PMID- 12803235
OWN - NLM
STAT- MEDLINE
DCOM- 20040115
LR  - 20190906
IS  - 0918-8959 (Print)
IS  - 0918-8959 (Linking)
VI  - 50
IP  - 2
DP  - 2003 Apr
TI  - Cushing's syndrome due to bilateral adrenocortical adenomas with unique 
      histological features.
PG  - 155-62
AB  - Cushing's syndrome due to bilateral cortisol-secreting adenomas rarely occurs. We 
      present a case of Cushing's syndrome due to bilateral adenomas. Both adenomas had 
      distinct cell compositions, and were compared with emphasis on 
      immunohistochemical and enzyme histochemical analysis for cytochrome P450(11beta) 
      and 3beta-hydroxysteroid dehydrogenase (3betaHSD). A 37 year-old female was 
      diagnosed with ACTH-independent Cushing's syndrome based on physical findings and 
      hormonal evaluation. High-resolution CT scan showed bilateral adrenocortical 
      adenomas and atrophied glands. 131I-methylnorcholesterol incorporation into both 
      glands suggested both adenomas were functional. Clinical diagnosis prior to 
      surgery was ACTH-independent Cushing's syndrome due to functioning bilateral 
      adenomas. The left adrenal gland was totally resected, while the right one was 
      partially resected by laparoscopic approach. Both adenomas were black on cut 
      sections, and were comparatively evaluated by immunohistochemical and enzyme 
      histochemical analysis for P450(11beta) and 3betaSD. The left adenoma was 1.6 cm 
      in diameter and had a complex cellular composition and enzyme expression similar 
      to that of primary pigmented nodular adrenocortical disease (PPNAD), while the 
      right adenoma was 1.8 cm in diameter with compact cells typical of a solitary 
      cortisol-producing adenoma. Adjacent bilateral adrenal cortex showed marked 
      atrophy, but contained several micronodules. Serum cortisol levels, both at basal 
      and after a low dodexamethasone, normalized thirteen months after surgery. In 
      conclusion, the present case of Cushing's syndrome with bilateral adrenal 
      adenomas demonstrated for the first time the simultaneous occurrence of two 
      distinct adenomas, an ordinary cortisol-producing adenoma and a PPNAD-like 
      adenoma. Further case reports of multiple adrenal adenomas should be 
      well-analyzed to clarify whether the results from this case represent a new 
      subgroup of ACTH-independent Cushing's syndrome.
FAU - Nomura, Kaoru
AU  - Nomura K
AD  - Department of Medicine, Institute of Clinical Endocrinology, Tokyo Women's 
      Medical University, School of Medicine, Tokyo 162-8666, Japan.
FAU - Saito, Hiroshi
AU  - Saito H
FAU - Aiba, Motohiko
AU  - Aiba M
FAU - Iihara, Masatoshi
AU  - Iihara M
FAU - Obara, Takao
AU  - Obara T
FAU - Takano, Kazue
AU  - Takano K
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - Japan
TA  - Endocr J
JT  - Endocrine journal
JID - 9313485
RN  - 0 (Glucocorticoids)
RN  - 7S5I7G3JQL (Dexamethasone)
RN  - WI4X0X7BPJ (Hydrocortisone)
SB  - IM
MH  - Adrenal Cortex Diseases/pathology
MH  - Adrenal Cortex Neoplasms/*complications/metabolism/*pathology/surgery
MH  - Adrenalectomy
MH  - Adrenocortical Adenoma/*complications/metabolism/*pathology/surgery
MH  - Adult
MH  - Cushing Syndrome/*etiology
MH  - Dexamethasone
MH  - Female
MH  - Glucocorticoids
MH  - Histocytochemistry
MH  - Humans
MH  - Hydrocortisone/metabolism
MH  - Immunohistochemistry
MH  - Neoplasms, Second Primary/*complications/metabolism/pathology/surgery
MH  - Radionuclide Imaging
MH  - Tomography, X-Ray Computed
EDAT- 2003/06/14 05:00
MHDA- 2004/01/16 05:00
CRDT- 2003/06/14 05:00
PHST- 2003/06/14 05:00 [pubmed]
PHST- 2004/01/16 05:00 [medline]
PHST- 2003/06/14 05:00 [entrez]
AID - 10.1507/endocrj.50.155 [doi]
PST - ppublish
SO  - Endocr J. 2003 Apr;50(2):155-62. doi: 10.1507/endocrj.50.155.