Is Capsule Endoscopy Indicated in Patients with Hereditary Hemorrhagic Telangiectasia and Normal Esophagoduodenoscopy Findings?

We read with interest the recent report by Ingrosso et al. [1], concerning the value of capsule endoscopy in patients with hereditary hemorrhagic telangiectasia (HHT). It represents a valuable effort to evaluate the diagnostic yield of this novel endoscopic tool in a specific disease, that is so frequently associated with acute or chronic anemia of obscure origin. According to their results, use of this new technique will probably change the etiological diagnosis of occult bleeding in HHT and may also be able to alter treatment strategies in HHT patients with gastrointestinal bleeding. Furthermore, capsule endoscopy will overcome the limitation of angiography in those patients without overt active bleeding.

However, in our opinion, more experience is needed before it can be stated that capsule endoscopy may be avoided in patients with HHT when conventional endoscopic explorations are negative. This caution is reinforced by our recent experience with a 53-year-old man, who was admitted to our hospital in May 2005 for evaluation of severe microcytic anemia. He had a personal and familial history (father, two brothers, and two sons) of recurrent epistaxis. Physical examination revealed a pale patient, with some telangiectases on the lingual and nasal mucosa, but with otherwise normal findings. On admission, a hemogram and a peripheral blood smear disclosed severe microcytic and hypochromic anemia, with a hemoglobin level of 4 g/dl, a red blood cell count of 2.4 × 10⁶/µl, a mean corpuscular volume of 53 fl, a serum iron level of 13 mg/dl, and a serum ferritin level of 16 ng/ml; white and platelet cell counts were normal. Liver and renal function tests, serum lactic dehydrogenase levels, a radiograph of the thorax, an esophagogastroduodenoscopy and colonoscopy, and a thoracic and abdominal computed tomography (CT) all showed normal results. Capsule endoscopy demonstrated a few small telangiectases in the distal duodenum and ileum and multiple large telangiectases in the jejunum. There was blood in the distal duodenum lumen, though no telangiectasic lesion was actively bleeding.

This case demonstrates that capsule endoscopy can disclose, in some patients, significant small-bowel involvement in HHT, even in the absence of positive findings after a conventional endoscopic work-up. This is in accordance with previous reports [2-4], relating to the group as a whole of patients with gastrointestinal bleeding of obscure origin, which suggest that capsule endoscopy should be considered as the next investigation after a nondiagnostic upper endoscopy and colonoscopy. We agree with Ingrosso et al. [1] about the difficulty of investigating the origin of blood losses in patients with HHT and associated epistaxis, and about the limitations of push enteroscopy as a screening procedure in these individuals. Therefore, we suggest that capsule endoscopy may be useful in this clinical setting, to evaluate the extent of small-bowel involvement and the need for more invasive diagnostic and/or therapeutic measures in all adult patients with HHT and severe anemia. Obviously, this statement is controversial, as the clinical impact and the cost-effectiveness of this strategy remain to be established, not only in the general group of patients, but also for specific diseases [5-7], including HHT and iron-deficiency anemia. This is, in our opinion, the key problem, which must be solved in future studies.

References

• 1 Ingrosso M, Sabba C, Pisani A. et al . Evidence of small-bowel involvement in hereditary hemorrhagic telangiectasia: a capsule-endoscopic study.  Endoscopy. 2004;  36 1074-1079
• 2 Selby W. Can clinical features predict the likelihood of finding abnormalities when using capsule endoscopy in patients with GI bleeding of obscure origin?.  Gastrointest Endosc. 2004;  59 782-787
• 3 Delvaux M, Fassler I, Gay G. Clinical usefulness of the endoscopic video capsule as the initial intestinal investigation in patients with obscure digestive bleeding: validation of a diagnostic strategy based on the patient outcome after 12 months.  Endoscopy. 2004;  36 1067-1073
• 4 Pennazio M. Small-bowel endoscopy.  Endoscopy. 2004;  36 32-41
• 5 Pennazio M, Santucci R, Rondonotti E. et al . Outcome of patients with obscure gastrointestinal bleeding after capsule endoscopy: report of 100 consecutive cases.  Gastroenterology. 2004;  126 643-653
• 6 Saurin J C, Delvaux M, Vahedi K. et al . Clinical impact of capsule endoscopy compared to push enteroscopy: 1-year follow-up study.  Endoscopy. 2005;  37 318-323
• 7 Herrerias J M, Caunedo A, Rodriguez-Tellez M. et al . Capsule endoscopy in patients with suspected Crohn’s disease and negative endoscopy.  Endoscopy. 2003;  35 564-568