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Intestinal neurofibromatosis in von Recklinghausen's disease: presenting as chronic anemia due to recurrent intestinal hemorrhage.

Hahn JS, Chung JB, Han SH, Lee SW, Noh SH, Lee JT, Chun SI, Kim GH - Korean J. Intern. Med. (1992)

Bottom Line: Neurofibromatosis (von Recklinghausen's disease) is a neuroectodermal disorder characterized by pigmentary changes of the skin (café-au-lait spots), cutaneous and visceral tumors (neurofibromas) and systemic abnormalities.The involvement of gastrointestinal tract in neurofibromatosis is not common.The most common symptoms, refer able to lesions in the gut, are hematemesis, melena and abdominal pain.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea.

ABSTRACT
Neurofibromatosis (von Recklinghausen's disease) is a neuroectodermal disorder characterized by pigmentary changes of the skin (café-au-lait spots), cutaneous and visceral tumors (neurofibromas) and systemic abnormalities. The involvement of gastrointestinal tract in neurofibromatosis is not common. The most common symptoms, refer able to lesions in the gut, are hematemesis, melena and abdominal pain. We experienced a case of intestinal neurofibroma in von Recklinghausen's disease. The patient was a 39 year-old female who had suffered from chronic iron deficiency anemia and recurrent gastrointestinal hemorrhage due to two neurofibromas of jejunum for 3 years, which was diagnosed by superior mesenteric and ileal arteriogram and 99mTc pertechnetate-labelled RBC scan, and treated by segmental resection of jejunum with end to end anastomosis.

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Radionuclide angiogram using 99mTc pertechnate-labelled RBC 20 mCi intravenously and taken 30 minutes after injecion showing an abnormal accumulation of radioactivity in the pelvic cavity (arrow).
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f3-kjim-7-2-137-10: Radionuclide angiogram using 99mTc pertechnate-labelled RBC 20 mCi intravenously and taken 30 minutes after injecion showing an abnormal accumulation of radioactivity in the pelvic cavity (arrow).

Mentions: After transfusion of 5 units of packed red cell, the hemoglobin was 9.9 g/dl, the hematocrit 31.1 percent. The fiberoptic gastroscopy showed mild diffuse chronic superficial gastritis. The UGI series and small bowel series (Fig. 2) revealed no specific abnormal findings. The radionuclide scanning, using 99mTc pertechnate-labelled RBC, revealed abnormal radioactivity in the right lower abdomen and increased activity by the time sequence, which was suggestive of lower gastrointestinal bleeding from the right hypogastric arterial branch (Fig. 3). On the flexible sigmoidoscopy, internal hemorrhoid without evidence of recent active bleeding was found. The superior mesenteric and ileal arteriogram showed bleeding from the left ileal arterial branches (Fig. 4). On the 15th hospital day, operation was performed under the diagnosis of small bowel tumor. There were two masses on jejunum located 60 and 120 cm from the ligament of Treitz, respectively. The distal mass was irregulary lobulated with some ulcerations on its surface, and tumor bleeding into the lumen was noted.


Intestinal neurofibromatosis in von Recklinghausen's disease: presenting as chronic anemia due to recurrent intestinal hemorrhage.

Hahn JS, Chung JB, Han SH, Lee SW, Noh SH, Lee JT, Chun SI, Kim GH - Korean J. Intern. Med. (1992)

Radionuclide angiogram using 99mTc pertechnate-labelled RBC 20 mCi intravenously and taken 30 minutes after injecion showing an abnormal accumulation of radioactivity in the pelvic cavity (arrow).
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4532108&req=5

f3-kjim-7-2-137-10: Radionuclide angiogram using 99mTc pertechnate-labelled RBC 20 mCi intravenously and taken 30 minutes after injecion showing an abnormal accumulation of radioactivity in the pelvic cavity (arrow).
Mentions: After transfusion of 5 units of packed red cell, the hemoglobin was 9.9 g/dl, the hematocrit 31.1 percent. The fiberoptic gastroscopy showed mild diffuse chronic superficial gastritis. The UGI series and small bowel series (Fig. 2) revealed no specific abnormal findings. The radionuclide scanning, using 99mTc pertechnate-labelled RBC, revealed abnormal radioactivity in the right lower abdomen and increased activity by the time sequence, which was suggestive of lower gastrointestinal bleeding from the right hypogastric arterial branch (Fig. 3). On the flexible sigmoidoscopy, internal hemorrhoid without evidence of recent active bleeding was found. The superior mesenteric and ileal arteriogram showed bleeding from the left ileal arterial branches (Fig. 4). On the 15th hospital day, operation was performed under the diagnosis of small bowel tumor. There were two masses on jejunum located 60 and 120 cm from the ligament of Treitz, respectively. The distal mass was irregulary lobulated with some ulcerations on its surface, and tumor bleeding into the lumen was noted.

Bottom Line: Neurofibromatosis (von Recklinghausen's disease) is a neuroectodermal disorder characterized by pigmentary changes of the skin (café-au-lait spots), cutaneous and visceral tumors (neurofibromas) and systemic abnormalities.The involvement of gastrointestinal tract in neurofibromatosis is not common.The most common symptoms, refer able to lesions in the gut, are hematemesis, melena and abdominal pain.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea.

ABSTRACT
Neurofibromatosis (von Recklinghausen's disease) is a neuroectodermal disorder characterized by pigmentary changes of the skin (café-au-lait spots), cutaneous and visceral tumors (neurofibromas) and systemic abnormalities. The involvement of gastrointestinal tract in neurofibromatosis is not common. The most common symptoms, refer able to lesions in the gut, are hematemesis, melena and abdominal pain. We experienced a case of intestinal neurofibroma in von Recklinghausen's disease. The patient was a 39 year-old female who had suffered from chronic iron deficiency anemia and recurrent gastrointestinal hemorrhage due to two neurofibromas of jejunum for 3 years, which was diagnosed by superior mesenteric and ileal arteriogram and 99mTc pertechnetate-labelled RBC scan, and treated by segmental resection of jejunum with end to end anastomosis.

Show MeSH
Related in: MedlinePlus