Ağustos 2015

53-57

Burcu BOZTEPE ¹, Orhan SEZGİN ² , Zeynep Ebru ESER ² , Serkan YARAŞ ³

¹ Department of Internal Medicine, Keşan State Hospital, Edirne
² Department of Gastroenterology, Mersin University Medical School, Mersin
³ Department of Gastroenterology, Niğde State Hospital, Niğde

Elliüç yaşında erkek hasta kaslarda güçsüzlük ve hipokalemik paralizi ile acil servise başvurdu. Hastanın başvuru esnasında hiçbir gastrointestinal semptomu olmamasına rağmen, yapılan araştırmalar sonucunda Crohn hastalığı tanısı aldı. Bu olgu bildiğimiz kadarı ile gastrointestinal semptomu olmadan hipokalemik paralizi tanısı alan literatürdeki ilk olgudur. Crohn hastalığı nutrisyonel eksiklikler ve metabolik hastalık ile başvuran hastaların ayırıcı tanısında düşünülmelidir.

Crohn hastalığı, hipokalemi, paralizi

Acute hypokalemic paralysis is a rare condition that can be potentially life threatening but resolves with potassium infusion, if recognized early. We report the unusual case of hypokalemic and hypophosphatemic paralysis in a male patient with Crohn’s disease

The syndrome of hypokalemic and hypophosphoremic paralysis represents a heterogeneous group of disorders characterised clinically with acute systemic weakness and hypokalemia. Most cases are familial or primary hypokalemic periodic paralysis; sporadic cases are associated with other various conditions including barium poisoning, hyperthyroidism, renal disorders, certain endocrinopathies and gastrointestinal potassium losses (1). Gastrointestinal potassium loss is a rare cause, although reported in celiac disease (2), tropical sprue (3), short bowel syndrome (4), and acute gastroenteritis (5). Underlying causes are anorexia, active inflammation, bile salt and fat malabsorption, decreased of absorption surface, rapid gastrointestinal transit, lack of fat-soluble vitamins (6-7). There has been reported only one case with hypokalemic paralysis caused by intestinal Crohn’s disease in the literature (8). We know our patient is the first report of hypokalemic paralysis caused by involvement in upper gastrointestinal system and ileocolic Crohn‘s disease, as far. Interestingly, the patient had no gastrointestinal symptoms at presentation, despite evidence of malabsorption. Malnutrition is an important complication and frequently observed in patients with Crohn’s Disease (CD), usually associated with nutritional deficiencies, especially vitamins (both water- and fat-soluble) and essential trace elements. It is often due to the disease activity; poor oral intake or restrictive diets, bacterial overgrowth, bile salt malabsorption, extensive small-bowel disease, fructose malabsorption, inflammation, lactose malabsorption, multiple resections, short-bowel syndrome and surgery (9). Anemia and osteoporosis are the most frequent complications generated by nutritional deficiencies in inflammatory bowel diseases (IBD). Osteoporosis is caused by low intakes of calcium and vitaminD; anemia, may be associated with chronic iron loss, long lasting inflammation and nutritional deficiencies suh as folate and vitamin B 12 (10,13). The cause of IBD iron deficiency anemia (IDA) is multifactorial and due to a combination of iron deficiency and chronic gastrointestinal blood loss and, in some cases, self-imposed dietary restrictions. A study showed that vitamins of A, C, D, E, B12 , folate and level of calcium, iron and potassium were low in blood with Crohn’s disease (11). Gastrointestinal bleeding is the most common etiology for iron-deficiency anemia in IBD. Although patients with ileal CD may be at particular risk for vitamin B 12 deficiency, the unaffected small bowel may adapt and increase its ability to absorb vitamin B 12 efficiently. Distal ileal resection or active disease in this area, ie. the last 60 cm of ileum where B 12 is absorbed, resuled in B 12 deficiency, as well. Vitamin B 12 should be given parenterally, in such cases, due to lack of functional absorptive area in the terminal ileum. Beside these, the diarrhea, high-output stoma or fistulae, short-bowel syndrome, malabsorption and vomiting result in excessive amount electrolyte losses such as potassium, sodium, magnesium, selenium and zinc (12). However, hypophosphatemia may occur due to both vitamin D deficiency and decreased intestinal absorption. Hypophosphatemia is commonly missed due to nonspecific signs and symptoms, but it causes considerable morbidity and even mortality. Phosphate is abundantly present in many foods. Isolated dietary phosphate deficiency is uncommon, and the deficiency usually occurs with generalized malnutrition. Intestinal phosphate absorption is up-regulated by 1,25(OH) vitD3. Generalized muscle weakness is the most common symptom of hypophosphatemia; weakness and fatigue are related with acquired hypophosphatemia. Rhabdomyolysis is one of the most important clinical consequences in hypophosphatemia (14). Calorie-protein malnutrition may lead in the humoral and cellular immunodeficiency. Its effects on the intestine: decreasing the efficiency of the mucosal barrier, leading alteration in the mucosa-associated lymphoid tissue functions and finally a greater risk of infection by bacterial translocation. Moreover, hypoplasia of the intestinal villi perpetuates malabsorption and increases the risk of infections (15). Our case had paralysis due to hypokalemiae and hypophosphatemia associated with malabsorbtion. Patients with Crohn’s disease may present with the malabsorbtion symptoms, solely. Therefore, one must consider IBD, espcially Crohn disease, in a patient, even if with no gastrointestinal symptom.

The syndrome of hypokalemic and hypophosphoremic paralysis represents a heterogeneous group of disorders characterised clinically with acute systemic weakness and hypokalemia. Most cases are familial or primary hypokalemic periodic paralysis; sporadic cases are associated with other various conditions including barium poisoning, hyperthyroidism, renal disorders, certain endocrinopathies and gastrointestinal potassium losses (1). Gastrointestinal potassium loss is a rare cause, although reported in celiac disease (2), tropical sprue (3), short bowel syndrome (4), and acute gastroenteritis (5). Underlying causes are anorexia, active inflammation, bile salt and fat malabsorption, decreased of absorption surface, rapid gastrointestinal transit, lack of fat-soluble vitamins (6-7). There has been reported only one case with hypokalemic paralysis caused by intestinal Crohn’s disease in the literature (8). We know our patient is the first report of hypokalemic paralysis caused by involvement in upper gastrointestinal system and ileocolic Crohn‘s disease, as far. Interestingly, the patient had no gastrointestinal symptoms at presentation, despite evidence of malabsorption. Malnutrition is an important complication and frequently observed in patients with Crohn’s Disease (CD), usually associated with nutritional deficiencies, especially vitamins (both water- and fat-soluble) and essential trace elements. It is often due to the disease activity; poor oral intake or restrictive diets, bacterial overgrowth, bile salt malabsorption, extensive small-bowel disease, fructose malabsorption, inflammation, lactose malabsorption, multiple resections, short-bowel syndrome and surgery (9). Anemia and osteoporosis are the most frequent complications generated by nutritional deficiencies in inflammatory bowel diseases (IBD). Osteoporosis is caused by low intakes of calcium and vitaminD; anemia, may be associated with chronic iron loss, long lasting inflammation and nutritional deficiencies suh as folate and vitamin B 12 (10,13). The cause of IBD iron deficiency anemia (IDA) is multifactorial and due to a combination of iron deficiency and chronic gastrointestinal blood loss and, in some cases, self-imposed dietary restrictions. A study showed that vitamins of A, C, D, E, B12 , folate and level of calcium, iron and potassium were low in blood with Crohn’s disease (11). Gastrointestinal bleeding is the most common etiology for iron-deficiency anemia in IBD. Although patients with ileal CD may be at particular risk for vitamin B 12 deficiency, the unaffected small bowel may adapt and increase its ability to absorb vitamin B 12 efficiently. Distal ileal resection or active disease in this area, ie. the last 60 cm of ileum where B 12 is absorbed, resuled in B 12 deficiency, as well. Vitamin B 12 should be given parenterally, in such cases, due to lack of functional absorptive area in the terminal ileum. Beside these, the diarrhea, high-output stoma or fistulae, short-bowel syndrome, malabsorption and vomiting result in excessive amount electrolyte losses such as potassium, sodium, magnesium, selenium and zinc (12). However, hypophosphatemia may occur due to both vitamin D deficiency and decreased intestinal absorption. Hypophosphatemia is commonly missed due to nonspecific signs and symptoms, but it causes considerable morbidity and even mortality. Phosphate is abundantly present in many foods. Isolated dietary phosphate deficiency is uncommon, and the deficiency usually occurs with generalized malnutrition. Intestinal phosphate absorption is up-regulated by 1,25(OH) vitD3. Generalized muscle weakness is the most common symptom of hypophosphatemia; weakness and fatigue are related with acquired hypophosphatemia. Rhabdomyolysis is one of the most important clinical consequences in hypophosphatemia (14). Calorie-protein malnutrition may lead in the humoral and cellular immunodeficiency. Its effects on the intestine: decreasing the efficiency of the mucosal barrier, leading alteration in the mucosa-associated lymphoid tissue functions and finally a greater risk of infection by bacterial translocation. Moreover, hypoplasia of the intestinal villi perpetuates malabsorption and increases the risk of infections (15). Our case had paralysis due to hypokalemiae and hypophosphatemia associated with malabsorbtion. Patients with Crohn’s disease may present with the malabsorbtion symptoms, solely. Therefore, one must consider IBD, espcially Crohn disease, in a patient, even if with no gastrointestinal symptom.

1. Sushil K Ahlawat, Anita Sachdev, Postgrad Med J 1999;75:193-197 2. Musselman BC, Wenzl JE, Groover RV. Potassium-depletion paralysis: associated with gluten-induced enteropathy. Am J Dis Child 1968; 116 :414-7. 3. Ghosh D,Dhiman RK, Kohli A e t al. Hypokalemic periodic paralysis in association with tropical sprue: a case report. Acta Neurol Scand 1994; 90: 371-3. 4. Manary MJ, Keating JP, Hirshberg GE. Quadriparesis due to potassium depletion. Crit Care Med 1986; 14: 750 5. Orman RA, Lewis JB Jr. Flaccid quadriparesis associated with Yersinia enterocolitis-induced hypokalemia. Arch Intern Med 1989; 149:1193-4.2 6. Hutchinson R,Tyrrell PN, Kumar D, Dunn JA, Li JK, Allan RN, Pathogenesis of gall Stone in Crohn’s disease, an alternative explanation. Gut 1994 Jan;35(1):94-7 7. N.H.Dyer, A.M.Dawson Malnutrition and malabsorption in crohn’s disease with reference to the effect of surgery British Journal of Surgery Society 1973;60:134-140 8. Sambit Sen, Kinesh P. Patel, Aseel M.N. Fattah, Simon M. Greenfield. Crohn ’ s Disease Presenting as Acute Hypokalemic Paralysis, Am J Gastroenterol doi:10.1038/ajg.2009.374 9. Krok KL & Lichtenstein GR (2003) Nutrition in Crohn disease. Curr Opin Gastroenterol 19, 148–153. 10. Ballesteros P, Vidal Casariego A, Calleja Fernandéz A, López Gómez JJ, Urioste Fondo A, Cano Rodríguez I. Impact of nutritional treatment in the evolution of inflammatory bowel disease. Nutr Hosp 2010; 25 (2):181-192 11. T. Davanço et al. Nutritional supplementation assessment with whey proteins and TGF-βin patients with Crohn’s disease Nutr Hosp. 2012;27(4):1286-1292 12. Gassull MA & Cabre E (2001) Nutrition in inflammatory bowel disease. Curr Opin Clin Nutr Metab Care 4, 561–569. 13. Miranda C. E. Lomer et. Al Nutrition in inflammatory bowel disease Dietary and nutritional considerations for inflammatory bowel diseaseProceedings of the Nutrition Society (2011), 70, 329-335 14. Approach to the Hypophosphatemic Patient J Clin Endocrinol Metab, March 2012, 97(3): 696-706 15. 15. Lucendo AJ, De Rezende LC Importance of nutrition in inflammatory bowel disease. Worl J Gastroenterol 2009 May 7;15(17):2081-8