Brown Bowel Syndrome Secondary to Jejunoileal Bypass: The First Case Report
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- Lee, H., Carlin, A.M., Ormsby, A.H. et al. OBES SURG (2009) 19: 1176. doi:10.1007/s11695-009-9872-0
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A 58-year-old woman with a surgical history of jejunoileal bypass in 1980 for weight reduction sought medical attention with multiple complaints. The patient had not been taking any nutritional supplements since her bypass surgery, 26 years previously. She was found to have osteomalacia, chronic diarrhea, secondary hyperparathyroidism, and hyperoxaluria with a frequent history of nephrolithiasis. Because of her severe osteodystrophy and metabolic complications, reversal of her jejunoileal bypass was recommended. Reversal of the jejunoileal bypass with a sleeve gastrectomy was performed. Laparotomy revealed brown discoloration of the entire alimentary limb with atrophy of the bypassed intestinal limb. Histologic examination of the resected small bowel demonstrated brown pigment deposits within smooth muscle cells of the bowel wall. The pigment stained positive with Fontana-Masson most likely representing lipofuscin. We report a case of brown bowel syndrome complicating jejunoileal bypass, the first case reported in the literature to the best of our knowledge.
KeywordsBariatric surgeryMalnutritionJejunoileal bypassBrown bowel syndromeLipofuscin
Nutritional deficiency is a well-known complication of bariatric surgery. Deficiencies of protein, iron, vitamin B12, folate, calcium, vitamin D, thiamine, and other fat-soluble vitamins and minerals have been reported following bariatric surgery [1–3]. Patients undergoing malabsorptive operations are more prone to develop nutritional deficiencies than those with restrictive procedures, and the degree of deficiency is proportional to the length of the malabsorptive area and to the percentage of weight loss .
Brown bowel syndrome is a rare condition characterized by deposition of lipofuscin pigment in smooth muscle cells. Brown bowel syndrome has been described in the setting of states of severe malnutrition such as celiac disease and endemic sprue [1, 2]. Although the pathogenesis is not fully understood, vitamin E deficiency has been considered to be a putative cause of brown bowel syndrome [4–7].
It seems intuitive to connect malnutrition secondary to jejunoileal bypass surgery and brown bowel syndrome resulting from nutritional deficiency. Nevertheless, we were not able to find a report of brown bowel syndrome following jejunoileal bypass. Herein, we report a case in which a patient had undergone jejunoileal bypass complicated by malnutrition and brown bowel syndrome.
A 58-year-old Caucasian woman (weight 77 kg, height 154.4 cm, BMI 32.2 kg/m2) was referred by her rheumatologist for evaluation of osteoporosis, vitamin D deficiency, nonspecific aches and pains, chronic diarrhea, and a skin rash of the intertriginous zones beneath her breasts. The patient had undergone jejunoileal bypass for weight reduction at the age of 32 (26 years previously), and since then had not been taking any nutritional supplements. Details regarding the primary operation and her preoperative nutritional status were not available as the surgeon who performed the bypass surgery had retired and the operative report and the surgeon were unable to be located. Postoperatively, she suffered from premature ovarian failure at the age of 33, multiple bouts of nephrolithiasis, osteoporosis, and severe iron deficiency anemia requiring transfusions and iron injections. Laboratory workup revealed low serum calcium [7.4 mg/dL (8.2–10.2)], vitamin D deficiency [25-hydroxy vitamin D <7 ng/mL (15–80)], secondary hyperparathyroidism [intact parathyroid hormone 205.6 pg/mL (10–75)], a mild decrease in her albumin of 3.6 g/dL (3.7–4.8), and hyperoxaluria. Transiliac bone biopsy revealed severe osteomalacia. Colonoscopy did not reveal any mucosal abnormalities. Despite supplemental treatment with large doses of calcium and vitamin D given orally, her symptoms did not improve. Because of her severe osteodystrophy and metabolic complications secondary to malnutrition, reversal of jejunoileal bypass was recommended.
The postoperative course of the patient was complicated by acute pulmonary edema, atrial fibrillation, urinary tract infection, and a wound hematoma that were managed accordingly. Subsequent upper gastrointestinal contrast study did not show any evidence of leakage. She was prescribed multivitamins and protein supplements and was last seen without major complication 7 months after the surgery.
Since the first end-to-end jejunoileal bypass operation by Kremen et al. in 1954 , technical aspects of bariatric surgery have markedly progressed especially over the last two decades . Although complication rates are relatively low with major complications in approximately 10% of the procedures , more cases of unusual complications are being documented due to the exponential growth in the number of patients undergoing bariatric operations with long-term follow-up [11–14].
Jejunoileal bypass was abandoned due to its severe complications including malnutrition, nephrolithiasis, cryptogenic cirrhosis, cholelithiasis, and chronic diarrhea . Malabsorption of vitamins and nutrients frequently manifests as clinical malnutrition. In addition, peripheral neuropathy, chronic infections of the bypassed limb, and migratory polyarthritis have been reported to be associated with jejunoileal bypass [10, 15, 16]. Another malabsorptive procedure, biliopancreatic diversion with duodenal switch, has replaced jejunoileal bypass to reduce complications; however, protein–calorie malnutrition remains a problematic complication of malabsorptive procedures .
Brown bowel syndrome is a rare condition associated with severe malnutrition, especially vitamin E deficiency . In the absence of vitamin E, working as an antioxidant, the phospholipid layer of the mitochondrial membrane is subject to injury by free oxygen radicals generated by oxidative phosphorylation, resulting in an aggregate of degraded mitochondrial membrane, in the form of lipofuscin. Loss of mitochondrial function and lack of energy lead to atrophy and atony of the smooth muscle layer [4, 18]. As such, brown bowel syndrome has been found in association with toxic colonic dilation, intestinal pseudo-obstruction, intussusception, and ileus [3, 4]. Also, a case of brown bowel syndrome associated with massive lower gastrointestinal bleeding has been reported . These authors postulated that mitochondrial malfunction in smooth muscle cells of the vasculature leads to bleeding. Moreover, three cases of gastrointestinal adenocarcinoma have been reported in association with brown bowel syndrome [19, 20]. Reynaert et al. speculated that vitamin E deficiency may be responsible for both brown bowel syndrome and the formation of dietary carcinogens . Because of the syndrome’s association with severe malnutrition and serious gastrointestinal complications, we would like to call attention to this unusual association.
In addition to assessing nutritional status before and after any bariatric procedure, it is imperative to rigorously apply recommendations and guidelines regarding prevention and management of malnutrition. This requires thorough patient education, patient compliance and physician supervision.
This report was possible due to the unusually long follow-up. Longer follow-up will likely lead to the identification of more nutritional complications related to previous bariatric operations. Therefore, brown bowel syndrome should be considered as one of the possible conditions that may lead to serious complications following jejunoilieal bypass. Again, emphasis should be placed on the importance of nutritional management after bariatric surgery to avoid this rare, but preventable condition.
The authors have no commercial associations that might pose a conflict of interest. All authors have made substantive contributions to the study and endorsed the data and conclusions.