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The University of Chicago Chicago, IL 60637
In the well-written and interesting case report "Appendiceal Ganglioneuromas and Pheochromocytoma in Neurofibromatosis Type I," Lockhart et al. [1] comment that since 1974 only five cases of neurofibromatosis with gastrointestinal ganglioneuromas have been reported. The authors neglected to quote the largest reported series describing the CT findings in adults with neurofibromatosis-1 by Tonsgard et al. [2]. In this series we documented the findings on CT of chest, abdomen, and pelvis in 91 of 125 consecutive adults with neurofibromatosis-1. We found three patients with isolated bowel neurofibromas. Four adrenal tumors were also reported: three pheochromocytomas and one malignant adrenal tumor. A wealth of other information valuable to radiologists can be found in this publication, particularly the often unrecognized fact that plexiform neurofibromas can appear as fat infiltration alone, particularly in the perirectal area.
References
University of Alabama at Birmingham Birmingham, AL 35249
We would like to thank Dr. Dachman for his interest in our case report [1]. Indeed, the article by Tonsgard et al. [2] provides an excellent description of the CT findings of neurofibromatosis in a large series of patients. In response to Dr. Dachman's concerns, two points merit discussion. First, our case report describes a patient with ganglioneuromas in association with neurofibromatosis. On the basis of discussion with our pathologists, we realized that there is a difference between ganglioneuromas and neurofibromas in these patients. Because the lesions in the three patients described in the article by Tonsgard et al. were neurofibromas, we did not incorporate them into the "Discussion" in our report. The distinction between ganglioneuromas and neurofibromas of the bowel is supported by Pathology of Tumors of the Nervous System [3], which was cited in our case report. The text suggests that visceral ganglioneuromatosis is an entity often associated with neurofibromatosis, but ganglioneuromas may also occur in patients without neurofibromatosis. Whether the pathologic distinction is important in the radiology literature is not certain, but this distinction was the reason we chose not to include all bowel fibromas in our report.
Secondly, although the article by Tonsgard et al. [2] is an excellent reference, a complete discussion of the literature is not possible within the constraints required by AJR for case reports. Many good references on neurofibromatosis could not be included. However, we did try to include several well-referenced articles as part of our brief discussion. We hope that our case report encourages the readership to delve more deeply into this interesting disease, and we have Dr. Dachman to thank as the catalyst.
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