Malakoplakia in a healthy young female patient
A 19-year-old female was referred to our hospital following the discovery of multiple polyps in the rectum with sigmoidoscopy. Her clinical symptoms were recurrent low abdominal pain and loose stool with blood and foul odor 4 to 5 times a day for 3 months. She complained of a squeezing type abdominal pain relieved after defecation. When the patient was admitted, she was in relatively good condition with no specific past medical history and did not complain of fever, chill, anorexia, weight loss, or cold sweating.\nOn physical examination, there were no definite abnormal findings except for mild epigastric tenderness and slightly increased bowel sounds. Blood pressure was 110/76 mmHg, pulse rate 106 beats per minute, body temperature 36.3℃. Laboratory tests revealed hemoglobin at 9.0 g/dL, white blood cell (WBC) count at 9,050/µL with 26.6% neutrophils, 27.2% lymphocytes, 15.2% monocytes, and 0.8% eosinophils. A peripheral blood smear showed normocytic normochromic anemia, Fe of 18 µg/dL, total iron binding capacity of 286 µg/dL, 0.8% reticulocytes, and stool occult blood over 1,000 ng/mL. Inflammatory markers such as C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR) were mildly elevated to 3.03 mg/dL and 42 mm/hr, respectively. Urine analysis with microscopic exam showed no abnormality. Stool acid-fast bacillus and tuberculosis polymerase chain reaction were negative, and gram stain showed WBC 10 to 25 and a number of gram-positive cocci and gram-negative bacilli. In the stool culture, Salmonella or Shigella was not isolated and blood cultures were negative. Rheumatic factor was normal, and antineutrophil cytoplasmic antibody was negative. Serum IgG, IgE, and hemolytic complement 50 (CH50) complement levels were slightly elevated to 1,800 mg/dL, 236 IU, and 54.2 U/mL, respectively. IgA, IgM, complement 3, and complement 4 were within normal range.\nColonoscopy revealed very diverse circular or flat elevated mucosal lesions up to 10 cm in size from the cecum to the descending colon, small nodules, mass-like lesions, and erosive small polypoid lesions throughout the colon, particularly in the sigmoid colon (). Biopsies were taken in eight different places. After colonoscopy, we initially suspected atypical lymphoma or malignancy. However, lactate dehydrogenase and tumor markers such as carcinoembryonic antigen, CA 125, CA 19-9, and α-fetoprotein were within normal range and an abdominopelvic computed tomography scan revealed no other abnormal findings, except for localized multiple small reactive lymph node enlargements along the mesentery. Histological examination of colon lesion biopsies revealed dense histiocyte infiltration with round inclusions of varying sizes in the cytoplasm. Many of these inclusions had round laminated structures with a central dark area and peripheral pale halo. These structures were stained with periodic acid-Schiff (PAS), Alcian blue, Prussian blue, and Gomori methenamine silver stains to confirm the presence of Michaelis-Gutmann bodies with pathognomonic features of malakoplakia (). These diagnostic tests confirmed the diagnosis of malakoplakia. During antibiotic treatment, diarrhea and abdominal pain improved so she was discharged with instructions to take 500 mg ciprofloxacin twice a day, 25 mg bethanecol three times a day, and 60 mg ascorbic acid once a day by mouth. After the treatment started, the symptoms of abdominal pain and diarrhea disappeared in a week. After 2 months, CRP, ESR, and hemoglobin were nearly normal with values of 0.32 mg/dL, 2 mm/hr, and 12.5 g/dL, respectively. The patient remained on this regimen for 6 months. During the treatment period, no drug side effects were observed. Previous abnormal colonoscopy findings were remarkably improved after 6 months of follow-up, and returned to normal after the end of 12 months (). Outpatient follow-up visits have confirmed that she is currently healthy.