SMALL INTESTINAL GANGLIONEUROMATOSIS IN A PATIENT WITH NEUROFIBROMATOSIS TYPE 1: A CASE REPORT
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- KIDA Yuichi
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
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- SAWADA Tsunaki
- Department of Endoscopy, Nagoya University Hospital.
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- ISHIKAWA Eri
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
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- SAKAKIBARA Ayako
- Department of Pathology and Laboratory Medicine, Nagoya University Hospital.
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- YAMAMURA Takeshi
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
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- MAEDA Keiko
- Department of Endoscopy, Nagoya University Hospital.
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- ESAKI Masaya
- Department of Endoscopy, Nagoya University Hospital.
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- HAMAZAKI Motonobu
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
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- MURATE Kentaro
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
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- NAKAMURA Masanao
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine.
Bibliographic Information
- Other Title
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- 小腸内視鏡下生検により診断した神経線維腫症1型に合併した腸管diffuse ganglioneuromatosisの1例
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Abstract
<p>A 50-year-old woman with neurofibromatosis type 1 who hospitalized for chronic diarrhea was transferred to our hospital for evaluation of small intestinal dilatation and thickening. Transoral double-balloon enteroscopy revealed jejunal dilatation and suppressed peristalsis; however, mucosal inflammation, such as ulcers or erosions were not detected. Transanal double-balloon enteroscopy detected the intestinal stenosis with inflammatory polyps and a longitudinal ulcer. Histopathological evaluation of duodenal, jejunal, and ileal specimens by biopsy revealed ganglion cells and Schwannian cells; therefore, the patient was diagnosed with ganglioneuromatosis with neurofibromatosis type 1. Small intestinal dilatation was associated with suppressed peristalsis caused by ganglioneuromatosis and was diagnosed as secondary chronic intestinal pseudo-obstruction. Abdominal distention persisted despite conservative therapy. However, she remained asymptomatic, and oral intake remained unaffected. Ganglioneuromatosis is rare; however, clinicians should be mindful that ganglioneuromatosis is an abdominal complication associated with systemic disease such as neurofibromatosis type 1 and multiple endocrine neoplasia type 2B.</p>
Journal
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- GASTROENTEROLOGICAL ENDOSCOPY
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GASTROENTEROLOGICAL ENDOSCOPY 65 (7), 1232-1238, 2023
Japan Gastroenterological Endoscopy Society
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Keywords
Details 詳細情報について
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- CRID
- 1390296829542492032
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- ISSN
- 18845738
- 03871207
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- Text Lang
- ja
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- Data Source
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- JaLC
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- Abstract License Flag
- Disallowed