REPORT OF CASE A 79-year-old white male, born in theNetherlands, was admitted to Mount Sinai Hospital withsevere anginal pain and anemia with a hematocrit of 24%.
He had a history of recurrent anemia and guaiac positivestools for at least 6 years, requiring repeated hospitalizationsand transfusions. No source for the gastrointestinal bleedingwas ever found. He had had a myocardial infarction 6 yearspreviously and felt well between bleeding episodes.
The family history disclosed several children and manygrandchildren denying cutaneous telangiectasis, epistaxis, orgastrointestinal bleeding episodes.
On physical examination, he was well developed, thin,and pale and complained of severe substernal chest pain.Other than the presence of bilateral inguinal hernias, noabnormalities were detected. The femoral pulses were active,but no popliteal or foot arterial pulsations were palpable.No cutaneous lesions were seen.
Following transfusion with 2 units of whole blood, theangina promptly disappeared.
The hematocrit on admission was 24% and on discharge,after transfusions, was 42%. Reticulocyte count was 4.4%on admission; stool guaiac was 4+ positive; serum iron was58 mcg, and total iron binding capacity was 370 mcg. Chestx-ray and barium enema were normal. The upper gastrointestinal series revealed 2 traction diverticula of the esophagus.Bone marrow examination revealed no specific disease. Visceral angiography was not performed because of the inherentrisk of arterial puncture in the absence of peripheral pulses.Sigmoidoscopy was normal.
85
Duodenal telangiectasis in an occultgastrointestinal bleeder
Harold W. Grotsky, M.D.Jerome D. Waye, M.D.
New York, New York
Duodenoscopy disclosed visceral telangiectasia as a probable causeof bleeding in an elderly man with recurring anemia.
The diagnosis of the exact cause of occult gastrointestinal Endoscopy was performed with an Olympus-JF duodenos-hemorrhage has long presented a difficult problem for the cope. The esophagus, stomach, and antrum were normal.physician. The duodenum was entered, and 3 separate but similar lesions
Visceral angiography added to the standard techniques of were seen. Each was noted to consist of a group of bloodbarium swallow, small bowel series, and barium enema has vessels emanating from a central point with radial distributionbeen of great aid in pinpointing the bleeding site in patients similar to a spider's legs. Each lesion was separated fromwith chronic active bleeding. In the elderly, angiography may the others by several centimeters of normal mucosa. Theirnot be feasible due to vascular disease, and other modalities distribution was in a random fashion around the walls ofof investigation must be sought. the descending duodenum. There was no active bleeding
The present report describes a patient with occult bleeding at the time of the endoscopic examination. These appearedupon whom angiography could not be performed, and similar to teliangectatic spiders as seen on the skin of cirrho-"standard" x-ray studies as well as "routine" endoscopic tics.examination failed to reveal any possible etiology of the blood The patient stopped bleeding spontaneously and is beingloss. followed with weekly hematocrits.
Duodenoscopic examination with one of the newer instru- DISCUSSION Familial hemorrhagic telangiectasis (Osler-ments permitted us to see telangiectases in the duodenum, Weber-Rendu syndrome), a syndrome of intestinal bleedingthereby extending our visual diagnostic ability into the upper with mucocutaneous telangiectases and recurrent epistaxis,small bowel. was popularized in 1901 when Sir William Osler first
appreciated the familial nature of the disease.' The first caseof this syndrome was published by Sutton in 1964.2
Isolated intestinal bleeding may occur as the presentingsymptom of the Osler-Weber-Rendu syndrome,3 and bleedingmay occur in the absence of cutaneous manifestations.'
Endoscopy has been useful in detecting the site of intestinalbleeding, and telangiectases have been recognized in theesophagus, stomach, and rectosigmoid.' Endoscopes mayalso be passed on the operating table through enterostomiesto ascertain the site of hemorrhage during surgery6,7 in patientswith Osler-Weber-Rendu syndrome.
Our patient had no cutaneous manifestations of telangiectases, but lesions were readily seen in the duodenum.Although these were not bleeding during the endoscopicprocedure, they reveal a highly potential bleeding sourceand the only site seen in a man with chronic gastrointestinal
blood loss. r~~::rl
REFERENCES1. OSLER W: On a family form of recurring epistaxis associated with multiple
telangiectases of the skin and mucous membranes. Bull Johns HopkinsHosp 12:333-337, 1901
2. SUnON HG: Epistaxis as an indication of impaired nutrition and degeneration of the vascular system. Med Mirror 1:769, 1864
3. VETTO RM: The management of multiple, diffuse telangiectasia of thesmall intestine. Surg Gyn Obst 115 :56·60, 1962
4. JACOBSON G, KRAUSE U: Hereditary haemorrhagic telangiectasia local·ized to the gastrointestinal tract. Scand J Gastroent 5:283-288,0000
5. Sanowski SANOWSKI RA: Hereditary hemorrhagic telangiectasia: A clinical and endoscopic study. Gastrointestinal Endoscopy 16:225-226, 1970
6. SWEETING jG: Fiberoptic examination of the small intestine in a case offamilial telangiectasia. Gastrointestinal Endoscopy 14:152-153, 1968
7. RENSHAW jF: Multiple hemorrhagic telangiectasia with special referenceto gastroscopic appearance. Clev Clin Quart 6:226, 1939
From the Department of Medicine, Division of Gastroenterology, The Mount Sinai MedicalSchool, New York, New York.
VOLUME 19, NO.2, 1972
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