ABSTRACT
Five children with ulcerative colitis for whom surgery was recommended were treated with cyclosporine. The five had received corticosteroids for 1-24 months. The group included two patients with acute-onset ulcerative colitis and three with acute exacerbations of intractable corticosteroid-dependent chronic ulcerative colitis. The average age at initiation of cyclosporine therapy was 13.8 years (range, 11.5-16); all five patients were boys. Cyclosporine was initiated in the hospital by continuous i.v. infusion. Trough levels of 400-600 ng/dl (measured by radioimmunoassay) were achieved, at which point oral cyclosporine was given and oral dosage was adjusted to similar levels. Significant hypertension requiring medical attention was seen in one patient. Of the two recently diagnosed acute cases, one failed to respond and required subtotal colectomy after 2 weeks of treatment, and the other, despite an initial response, had a subtotal colectomy 10 months later. Of the three corticosteroid-dependent children, none was able to be weaned from corticosteroids and all underwent subtotal colectomy. Our experience emphasizes that the appropriate role of cyclosporine as therapy for children with ulcerative colitis is yet to be determined. Cyclosporine was not effective as an alternative to surgery in our patients.
Subject(s)
Colitis, Ulcerative/drug therapy , Cyclosporine/therapeutic use , Acute Disease , Adolescent , Adrenal Cortex Hormones/therapeutic use , Child , Humans , Male , Prednisone/therapeutic use , Treatment FailureSubject(s)
Cyclosporine/therapeutic use , Gastrointestinal Diseases/chemically induced , Immunosuppressive Agents/antagonists & inhibitors , Liver Transplantation/immunology , Tacrolimus/toxicity , Adult , Aged , Female , Graft Rejection/prevention & control , Humans , Tacrolimus/antagonists & inhibitorsSubject(s)
Liver Transplantation/immunology , Tacrolimus/therapeutic use , Adolescent , Bilirubin/blood , Child , Child, Preschool , Cyclosporine/adverse effects , Female , Graft Survival , Humans , Hyperkalemia/etiology , Immunosuppression Therapy/adverse effects , Immunosuppression Therapy/methods , Infant , Infections/etiology , Liver Transplantation/physiology , Lymphoproliferative Disorders/etiology , Male , Safety , Tacrolimus/adverse effectsABSTRACT
Microvillous inclusion disease is a specific disorder recognized as a cause of intractable diarrhea of infancy. We studied three cases by light microscopy, electron microscopy, and immunostaining for polyclonal carcinoembryonic antigen (CEA). Histologically, all cases had villous atrophy and abnormal accumulation of periodic acid-Schiff-positive material in surface enterocytes. Ultrastructurally, poorly developed brush-border and intracytoplasmic inclusions lined by intact microvilli were present in surface enterocytes. Crypt cells showed well-preserved surface microvilli. Carcinoembryonic antigen immunostaining showed prominent intracytoplasmic reactivity in surface enterocytes and linear brush-border reactivity in crypt cells. Normal and diseased small bowel biopsy specimens used as controls revealed linear brush-border reactivity without intracytoplasmic staining. Intracytoplasmic positivity for carcinoembryonic antigen in microvillous inclusion disease is explained by its presence in the glycocalyx within the microvillous inclusions. The demonstration of a distinct staining pattern for polyclonal carcinoembryonic antigen in routinely processed small bowel biopsy specimens provides a new useful criterion that complements other established techniques for accurate diagnosis of microvillous inclusion disease.
