Saudi Surgical Journal

CASE REPORT
Year
: 2019  |  Volume : 7  |  Issue : 4  |  Page : 163--166

Multiple wandering jejunal intussusceptions in an adult patient with celiac disease: Atypical presentation


Abdullah Mohammed Albishi1, Yahia Atiah Alhagawi1, Nasser Ibraheem Alqahtani1, Rafaat Chakik1, Mohamed F Bazeed2, M Ezzedien Rabie3, Mohammed Tagelsir Elmaki4, Bushra Mofreh Asiri4, Mubarak Mohammed Alshahrani5,  
1 Department of Gastroenterology and Endoscopy, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia
2 Department of Radiology, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia
3 Department of General Surgery, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia
4 Department of Internal Medicine, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia
5 Family Medicine Primary Health Care, Ministry of Health, Abha, Saudi Arabia

Correspondence Address:
Abdullah Mohammed Albishi
Department of Gastroenterology and Endoscopy, Armed Forces Hospital Southern Region, Khamis Mushayt
Saudi Arabia

Abstract

Intussusception is a rare presentation of celiac disease. In this report, we describe the condition of a 28-year-old Saudi male known case of brucellosis diagnosed and treated 10 months ago. The patient presented to the hospital complaining of diffuse intermittent dull abdominal pain with mild abdominal distention for 1-month duration, which starts gradually with progressive course. Patient also was given history of generalized fatigability for the same duration. These symptoms were associated with a weight loss about 10 kg in the last 6 months. Abdominal examination revealed soft and lax abdomen with mild tenderness at epigastria area. Computed tomography (CT) abdomen showed jejunal loop intussusceptions with diffuse jejunal wall thickening. Gastroscopy was done and showed fissuring of duodenal folds. Biopsy report came later as duodenal villa atrophy with increased intraepithelial lymphocytosis. The patient diagnosed as celiac disease. After establishing the diagnosis, dietary advice was given and he was discharged for outpatient department follow-up. Currently, the patient remains well and repeat CT scan showed persistence of multiple intussusceptions, though at different sites of the jejunum.



How to cite this article:
Albishi AM, Alhagawi YA, Alqahtani NI, Chakik R, Bazeed MF, Rabie M E, Elmaki MT, Asiri BM, Alshahrani MM. Multiple wandering jejunal intussusceptions in an adult patient with celiac disease: Atypical presentation.Saudi Surg J 2019;7:163-166


How to cite this URL:
Albishi AM, Alhagawi YA, Alqahtani NI, Chakik R, Bazeed MF, Rabie M E, Elmaki MT, Asiri BM, Alshahrani MM. Multiple wandering jejunal intussusceptions in an adult patient with celiac disease: Atypical presentation. Saudi Surg J [serial online] 2019 [cited 2020 Oct 30 ];7:163-166
Available from: https://www.saudisurgj.org/text.asp?2019/7/4/163/272845


Full Text

 Introduction



Coeliac disease or celiac disease is a long-term autoimmune disorder that primarily affects the small intestine.[1] Coeliac disease is caused by a reaction to gluten, a group of various proteins found in wheat and in other grains such as barley and rye.[2],[3],[4] Classic symptoms include gastrointestinal (GI) problems such as chronic diarrhea, abdominal distention, malabsorption and among children failure to grow normally.[5] Nonclassic symptoms are more common, especially in people older than 2 years. There may be mild or absent GI symptoms, a wide number of symptoms involving any part of the body or no obvious symptoms.[6],[7],[8] Intussusception is a medical condition in which a part of the intestine folds into the section immediately beside it. It typically involves the small bowel and less commonly the large bowel.[9],[10] Intussusception is rare in adults. Reported cases of intussusception in celiac disease suggest that it may be asymptomatic, transient, and limited to the small intestine and rarely requires surgical intervention;[11] however, enteropathy-associated T-cell lymphoma should be considered in the differential diagnosis.[12]

 Case Report



A 28-year-old Saudi male with past history of brucellosis diagnosed and treated 10 months ago. The patient presented to the hospital complaining of diffuse intermittent dull abdominal pain with mild abdominal distention 1-month duration which starts gradually with progressive course. Patient also was given history of generalized fatigability for the same duration. Nausea with occasional vomiting was also recorded by the patient. These symptoms were associated with weight loss of about 10 kg in the last 6 months. No history of fever or night sweating was observed. No history of GI bleeding was noted. No history of skin rash or joint pain or bone pain was also observed. The patients had neither jaundice nor history of chest pain, shortness of breath, or cough. No history of dysuria or change in color or frequency of urine was noted. No history of similar attack before. No significant family history. In addition, surgery or drug history was negative.

The patient was diagnosed as brucellosis case 10 months ago as he complained of fever and arthralgia, which started on antibiotics, but they noticed that time he had picture of unexplained iron deficiency anemia, and then patient missed the follow-up.

Clinical examination

The patient was conscious, oriented, pale, but neither jaundice nor lymphadenopathy. Body mass index was 19.5. The patient was vitally stable and afebrile. Chest and cardiovascular examinations were free.

Abdominal examination revealed soft and lax abdomen with mild tenderness at epigastria area. No organ enlargement was detected, but bowel sound was recorded. Lower limb examination was normal.

Investigation

Laboratory investigations revealed white blood cell = 9.7, Hb = 10.5 g/dl, MCV = 66.1, MCH = 19.1, PLT = 306, and INR = 1.1.

Liver function test and renal profile were normal.

Ferritin was low, normal thyroid function test, tissue transglutaminase IgA was positive with high titer.

Imaging

Computed tomography (CT) abdomen showed jejunal loop intussusceptions with diffuse jejunal wall thickening [Figure 1]. Gastroscopy was done and showed mild gastric erythematous mucosa at gastric antrum with fissuring and scalloping of duodenal folds of second part of duodenum (5 biopsies were taken) [Figure 2].{Figure 1}{Figure 2}

Duodenal biopsy report showed duodenal villa atrophy with increase intraepithelial lymphocytosis which going with celiac disease. The patient diagnosed as celiac disease.

Treatment

After final diagnosis, the patient started on gluten-free diet and dietician referral was done, while micronutrient deficiencies were also corrected. The patient followed by general surgeon regarding the intussusception, which does not need any surgical intervention.

Outcome and follow-up

The patient was seen after 3 months, clinical symptoms were improved, with neither more abdominal pain nor distention. The patient had good appetite and no more fatigability. His body mass index was improved to 24, and his abdomen was soft lax with no tenderness. Patient laboratory investigations also were improved as Hg was increased to 12 g/dl, and the celiac serology titer was improved while patient was on strict gluten free diet. Follow-up CT abdomen revealed persistence of multiple intussusceptions, though at different sites of the jejunum. The patient instructed to be on strict gluten-free diet with follow-up in clinic after 2 months.

 Discussion



Coeliac disease is an autoimmune GI disorder caused by permanent intolerance to ingested gluten in genetically susceptible individuals.[13],[14],[15] Its magnitude was reported among Caucasians in Europe, North and South America, Australia, and the Middle East to be as high as 1 in 100.[16],[17] Conventionally, coeliac disease clinically presented with symptoms of intestinal malabsorption disorder, resulting in weight loss, diarrhea, steatorrhea, or abdominal distension. Regarding these typical clinical presentations, coeliac disease can be presented by other atypical complaints such as isolated subclinical iron deficiency anemia, osteoporosis, neurologic disease, nonspecific abdominal symptoms, dermatitis herpetiformis, or even intussusception.[18],[19] In recent years, there has been increasing recognition that the mode of presentation of coeliac disease may be changing.[20] It often presents with symptoms not previously considered to be characteristic of the disease.[21],[22]

Intussusception is not a commonly recorded complication of celiac disease. In 1968, Ruoff et al.[23] stated the occurrence of intussusception in adult celiac disease for the first time. An occurrence of intussusception among adults with celiac disease had been reported. Although more common among children, intussusception not confirmed among childhood celiac disease.[24] Germann et al.[11] in 1997 recorded celiac disease as an uncommon cause of recurrent intussusception in children for first time. Then, Mushtaq et al.[25] reported three children with intestinal intussusception in conjunction with celiac disease and recommended that the finding of transient small bowel intussusception should be considered clinically for celiac disease. Another many cases of intussusception among cases of coeliac disease were recorded.[26],[27],[28],[29],[30]

All these reported associations between intussusception and coeliac disease were in concordance with these cases reported in this study as the patient reported here also complained of abdominal pain with bowel motion disturbances, especially constipation, and after clinical examination and biopsy, he was confirmed as a case of intussusception for coeliac disease patient.

Intussusception can be a clinical complication of celiac disease so that celiac disease should be considered in patients with intussusception and growth failure, especially in unusual age range.

 Conclusion



Small bowel intussusception in adult with suspected celiac disease initially should be considered and managed expectantly rather than by early surgical intervention. The finding of transient small bowel intussusception should prompt investigation for celiac disease.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1National Institute of Diabetes and Digestive and Kidney. Archived from the Original on 13 March 2016. National Institute of Diabetes and Digestive and Kidney; June, 2015. Available from: https://www.niddk.nih.gov/health-information/digestive-diseases/celiac-disease. [Last retrieved 2016 Mar 17].
2Tovoli F, Masi C, Guidetti E, Negrini G, Paterini P, Bolondi L, et al. Clinical and diagnostic aspects of gluten related disorders. World J Clin Cases 2015;3:275-84.
3Penagini F, Dilillo D, Meneghin F, Mameli C, Fabiano V, Zuccotti GV, et al. Gluten-free diet in children: An approach to a nutritionally adequate and balanced diet. Nutrients 2013;5:4553-65.
4Di Sabatino A, Corazza GR. Coeliac disease. Lancet 2009;373:1480-93.
5Fasano A. Clinical presentation of celiac disease in the pediatric population. Gastroenterology 2005;128:S68-73.
6Rostami Nejad M, Hogg-Kollars S, Ishaq S, Rostami K. Subclinical celiac disease and gluten sensitivity. Gastroenterol Hepatol Bed Bench 2011;4:102-8.
7Tonutti E, Bizzaro N. Diagnosis and classification of celiac disease and gluten sensitivity. Autoimmun Rev 2014;13:472-6.
8Newnham ED. Coeliac disease in the 21st century: Paradigm shifts in the modern age. J Gastroenterol Hepatol 2017;32 Suppl 1:82-5.
9Marsicovetere P, Ivatury SJ, White B, Holubar SD. Intestinal intussusception: Etiology, diagnosis, and treatment. Clin Colon Rectal Surg 2017;30:30-9.
10Gluckman S, Karpelowsky J, Webster AC, McGee RG. Management for intussusception in children. Cochrane Database Syst Rev 2017;6:CD006476.
11Germann R, Kuch M, Prinz K, Ebbing A, Schindera F. Celiac disease: An uncommon cause of recurrent intussusception. J Pediatr Gastroenterol Nutr 1997;25:415-6.
12Maconi G, Radice E, Greco S, Bezzio C, Bianchi Porro G. Transient small-bowel intussusceptions in adults: Significance of ultrasonographic detection. Clin Radiol 2007;62:792-7.
13Marsh MN. Mucosal pathology in gluten sensitivity. In: Marsh MN, editor. Coeliac Disease. Oxford: Blackwell Scientific; 1992:136-91.
14Gee S. On the coeliac disease. St Bart Hosp Rep 1888;24:17-20.
15Mäki M, Collin P. Coeliac disease. Lancet 1997;349:1755-9.
16Csizmadia CG, Mearin ML, von Blomberg BM, Brand R, Verloove-Vanhorick SP. An iceberg of childhood coeliac disease in the Netherlands. Lancet 1999;353:813-4.
17Mäki M, Mustalahti K, Kokkonen J, Kulmala P, Haapalahti M, Karttunen T, et al. Prevalence of celiac disease among children in finland. N Engl J Med 2003;348:2517-24.
18West J, Logan RF, Hill PG, Lloyd A, Lewis S, Hubbard R, et al. Seroprevalence, correlates, and characteristics of undetected coeliac disease in England. Gut 2003;52:960-5.
19Catassi C, Bearzi I, Holmes GK. Association of celiac disease and intestinal lymphomas and other cancers. Gastroenterology 2005;128:S79-86.
20Logan RF, Tucker G, Rifkind EA, Heading RC, Ferguson A. Changes in clinical features of coeliac disease in adults in Edinburgh and the Lothians 1960-79. Br Med J (Clin Res Ed) 1983;286:95-7.
21Hin H, Bird G, Fisher P, Mahy N, Jewell D. Coeliac disease in primary care: Case finding study. BMJ 1999;318:164-7.
22Zipser RD, Patel S, Yahya KZ, Baisch DW, Monarch E. Presentations of adult celiac disease in a nationwide patient support group. Dig Dis Sci 2003;48:761-4.
23Ruoff M, Lindner AE, Marshak RH. Intussusception in sprue. Am J Roentgenol Radium Ther Nucl Med 1968;104:525-8.
24Reilly NR, Aguilar KM, Green PH. Should intussusception in children prompt screening for celiac disease? J Pediatr Gastroenterol Nutr 2013;56:56-9.
25Mushtaq N, Marven S, Walker J, Puntis JW, Rudolf M, Stringer MD, et al. Small bowel intussusception in celiac disease. J Pediatr Surg 1999;34:1833-5.
26Martinez G, Israel NR, White JJ. Celiac disease presenting as entero-enteral intussusception. Pediatr Surg Int 2001;17:68-70.
27Lastennet F, Piloquet H, Camby C, Moussally F, Siret D. Acute intestinal invagination revealing celiac disease in a 9-month-old infant. Arch Pediatr 2002;9:151-4.
28Fishman DS, Chumpitazi BP, Ngo PD, Kim HB, Lightdale JR. Small bowel intussusception in celiac disease: Revisiting a classic association. J Pediatr Gastroenterol Nutr 2010;50:237.
29Altaf MA, Grunow JE. Atypical presentations of celiac disease: Recurrent intussusception and pneumatosis intestinalis. Clin Pediatr (Phila) 2008;47:289-92.
30Al Furaikh S, Al Zaben AA. Recurrent small bowel intussusceptions: An uncommon presentation of celiac disease in an Arab child. Trop Gastroenterol 2005;26:38-9.