PMID- 16484744 OWN - NLM STAT- MEDLINE DCOM- 20060710 LR - 20190606 IS - 1349-7235 (Electronic) IS - 0918-2918 (Linking) VI - 45 IP - 2 DP - 2006 TI - Multiple endocrine neoplasia type I and Cushing's syndrome due to an aggressive ACTH producing thymic carcinoid. PG - 81-6 AB - Thymic carcinoid in multiple endocrine neoplasia type 1 (MEN 1) is previously reported as a non-ACTH producing tumor. The present case is a 39-year-old man with mortal outcome from thymic carcinoid and Cushing's syndrome with high plasma ACTH. The symptom was first observed at age 29 and was relieved after extended thymectomy, with reduction of ACTH level. The tumor was positive for ACTH, Grimelius silver staining and Chromogranin A. The finding of primary hyperparathyroidism, pituitary adenoma, and a novel germline nonsense mutation (W423X) established the diagnosis of MEN 1. Cushing's syndrome due to ACTH producing thymic carcinoid should be also considered as one phenotype of the MEN 1 spectrum. FAU - Takagi, Junko AU - Takagi J AD - Department of Laboratory Medicine, Aichi Medical University School of Medicine, Nagakute, Aichi. FAU - Otake, Kazuo AU - Otake K FAU - Morishita, Munehiko AU - Morishita M FAU - Kato, Harumichi AU - Kato H FAU - Nakao, Naoki AU - Nakao N FAU - Yoshikawa, Kazuhiro AU - Yoshikawa K FAU - Ikeda, Hiroshi AU - Ikeda H FAU - Hirooka, Yoshifumi AU - Hirooka Y FAU - Hattori, Yoshinobu AU - Hattori Y FAU - Larsson, Catharina AU - Larsson C FAU - Nogimori, Tsuyoshi AU - Nogimori T LA - eng PT - Case Reports PT - Journal Article DEP - 20060215 PL - Japan TA - Intern Med JT - Internal medicine (Tokyo, Japan) JID - 9204241 RN - 0 (Antineoplastic Agents, Hormonal) RN - 9002-60-2 (Adrenocorticotropic Hormone) RN - RWM8CCW8GP (Octreotide) SB - IM MH - ACTH Syndrome, Ectopic/blood/*etiology MH - Adrenocorticotropic Hormone/blood MH - Adult MH - Antineoplastic Agents, Hormonal/therapeutic use MH - Antineoplastic Combined Chemotherapy Protocols/administration & dosage MH - Carcinoid Tumor/blood/*complications/pathology/therapy MH - Combined Modality Therapy MH - Cushing Syndrome/blood/*etiology MH - Fatal Outcome MH - Germ-Line Mutation MH - Humans MH - Male MH - Multiple Endocrine Neoplasia Type 1/blood/*diagnosis/genetics/therapy MH - Octreotide/therapeutic use MH - Radiotherapy, Adjuvant MH - Thymectomy MH - Thymus Neoplasms/blood/*complications/pathology/therapy EDAT- 2006/02/18 09:00 MHDA- 2006/07/13 09:00 CRDT- 2006/02/18 09:00 PHST- 2006/02/18 09:00 [pubmed] PHST- 2006/07/13 09:00 [medline] PHST- 2006/02/18 09:00 [entrez] AID - JST.JSTAGE/internalmedicine/45.1427 [pii] AID - 10.2169/internalmedicine.45.1427 [doi] PST - ppublish SO - Intern Med. 2006;45(2):81-6. doi: 10.2169/internalmedicine.45.1427. Epub 2006 Feb 15.