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Dig Dis Sci. 2003 Aug;48(8):1600-6

Autoimmune enteropathy and colitis in an adult patient.

Carroccio A, Volta U, Di Prima L, Petrolini N, Florena AM, Averna MR,
Montalto G, Notarbartolo A.

The Department of Internal Medicine, University Hospital of Palermo,
Palermo, Italy. [log in to unmask]

The presence of circulating autoantibodies to gut enterocytes has been very
rarely described in adults and is considered a possible cause of refractory
sprue. Our aims was to describe the case of an adult patient with serum
anti-enterocyte autoantibodies associated with a clinical picture
characterized by involvement of both the small intestine and colon. A
female, age 50, had suffered from diarrhea with mucus and blood, abdominal
pain, thinness, anemia, and leukopenia since the age of 20. She also
suffered from HCV infection and had mild chronic hepatitis. Family history
was positive for autoimmunity. Symptoms were reported to worsen after
eating gluten-containing foods, but anti-transglutaminase and anti-
endomysial antibodies were negative. Intestinal histology showed mild,
patch villous atrophy with a high intraepithelial lymphocyte count, but a
normal number of intraepithelial lymphocytes carrying the gamma/delta+
receptor. HLA was: A11, A31 (19), B52 (5), DR 15 (2), DR 14 (6), DR 51, DR
52, DQ1. Colonoscopy did not show ulcerations or erosions and colon
histology showed a moderate inflammatory infiltrate without minor crypt
distortion or granuloma. RAST tests were positive for lactalbumin,
lactoglobulin, casein, egg, and gliadin. After commencement of an
oligoantigenic diet, stool frequency initially decreased, but the presence
of mucus in the stools persisted, with episodes of bloody diarrhea. After
one year of diet, nutritional parameters were low and anemia associated
with a low leukocyte count persisted. Upper and lower gastrointestinal
endoscopy and histology of the small intestine and colon were virtually
unchanged. Consequently, natural autoantibodies and enterocyte
autoantibodies were assayed. The patient was positive for IgG class
enterocyte autoantibodies at a titer of 1:34. No other organ-specific or
non-organ-specific autoantibodies were positive. Prednisolone treatment was
started and the symptoms improved. After one year of this treatment plus
elimination diet she was reevaluated. Bowel movement frequency was normal,
body weight increased, and the asthenia had completely regressed. IgG anti-
enterocyte autoantibodies were absent. Histology of the distal duodenum
showed a normal villus/crypt ratio and IEL infiltration was reduced. Colon
histology showed a reduction in inflammatory infiltrate in the lamina
propria. In conclusion, we report a case of generalized gut disorder in an
adult patient, affecting both the small intestine and the colon and
characterized by the presence of circulating anti-enterocyte
autoantibodies. Systematic testing for enteroctye autoantibodies should be
performed not only in patients with refractory sprue, but also in subjects
with upper and lower intestinal symptoms who have not been definitively
diagnosed.

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