical treatment with partial gastrectomy and selective vagotomy. One year later, exclusive enteral feeding with an elemental formula (Neocate One Plus ®) was initiated through a gastrostomy tube. Abdominal pain and vomiting recurred with any oral ingestion of food or when prednisone was tapered below 20 mg daily. Eosinophil counts remained between 330670 cells/~L on prednisone therapy. Two years after onset of symptoms, an immunology consult was requested to suggest alternative therapies to minimize corticosteroid side effects. Subcutaneous IFNot-2b 500,000 units daily was initiated. Within 2 weeks, the eosinophil count decreased to 146 cells/gtL and symptoms improved. Prednisone dosage was tapered over 21⁄2 years, and a normal diet was gradually reintroduced, except for dairy and eggs. IFNt~ was tapered over 4 months after prednisone was discontinued. The patient has not taken prednisone for 9 months or IFNct for 5 months. He is tolerating a regular diet and has gained 25 pounds, without abdominal discomfort or diarrhea. Eosinophil counts without therapy are remaining normal. 1 ? Use of Interferon7 in a Patient With Severe Molluscum Conta/ giosum, Severe Atopic Dermatitis and Undefined ToCell Deftciency Fabienne Dayer Pastore*, Javier Chinen*, WT Shearer§, SE Pacheco§ *Baylor College of Medicine, Houston, TX §Baylor College of MedicineTexas Children's Hospital, Houston, TX INTRODUCTION: Immunodeficiencies like Omenn syndrome, hyper-IgE syndrome or HIV infection may manifest an imbalance of Thl/Th2 type immune phenotype. Skin conditions such as molluscum contagiosum (MC) or atopic dermatitis tAD) are manifestations of a Th2 imbalance. Among our population of immunodeficient patients in the Allergy/Immunology Service at Texas Children's Hospital, we have seen severe MC in patients with Leukocyte Adhesion Deficiency, hyper-IgE syndrome and twins with an ill-defined T-cell deficiency. MC infection is caused by a defective pox virus that produces IL18 binding proteins. IL18 is an important cytokine regulating interferon-y (IFN-y) production and therefore its binding leads to decreased levels of IFN-'/. RATIONALE: IFN-ytherapy has been described to be effective in AD through immunomodulation and counteraction of Th2 immune response. In the case of MC, decreased IFN-yproduction leads to a shift towards Th2 immunity. Therefore, we hypothesized that treatment with IFN7 would be beneficial in patients with AD and MC, through immunomodulation and counteraction of Th2 related cytokines. STUDY: A 9-year-old Latin-American female with severe MC, severe AD, undefined T-cell deficiency and asthma had been hospitalized several times per year, mainly for bacterial skin superinfections of AD and MC. Baseline laboratory studies demonstrated high eosinophil count and very high IgE level (see table). In November 2000, she was admitted for severe bacterial superinfection of MC lesions. Subcutaneous injection of IFN-y at 50 lag/m2/dose three times/week was initiated in addition to conventional antibiotic therapy and atopic dermatitis skin care. Ten months after initialization of IFN7 treatment the patient has had great improvement in AD symptoms and minor decrease in MC lesions. She has not required hospitalizations related to AD or MC. A decrease in eosinophil number and IgE level has been observed (see table). CONCLUSION: IFN-Ttherapy may have been beneficial in controlling AD and MC in this immunodeficient patient because of its immunomodulatory effect and counteraction of the Th2 immune response. (Supported by the David Fund of Texas Children's Hospital) Test IgE (0-90 IU/ml) Eosinophil count (cells/ram3) 11106100 41,66
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