CASE REPORT

Spontaneous Remission of Hypertrophic Lymphocytic Gastritis Associated with Hypoproteinemia Masaki Amenomori, Tomio Umemoto*, Ryoji Kushima** and Takanori Hattori* *

A 54-year-old womancame to our office because ofpretibial edema. She had no gastrointestinal symptoms. Laboratory tests revealed severe hypoproteinemia. Upper gastrointestinal endoscopy demonstrated enlarged gastric folds and multiple aphthoid nodules on the body and the fornix of the stomach. The biopsy specimen revealed a large numberof CD8positive intraepithelial T- lymphocytes infiltrating the gastric mucosa. Both serum total protein and the gastric lesions improved eight months after her first visit without any therapy for peptic ulcer or eradication of Helicobacter pylori The data suggest that spontaneous remission mayoccur in lymphocytic gastritis without any gastrointestinal symptoms. (Internal Medicine 37: 1019-1022, 1998) Key words: cytotoxic T-lymphocyte, Helicobacterpylori

Introduction (Fig. 1A, B). This pattern is consistent with the diagnosis of varioliform gastritis. Lymphocytic gastritis (LG) is a rare disorder characterized The stomach biopsy specimen revealed the presence of a by diffuse infiltration in the surface and foveolar epithelium of large numberof lymphocytes infiltrating the surface, foveolar gastric mucosa by T lymphocytes (1). LG correlates with epithelium, and foveolar hyperplasia of gastric mucosa (Fig. diffuse varioliform gastritis characterized by widespread mu- 2A, B). The number oflymphocytes per 100 epithelial cells was cosal nodules, erosions, and thickened rugal folds. The more than 50. Immunological staining showed these pathogenesis and treatment of LG, however, remains unclear. intraepithelial lymphocytes to be CD8positive cytotoxic T- Wereport a case of LGwith severe hypoproteinemia which lymphocytes. H. pylori were seen in the body and astral mucus. remitted without therapy for peptic ulcer or eradication of Thepathological appearance was pathognomonicof LG. Helicobacter pylori (H. pylori). Twoweeks after taking furosemide and spironolactone her edema disappeared. Although furosemide and spironolactone were stopped, the level of serum total protein gradually im- Case Report proved and was normalized six months after her first visit. Eight A 54-year-old womancame to our office after experiencing months after her first visit endoscopic examination revealed pretibial edema for two weeks. She had gained 5 kg in weight normal gastric folds (Fig. 1C, D). The second biopsy specimen over the previous two months, but denied any abdominal of the body showed a few intraepithelial lymphocytes and no symptoms.Her appetite was good. Onexamination there were foveolar hyperplasia (Fig. 2C, D). The number of //, pylori in no remarkable findings except for the presence of symmetrical the body and antral mucus, however, was more than in the first lower limb edema. Laboratory data (Table 1) revealed serum biopsy specimen. The patient remains asymptomatic after 15 total protein 4.4 g/dl, albumin 2.2 g/dl, calcium 8.3 mg/dl, months. Shehad taken neither antibiotics nor other drugs except immunoglobulin G (IgG) 601 mg/dl, immunoglobulin A (IgA) for furosemide and spironolactone since her first visit. 76 mg/dl. Liver function tests and renal function tests were within the normal range. The urine contained no protein. Discussion Upper gastrointestinal endoscopy showed thickened longi- tudinal rugal folds, confined on the gastric body and fornix. Here, we describe a patient with hypoproteinemia and the Gastric folds were topped by small rounded, aphthoid nodules specific endoscopic and histological findings similar to LG(1).

From Ryuo Medical Center Yuge Clinic, Shiga, *Ryuo Medical Center, Shiga and **the First Department of Pathology, Shiga University of Medical Science, Otsu Received for publication March 16, 1998; Accepted for publication August 29, 1998 Reprint requests should be addressed to Dr. Masaki Amenomori, Ryuo Medical Center Yuge Clinic, 1825 Yuge, Ryuocho, Gamogun, Shiga 520-2501

Internal Medicine Vol. 37, No. 12 (December 1998) 1019 Amenomoriet al

Table 1. Laboratory Data on Her First Visit U rine C hem istry P ro tein (- ) T otal pro tein 4.4 g/dl G lu co se (- ) A lb um in >.6 g/dl Occult blood (- ) Urea nitrogen 4.3 mg/dl H em ato logy C reatin in e 0.8 mg/dl White blood cell 4,500/ul N a 144 mEq// Red blood cell 4 5 3x lO 4/u l K 3.9 mEq// H em o glob in 13.8 g/dl c ¥ 107 mEq// Platel et 1 9.2x 104/jlil C a 8.3 mg/dl S ero log y G luco se 97 mg/dl Im mu n og lo b ul m G 601 mg/dl Aspartate aminotransferase 22 IU/Z Imm unog lob ulin A 76 mg/dl Alanine aminotransferase 17 IU // Imm uno gl obu li n M 188 mg/dl Lactate dehydrogenase 444 IU/Z C 3 65 mg/dl Alkaline phosphatase 117 IU/Z C 4 2 1 m g/d l Uric acid 4.3 mg/dl C H 5 0 45 U /ml T o tal cho le stero l 187 mg/dl Anti-nuclear antibody (- ) T riglyceride 160 mg/dl

LG is characterized by the presence of a large number of pylori, suggesting that H. pylori maynot be relevant to her lymphocytes infiltrating the surface and foveolar gastric epithe- clinical course. lium. Our patient fulfilled the diagnostic criteria for LGde- With regard to the treatment of LG,therapy for peptic ulcer scribed as greater than 30 intraepithelial lymphocytes per 100 such as H2-receptor antagonists (2, 12), a proton pumpinhibitor epithelial cells (1). Our patient had hypoproteinemia without (2), and the eradication of//, pylori (5, 6) were reported to be gastrointestinal symptom. The commonpresenting symptoms effective. Although spontaneous remissions were reported, of LGhave been reported as epigastric pain, nausea, anorexia, clinical data in these cases was insufficient (2). Crampton et al and weight loss (2). Lack ofgastrointestinal symptoms (3), and reported a case of LGwith hypoproteinemia controlled by lower limb edema ascribed to hypoproteinemia have been diuretics similar to the present case (3). Their case also demon- reported as rare symptoms(2-6). Haot et a] reported only 4 strated a lack of gastrointestinal symptoms as did ours. This patients with hypoproteinemia out of 160 LGpatients (4). suggests that spontaneous remission mayoccur in LGwithout Hypoproteinemia in LGis due to protein-losing gastropathy any gastrointestinal symptoms.Accumulationof similar cases (2-6), which is derived from elevated gastric permeability (7). mayhelp to establish the etiology and appropriate treatment of Wecould not performexaminationsto confirmthe excessive LG. protein leakage into the gastrointestinal lumen, such as Alphal- antitrypsin clearance. Our patient, however, is thought to have protein-losing gastropathy because she had no evidence of decreasing protein production or leaking protein except into the gastrointestinal tract. The etiology and pathogenesis ofLG remains unknown. The relationship between LG and H. pylori has been debated (8). The resolution of the clinical, endoscopic, and histological abnormalities with eradication of H. pylori has been reported (2, 5, 6). LG patients have been reported to have U high prevalence of specific antibody to H. pylori (9, 10). Nkniela et al reported that the appearance of gastric mucosa in LGpatients whohad specific antibody to H. pylori was associated With an increase in the grade of gastritis and intestinal metaplasia during a ten-year interval (10). Onthe other hand, the prevalence ofH. pylori in LGIias been reported to be lower than that in chronic active gastritis (9). From long-term follow-up study, the clinical symptom^, endo- scopic, and histological abnormalities remained unchanged and conventional peptic ulcertherapy failed (1 1). In ourpatient, the serum total protein, endoscopic, and histologic abnormali- ties improved spontaneously in spite of the existence of H.

1020 Internal Medicine Vol 37, No 12 (December 1998) Hypertrophic Lymphocytic Gastritis A B D c

Figure 1. Endoscopic view. Thickened longitudinal rugal folds A) with small rounded, aphthoid nodules B) on the gastric body and fornix on her first visit improved 8 months after her first visit C, D).

Internal Medicine Vol. 37, No. 12 (December 1998) 1021 Amenomoriet al B A D c

Figure 2. HE-stained sections of the stomach. A, B) On her first visit: The infiltration of lymphocytes and the foveolar hyperplasia were observed A) (x40). Numerous small lymphocytes surrounded by a clear halo were observed B) (x200). C, D) 8 months after her first visit: Foveolar hyperplasia improved C) (x40). Few small lymphocytes were observed D) (x200).

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