Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 


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

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


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

Date of Submission09-Apr-2019
Date of Acceptance05-Nov-2019
Date of Web Publication12-Dec-2019

Correspondence Address:
Abdullah Mohammed Albishi
Department of Gastroenterology and Endoscopy, Armed Forces Hospital Southern Region, Khamis Mushayt
Saudi Arabia
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ssj.ssj_18_19

Rights and Permissions
  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.

Keywords: Abdominal pain, coeliac disease, intussusception, repeated, small bowel disease


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;7:163-6

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 Jul 5];7:163-6. Available from: http://www.saudisurgj.org/text.asp?2019/7/4/163/272845


  Introduction Top


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 Top


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: Computed tomography abdomen showing jejunal loop intussusceptions with diffuse jejunal wall thickening

Click here to view
Figure 2: Upper gastrointestinal endoscopy showing mild fissuring and scalloping of duodenal folds in the second part of duodenum

Click here to view


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 Top


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 Top


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 Top

1.
National 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].  Back to cited text no. 1
    
2.
Tovoli 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.  Back to cited text no. 2
    
3.
Penagini 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.  Back to cited text no. 3
    
4.
Di Sabatino A, Corazza GR. Coeliac disease. Lancet 2009;373:1480-93.  Back to cited text no. 4
    
5.
Fasano A. Clinical presentation of celiac disease in the pediatric population. Gastroenterology 2005;128:S68-73.  Back to cited text no. 5
    
6.
Rostami Nejad M, Hogg-Kollars S, Ishaq S, Rostami K. Subclinical celiac disease and gluten sensitivity. Gastroenterol Hepatol Bed Bench 2011;4:102-8.  Back to cited text no. 6
    
7.
Tonutti E, Bizzaro N. Diagnosis and classification of celiac disease and gluten sensitivity. Autoimmun Rev 2014;13:472-6.  Back to cited text no. 7
    
8.
Newnham ED. Coeliac disease in the 21st century: Paradigm shifts in the modern age. J Gastroenterol Hepatol 2017;32 Suppl 1:82-5.  Back to cited text no. 8
    
9.
Marsicovetere P, Ivatury SJ, White B, Holubar SD. Intestinal intussusception: Etiology, diagnosis, and treatment. Clin Colon Rectal Surg 2017;30:30-9.  Back to cited text no. 9
    
10.
Gluckman S, Karpelowsky J, Webster AC, McGee RG. Management for intussusception in children. Cochrane Database Syst Rev 2017;6:CD006476.  Back to cited text no. 10
    
11.
Germann 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.  Back to cited text no. 11
    
12.
Maconi 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.  Back to cited text no. 12
    
13.
Marsh MN. Mucosal pathology in gluten sensitivity. In: Marsh MN, editor. Coeliac Disease. Oxford: Blackwell Scientific; 1992:136-91.  Back to cited text no. 13
    
14.
Gee S. On the coeliac disease. St Bart Hosp Rep 1888;24:17-20.  Back to cited text no. 14
    
15.
Mäki M, Collin P. Coeliac disease. Lancet 1997;349:1755-9.  Back to cited text no. 15
    
16.
Csizmadia 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.  Back to cited text no. 16
    
17.
Mäki 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.  Back to cited text no. 17
    
18.
West 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.  Back to cited text no. 18
    
19.
Catassi C, Bearzi I, Holmes GK. Association of celiac disease and intestinal lymphomas and other cancers. Gastroenterology 2005;128:S79-86.  Back to cited text no. 19
    
20.
Logan 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.  Back to cited text no. 20
    
21.
Hin H, Bird G, Fisher P, Mahy N, Jewell D. Coeliac disease in primary care: Case finding study. BMJ 1999;318:164-7.  Back to cited text no. 21
    
22.
Zipser 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.  Back to cited text no. 22
    
23.
Ruoff M, Lindner AE, Marshak RH. Intussusception in sprue. Am J Roentgenol Radium Ther Nucl Med 1968;104:525-8.  Back to cited text no. 23
    
24.
Reilly NR, Aguilar KM, Green PH. Should intussusception in children prompt screening for celiac disease? J Pediatr Gastroenterol Nutr 2013;56:56-9.  Back to cited text no. 24
    
25.
Mushtaq 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.  Back to cited text no. 25
    
26.
Martinez G, Israel NR, White JJ. Celiac disease presenting as entero-enteral intussusception. Pediatr Surg Int 2001;17:68-70.  Back to cited text no. 26
    
27.
Lastennet 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.  Back to cited text no. 27
    
28.
Fishman 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.  Back to cited text no. 28
    
29.
Altaf MA, Grunow JE. Atypical presentations of celiac disease: Recurrent intussusception and pneumatosis intestinalis. Clin Pediatr (Phila) 2008;47:289-92.  Back to cited text no. 29
    
30.
Al 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.  Back to cited text no. 30
    


    Figures

  [Figure 1], [Figure 2]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed410    
    Printed36    
    Emailed0    
    PDF Downloaded62    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]