Chalapathi Rao

Deepak K Bhasin

Kartar Singh

Surinder S Rana

Vishal Sharma

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© 2012 Hellenic Society of Gastroenterology www.annalsgastro.gr
Obscure gastrointestinal bleeding persisting for a decade:  
a rare manifestation of a common disease
Surinder S. Rana, Vishal Sharma, Chalapathi Rao, Kartar Singh, Deepak K. Bhasin
Post Graduate Institute of Medical Education and Research (PGIMER), Chandigarh, India
Annals of Gastroenterology (2012) 25, 1-3
Introduction
Celiac disease is an autoimmune enteropathy precipitated 
by exposure to gluten. Although initially considered a rare 
malabsorption syndrome in children, it is now recognized as 
a systemic disorder with varying presentation and multiple 
manifestations affecting both pediatric and adult populations 
[1]. It commonly presents with diarrhea in children, whereas 
in adults the presentation is variable and may include anemia, 
osteoporosis, and liver function abnormalities [1,2]. Although 
occult gastrointestinal blood loss is common [3], overt bleed-
ing is uncommon. We present the case of a 60-year-old lady 
who presented with obscure gastrointestinal blood loss for 
more than a decade necessitating multiple transfusions until 
celiac disease was eventually diagnosed. 
Case report
A 60-year-old lady was referred to us for melena of 12 
years duration. She had earlier undergone repeat evaluations 
at multiple private hospitals. She was initially diagnosed with 
duodenal ulcer and underwent exploratory laparotomy with 
pyloroplasty and truncal vagotomy. However, she rebled 
and thereafter underwent antrectomy with retrocolic gas-
trojejunostomy. She also received proton pump inhibitors, 
and eradication therapy for Helicobacter pylori. Despite the 
aggressive treatment, the patient continued to have melena 
episodes and required multiple blood transfusions (~170 
transfusions over a decade). Her esophagogastroduodenoscopy, 
colonoscopy and barium meal follow through did not reveal 
any abnormality. Contrast enhanced computed tomography 
(CECT) of abdomen and the technetium-99m red blood cell 
scan were also non-contributory. A capsule endoscopy was 
performed by placing capsule in efferent limb under endo-
scopic guidance (Fig. 1). It revealed multiple ulcers of varying 
sizes throughout the small bowel (Fig. 2). As there was no 
history of non steroidal anti inflammatory drugs (NSAID) 
ingestion, a possibility of chronic non-specific small bowel 
ulcers was kept. Antinuclear antibodies and anti-neutrophil 
cytoplasmic antibodies were negative. However, IgA anti-tissue 
transglutaminase and anti-gliadin antibodies were positive. 
Antegrade double balloon enteroscopy was performed and 
biopsies were obtained from ulcer edge as well as mucosal 
folds. Histological examination of the biopsies from the ulcer 
edge revealed chronic inflammation and from the mucosal 
folds revealed increased intraepithelial lymphocytes. She 
was started on prednisolone 40 mg daily along with gluten-
free diet. The melena subsided and thereafter steroids were 
tapered off. She remains well after one-year follow up, there 
has been no recurrence of bleeding and her hemoglobin has 
risen to 13.4 mg/dL. 
Discussion
Celiac disease is an autoimmune disorder characterized 
by sensitivity to gluten. It can be complicated by occurrence 
Department of Gastroenterology, Post Graduate Institute of Medical 
Education and Research (PGIMER), Chandigarh 160012, India
Conflict of Interest: None
Correspondence to: Dr Surinder Singh Rana, Asst. Prof., 
Department of Gastroenterology, Postgraduate Institute of Medical 
Education and Research (PGIMER), Chandigarh, India 160012,  
Tel: +91 172 2749123, Fax: +91 172 2744401,  
e-mail: drsurinderrana@yahoo.co.in, sonalisurinder@yahoo.co.in
Received 16 February 2012; accepted 26 March 2012
CASE REPORT
Abstract Celiac disease commonly presents with diarrhea but variable presentation with anemia, osteo-
porosis, incidental recognition, and liver function abnormalities is also known. Overt blood 
loss is uncommon in celiac disease. We present the case of a 60-year-old female who presented 
with obscure gastrointestinal blood loss for more than a decade necessitating multiple transfu-
sions and was eventually diagnosed to have celiac disease. After introduction of gluten-free 
diet, her symptoms improved and there has been no recurrence of gastrointestinal bleeding.
Keywords celiac disease, capsule endoscopy, gastrointestinal bleeding
Ann Gastroenterol 2012; 25 (3): 1-3
Annals of Gastroenterology 25
2   S.S. Rana et al
of ulcerative jejunoileitis or malignant transformation [1]. 
Celiac disease has been associated with occult gastrointestinal 
blood loss [3,4]. In a study in children, occult gastrointestinal 
blood loss was reported in around one-fourth of patients and 
improved with gluten-free diet [3]. However, another study 
which utilized the more specific direct radiochromium-labeled 
red blood cells indicated that the blood loss was infrequent and 
the traditional colorimetric test were false positive probably 
due to excess intestinal cell loss or malabsorption affecting 
peroxidase containing foods [5]. Overt gastrointestinal blood 
loss in a patient with celiac disease may point to presence of 
Figure 2 Ulcer in the jejunum (A) and ulcer with normal surrounding small bowel mucosa (B)
Figure 1 Videocapsule endoscope being deployed in the efferent loop. 
Wires of the dormia seen around the capsule endoscope
A B
malignancy like enteropathy-associated T cell lymphoma or 
adenocarcinoma, ulcerative jejunoileitis, vitamin K deficiency, 
occasionally due to varices, which may result from associ-
ated portal vein thrombosis or chronic liver disease, or other 
unrelated causes [6-9].
In our patient the capsule endoscopy revealed multiple 
ulcers in the small bowel. She was initially thought of having 
chronic non-specific ulceration of the small bowel. However, 
eventually celiac disease was found to be the cause of overt 
gastrointestinal bleeding in our patient. She improved after 
treatment with steroids and initiation of gluten-free diet and 
remained well with a rise in hemoglobin even after steroids 
were tapered. The long duration of the disease, the complete 
response after the introduction of gluten-free diet, and the 
absence of refractory symptoms suggest that these ulcers may 
have been benign. The etiology of these ulcers is not clear 
but,similar to the aphthous ulcers in the oral cavity of celiac 
disease patients, they might be immunologically mediated. 
Absence of relapse on gluten-free diet also suggests that the 
exclusion of the antigen from the diet may have led to remission.
The surgically altered proximal bowel also complicated 
the evaluation of the patient. Capsule endoscopy may be 
indicated in patients with celiac disease for evaluation of 
acute abdominal pain or occult gastrointestinal blood loss 
and is particularly useful in evaluation of complicated 
celiac disease [10,11]. In certain cases of iron deficiency 
anemia the diagnosis of celiac disease may be established 
only after capsule endoscopy [12]. The present report is 
also important as it highlights celiac disease with mucosal 
ulcerations as a cause of overt gastrointestinal blood loss. 
An earlier report had implicated celiac-related lymphocytic 
gastritis as a cause of fatal gastrointestinal blood loss even 
in the absence of malignant transformation [13]. Mucosal 
ulceration of the small intestine, especially jejunum, is 
common in celiac disease [14]. To conclude, celiac disease 
Annals of Gastroenterology 25
Long-term overt GI bleeding in celiac disease   3
must be considered in the differential diagnosis in patients 
with obscure gastrointestinal bleeding and when ulcers are 
visualized in the small bowel. 
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Obscure gastrointestinal bleeding persisting for a decade: a rare manifestation of a common disease

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Obscure gastrointestinal bleeding persisting for a decade: a rare manifestation of a common disease

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