Increase of cerebrospinal fluid soluble interleukin‐2 receptor index in a patient with atopy and syringohydromyelia

Abstract

Key wordsatopic dermatitis, atopic myelitis, syringohydromyelia, Chiari malformation type I, soluble interleukin-2 receptor.Atopic myelitis, described by in 1997, comprises atopic dermatitis, hyper-immunoglobulin (IgE)-emia and spinal involvement. The etiology, preventive methods and treatments were not established. Herein, we present a patient with severe atopic dermatitis, hyper-IgE-emia, Chiari malformation type I, and syringohydromyelia. Syringohydromyelia is sometimes complicated with Chiari malformation. However, the patient's cerebrospinal fluid (CSF) soluble interleukin-2 receptor (sIL-2R) index increased. In our data and former reports, 4,5 CSF sIL-2R increased in patients with multiple sclerosis, but did not in other patients with syringohydromyelia or atopic dermatitis. After treatment with skin care, food elimination and environment upgrading for 2 months, the CSF sIL-2R index decreased to undetectable levels. Neurological improvement was slight but certain. The patient then had foramen magnum decompression and a C1 laminectomy. At 2 months after the operation, his syringohydromyelia was still observed and muscle weakness was gradually improving.Syringohydromyelia is sometimes complicated with Chiari malformation. However, the patient's CSF sIL-2R index is hard to explain with the constriction of the central canal. The findings and data suggested one possibility, that atopic inflammation exacerbated syringohydromyelia. Case reportA 12-year-old male patient had suffered from severe generalized atopic dermatitis and allergic rhinitis since infancy. His psychomotor development was normal until 6 months ago when he began to show insidiously progressive muscle weakness and atrophy in the distal parts of the bilateral forearms, with right side dominancy (Fig. 1a,b). Asymmetrical and reduced grasp powers were noted; 2 kg and 7 kg for the right and left, respectively. Bilateral hyperalgesia and thermal-hypoesthesia were also observed at lesion of C5-C8. Bilateral deep tendon reflex, biceps, triceps, brachioradialis, patellar and Achilles, were decreased (left) or not noted (right). His intelligence quotient (IQ) was close to the normal range; Weschsler Intelligence Scale for Children (WISC) III performance IQ = 94, verbal IQ = 103 and total IQ = 99.A peripheral blood examination yielded a leukocyte count of 5900/mm 3 , eosinophils 24.9%. The routine serum biochemical laboratory datum were all within normal ranges. The serum IgE level was 11 400 IU/mL (<200). Serum IgE specific for Dermatophagoides farinae , Dermatophagoides pteronyssinus , egg white, milk, soybean, wheat, rice, pork, candida and Staphylococcus aureus toxin were strongly positive. Peripheral B cells increased to 18% (4-13%). Populations of T cell, monocyte, natural killer cell, and CD4/8 ratio were all within the normal range. T-cell proliferation to phytohemagglutinin as measured by [ 3 H]-thymidine incorporation resulted in 59 770 cpm (26 000-53 000) with a stimulation index of 424, and in response to concanavalin-A gave 37 083 cpm (20 000-...

Human γδ T cells modulate the mite allergen‐specific T‐helper type 2‐skewed immunity

“…In spite of many reports on allergic diseases and their therapeutic strategies, it still remains difficult to control such diseases [1,3,23]. Therefore, various kinds of immunotherapy have been conducted [21,24].…”