Diffuse Idiopathic Pulmonary Neuroendocrine Hyperplasia Research Articles

DIFFUSE IDIOPATHIC PULMONARY NEUROENDOCRINE HYPERPLASIA IN A PATIENT WITH MYCOBACTERIUM AVIUM COMPLEX

TOPIC: Lung Cancer TYPE: Medical Student/Resident Case Reports INTRODUCTION: Diffuse idiopathic pulmonary neuroendocrine hyperplasia (DIPNECH) is a preneoplastic lesion that can progress to carcinoid tumors. It differs from reactive hyperplasia in its expression of tumor antigens such as Ki67 as well as the possibility of progression to carcinoid tumors. We present a case of an elderly female with a dry cough who was found to have a lung nodule showing granulomatous inflammation and a focus of neuroendocrine cell hyperplasia suggesting DIPNECH. CASE PRESENTATION: A 78-year-old female with a history of rheumatoid arthritis, breast cancer post excision and radiation therapy, presented with dry cough for a few months. She had a ten-pack-year history of smoking and quit eight years ago. She denied fevers, chills, night sweats, or acute weight changes. Chest CT and PET scan revealed a right middle lobe (RML) lung nodule with increased metabolic activity. EBUS and bronchoscopy specimens did not reveal any organisms or abnormal cytology. The patient underwent wedge resection of the RML. Pathology revealed extensive chronic inflammatory infiltrate with necrotizing granulomas and sparse foreign body giant cells, and a focus of bland appearing cells that stained positive for chromogranin and synaptophysin indicating neuroendocrine differentiation. MiB-1 proliferation index was 1%. Tissue culture grew MAC. A diagnosis of DIPNECH was established. On resection of the nodule, our patient's symptoms resolved, and she required no further therapy. On follow-up at 1.5 years, she was asymptomatic. DISCUSSION: DIPNECH is a rare lung condition presenting mainly with dry cough. CT chest can show solitary or bilateral pulmonary nodules, mosaic perfusion, bronchial wall thickening, ground-glass opacities, air trapping, or bronchiectasis. MiB-1 (antibody to Ki67) expressed in her case indicates tumor antigen expression supporting a diagnosis of DIPNECH vs. reactive hyperplasia. The miB-1 index was 1%, predicting low mitotic activity and a better prognosis. Tissue cultures of her resected specimen grew MAC. MAC is not known to be associated with such a preneoplastic condition. As per our literature review, only two other cases have been reported that describe the concurrence of pulmonary neuroendocrine growth and lung infection. MAC was associated in both these cases. Overall, most patients with DIPNECH experience a stable clinical course. However, severe clinical decline has been reported in a few. CONCLUSIONS: DIPNECH is a rare preneoplastic condition with a good prognosis, although it can sometimes progress to carcinoid tumors with clinical deterioration. Identification of risk factors may be helpful in timely diagnosis and treatment of DIPNECH. MAC is one such factor that seems to be associated with DIPNECH. Further studies should be undertaken to investigate their relationship. REFERENCE #1: Böhm J, Koch S, Gais P, Jütting U, Präuer HW, Höfler H. Prognostic value of MIB-1 in neuroendocrine tumours of the lung. J Pathol. 1996;178(4):402-409. doi:10.1002/(SICI)1096-9896(199604)178:4<402::AID-PATH498>3.0.CO;2-5 REFERENCE #2: Chassagnon G, Favelle O, Marchand-Adam S, De Muret A, Revel MP. DIPNECH: when to suggest this diagnosis on CT. Clin Radiol. 2015;70(3):317-325. doi:10.1016/j.crad.2014.10.012 REFERENCE #3: Krvavac A, Patel PH, Kamel G, Najmuddin M, Nayak RP, Patolia S. Pulmonary Neuroendocrine Cell Hyperplasia in a Patient with Mycobacterium avium Complex. Am J Respir Crit Care Med. 2018 Nov 1;198(9):e109-e110. doi: 10.1164/rccm.201801-0199IM. PMID: 30130431. DISCLOSURES: No relevant relationships by Lipisha Agarwal, source=Web Response No relevant relationships by Divyansh Bajaj, source=Web Response No relevant relationships by Chinmay Jani, source=Web Response No relevant relationships by Joseph Khoory, source=Web Response No relevant relationships by Abdullah Melhim, source=Web Response No relevant relationships by Gabriel Ryan, source=Web Response No relevant relationships by Harpreet Singh, source=Web Response

High-grade Neuroendocrine Carcinoma of the Lung With Carcinoid Morphology: A Study of 12 Cases.

Twelve lung neuroendocrine tumors with morphologic features of carcinoid tumors but with mitotic count >10/2 mm are reported. There were 7 males and 5 females, with age ranging from 56 to 78 years. Four cases were from never-smokers. All tumors showed architectural and cytomorphologic features of carcinoid tumor, including organoid nesting, insular, trabecular, or acinar growth, and tumor cells with low nucleocytoplasmic ratio, abundant cytoplasm, ovoid to round nuclei, and salt and pepper chromatin. Angulated or confluent nesting, insular or lobular growth pattern was also seen. Nuclear irregularities and anisonucleosis were focally present. Mitotic count ranged from 11 to 61/2 mm. Punctate-type necrosis was present in 8 tumors. Anaplastic cytology, large infarct-type necrosis, desmoplasia, or marked inflammatory infiltrate was not found in any of the tumors. One tumor occurred in the background of diffuse idiopathic pulmonary neuroendocrine hyperplasia. All tumors were treated by resection, and all but 1 patient subsequently developed metastasis, and 7 died of the tumor. For metastatic tumors, 4 patients were treated by platinum-based chemotherapy with no apparent response, whereas 3 other patients were treated by combined capecitabine and temozolomide-novel chemotherapy for well-differentiated neuroendocrine tumor/carcinoid tumor-2 of them responded. This subset of tumor would be classified as large cell neuroendocrine carcinoma according to the current WHO classification scheme, but their clinical and pathologic features appear to have more in common with the carcinoid tumor group than large cell neuroendocrine carcinoma, therefore, identification of this subset may be relevant for further therapeutic management.

Functional Imaging to Differentiate Pulmonary Carcinoids

TO THE EDITOR: We read with great interest the article by Kayani et al. ([1][1]). They evaluated 11 cases of typical carcinoids, 2 of atypical carcinoids, 1 of small cell neuroendocrine tumor, 1 of large cell neuroendocrine tumor, 2 of diffuse idiopathic pulmonary neuroendocrine hyperplasia, and 1

MTP22-01: Neuroendocrine tumors

MTP22-01: Neuroendocrine tumors