naa a22 4500 510773524 CHVBK 20180411083222.0 cr unu---uuuuu 180411e20131201xx s 000 0 eng 10.1007/s12022-013-9265-8 doi (NATIONALLICENCE)springer-10.1007/s12022-013-9265-8 Diffuse Idiopathic Pulmonary Neuroendocrine Cell Hyperplasia with a Central and Peripheral Carcinoid and Multiple Tumorlets: A Case Report Emphasizing the Role of Neuropeptide Hormones and Human Gonadotropin-Alpha [Elektronische Daten] [Hanako Oba, Kazunori Nishida, Shingo Takeuchi, Hirohiko Akiyama, Koji Muramatsu, Masafumi Kurosumi, Toru Kameya] We report a case of diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNECH). We performed immunohistochemical analysis of 17 neuropeptides and human gonadotropin-alpha (hCGα), a trophoblastic peptide that promotes the proliferation of neuroendocrine cells. A 51-year-old woman with no history of smoking was found to have a nodule in the right middle lobe. Upon examination, the nodule was found to comprise diffuse linear and nodular neuroendocrine cell hyperplasia (NECH), numerous pulmonary tumorlets merging with one peripheral carcinoid, and an additional central carcinoid. Immunohistochemical analysis revealed diffuse but intense expression of the general neuroendocrine markers CD56, synaptophysin, and chromogranin A, together with gastrin-releasing peptide (GRP), calcitonin, and hCGα throughout the carcinoids, tumorlets, and NECH. Positive staining was also noted for adrenocorticotropic hormone, corticotropin-releasing hormone, met-enkephalin, vasoactive intestinal polypeptide, neurotensin, and growth hormone-releasing hormone in a few isolated cells of the carcinoids and the tumorlets, but staining for these proteins was entirely negative in the NECH lesions. The presence of these neuropeptides in neuroendocrine tumors might explain the presence of neuropeptide-producing tumors of the lungs, cases of which have been reported over the last 30years. The preoperative serum proGRP level was high but returned to normal after surgical intervention, indicating that GRP was produced and secreted by carcinoids, tumorlets, and/or NECH lesions. It is also probable that neuroendocrine cells secreted GRP into the interstitium in a paracrine manner, leading to the development of dense fibrosis around the tumorlets. During the preoperative and postoperative periods, no evidence of bronchiolitis obliterans was noted, in contrast to some previously reported cases of DIPNECH. Springer Science+Business Media New York, 2013 Lung tumor nationallicence Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNECH) nationallicence Pulmonary neuroendocrine cell hyperplasia (PNECH) nationallicence Tumorlet nationallicence Carcinoid tumor nationallicence Neuropeptides nationallicence Oba Hanako Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut Nishida Kazunori Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut Takeuchi Shingo Division of Thoracic Surgery, Department of Surgery, Nippon Medical School, Tokyo, Japan aut Akiyama Hirohiko Department of Thoracic Surgery, Saitama Cancer Center, Saitama, Japan aut Muramatsu Koji Pathology Division, Shizuoka Cancer Center Hospital and Research Institute, Shizuoka, Japan aut Kurosumi Masafumi Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut Kameya Toru Pathology Division, Shizuoka Cancer Center Hospital and Research Institute, Shizuoka, Japan aut Endocrine Pathology Springer US; http://www.springer-ny.com 24/4(2013-12-01), 220-228 1046-3976 24:4<220 2013 24 12022 https://doi.org/10.1007/s12022-013-9265-8 text/html Onlinezugriff via DOI 1 research-article jats NATIONALLICENCE 856 40 https://doi.org/10.1007/s12022-013-9265-8 text/html Onlinezugriff via DOI NATIONALLICENCE 700 1- Oba Hanako Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut NATIONALLICENCE 700 1- Nishida Kazunori Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut NATIONALLICENCE 700 1- Takeuchi Shingo Division of Thoracic Surgery, Department of Surgery, Nippon Medical School, Tokyo, Japan aut NATIONALLICENCE 700 1- Akiyama Hirohiko Department of Thoracic Surgery, Saitama Cancer Center, Saitama, Japan aut NATIONALLICENCE 700 1- Muramatsu Koji Pathology Division, Shizuoka Cancer Center Hospital and Research Institute, Shizuoka, Japan aut NATIONALLICENCE 700 1- Kurosumi Masafumi Department of Pathology, Saitama Cancer Center, 818, Komuro, Ina, Kita-adachi, 362-0806, Saitama, Japan aut NATIONALLICENCE 700 1- Kameya Toru Pathology Division, Shizuoka Cancer Center Hospital and Research Institute, Shizuoka, Japan aut NATIONALLICENCE 773 0- Endocrine Pathology Springer US; http://www.springer-ny.com 24/4(2013-12-01), 220-228 1046-3976 24:4<220 2013 24 12022 Metadata rights reserved Springer special CC-BY-NC licence nationallicence BK010053 XK010053 XK010000 NATIONALLICENCE NATIONALLICENCE NL-springer