Paraneoplastic Cushing syndrome due to adrenal neuroblastoma

Espinasse-Holder, M.; Defachelles, Anne‐Sophie; Weill, Jacques; Keyzer, Yves de; Lasalle, E. Martin de; Nelken, Brigitte

doi:10.1002/(sici)1096-911x(200003)34:3<231::aid-mpo17>3.0.co;2-w

Cited by 14 publications

(12 citation statements)

References 20 publications

(22 reference statements)

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“…As for the ectopic hormone production by olfactory neuroblastoma, three cases with syndromes of inappropriate secretion of antidiuretic hormone have been reported [11][12][13]. Although four cases of ectopic ACTH-producing adrenal neuroblastoma were reported in infants [14][15][16][17], only one case with ectopic ACTH-producing olfactory neuroblastoma in adulthood has been reported thus far [5]. Thus, our case adds to the literature a second rare case report of ectopic ACTH-producing olfactory neuroblastoma.…”

Section: Discussionmentioning

confidence: 68%

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Olfactory Neuroblastoma Causing Ectopic ACTH Syndrome

et al. 2005

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Abstract.A 39-year-old woman who presented with typical Cushingoid appearance (moon facies, central obesity, purpura) was admitted to our hospital because of pulmonary infection. She was found to have hypertension, severe hypokalemia, and metabolic alkalosis. Endocrine data revealed elevated plasma levels of ACTH and cortisol with lack of circadian rhythm, non-suppressibility to high-dose dexamethasone, and hyperresponsiveness to CRH stimulation. Although no pituitary mass was detected by MRI of the brain, inferior petrosal sinus sampling showed a step-up of central to peripheral ACTH levels; these data are consistent with the diagnosis of Cushing's disease. She was successfully treated with metyrapone to control hypercortisolemia. Ten months later, a mass was detected in the ethmoid sinus, which was surgically removed. After resection of the ethmoid sinus tumor, her Cushingoid features and hypercortisolemia disappeared, but recurred after enlargement of a second mass in the maxillary sinus. After resection of the maxillary sinus tumor, her hypercortisolemia subsided. Histologically, the tumor tissues from both the ethmoid and maxillary sinus were identical and consistent with the diagnosis of olfactory neuroblastoma. Immunohistochemically, the immunoreactivities of ACTH and POMC were positive in the cytoplasm of tumor cells, and immunoreactive ACTH was demonstrated in both tumor tissues. Thus, this is the second rare case with ectopic ACTH syndrome caused by olfactory neuroblastoma thus far reported.

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Section: Discussionmentioning

confidence: 68%

“…Plasma and tissue ACTH concentrations were determined by immunoradiometric assay (IRMA) using Yuka ACTH IRMA kit (Mitsubishi Yuka, Tokyo, Japan) which showed full crossreactivity with ACTH (1-39), but none with ACTH fragments (1-13), , (11)(12)(13)(14)(15)(16)(17)(18)(19)(20)(21)(22)(23)(24), , b-lipotropin or b-endorphin [6].…”

Section: Acth Measurementmentioning

confidence: 99%

Olfactory Neuroblastoma Causing Ectopic ACTH Syndrome

et al. 2005

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“…POMC produced by neoplasms other than pituitary tumors is reported to be processed to smaller-sized ACTH precursors than normal ACTH, which can cause the clinical feature of EAS despite their lower biological activity [21]. Interestingly, one report of an adrenal neuroblastoma producing increased levels of POMC did not show any increased ACTH level [22]. In addition, by Raux Demay et al, an ACTH-producing thymic carcinoid tumor was found to be responsible for the Cushing's syndrome [23].…”

Section: Discussionmentioning

confidence: 99%

Two Cases of Ectopic Adrenocorticotropic Hormone Syndrome with Olfactory Neuroblastoma and Literature Review

Koo

Jeon

et al. 2008

Endocr J

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are equally contributed to this journal.Abstract. Olfactory neuroblastomas are rare, slow-growing malignant tumors, usually diagnosed at advanced stages. Ectopic adrenocorticotropic hormone (ACTH) syndrome caused by an olfactory neuroblastoma is extremely rare. We reported two Korean women who suffered from ectopic ACTH syndrome (EAS) caused by olfactory neuroblastomas. The first patient was a 66-year-old woman who had been diagnosed as olfactory neuroblastoma and refused the management two years before and the second patient was a 37-year-old woman on chemotherapy for olfactory neuroblastoma. In the first case, she presented the Cushingoid appearance with systemic edema and her tumor was removed surgically. ACTH secretion by the tissue was confirmed by immunohistochemistry. By contrast, the second patient presented as severe pneumonia caused by cytomegalovirus and was treated with anti-viral agent followed by chemotherapy and radiotherapy, and her residual mass remained. However, after treatment, both patients' plasma ACTH and cortisol levels returned to normal without any adrenolytic therapy. Considering the causative tumors of EAS can be rarely cured and EAS increases the susceptibility to infections, it is prudent to suppress any hypercortisolemia initially, apart from treating the causal malignancy.

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“…In the pediatric population, a variety of neoplasm, including neuroblastoma [5], Ewing's sarcoma [6], malignant carcinoid tumor of the lungs [7], and thymic carcinoid tumor [8], produce one kind of cortisol, ACTH and CRH, triggering the occurrence of Cushing syndrome. Based on a survey of the literature, ACTH secretion is the most frequent cause [7,8], and CRH secretion the rarest [6].…”

Section: Discussionmentioning

confidence: 99%

Paraneoplastic Cushing syndrome because of corticotrophin-releasing hormone–secreting Wilms' tumor

Zhang¹

2008

Journal of Pediatric Surgery

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Paraneoplastic Cushing syndrome due to adrenal neuroblastoma

Cited by 14 publications

References 20 publications

Olfactory Neuroblastoma Causing Ectopic ACTH Syndrome

Olfactory Neuroblastoma Causing Ectopic ACTH Syndrome

Two Cases of Ectopic Adrenocorticotropic Hormone Syndrome with Olfactory Neuroblastoma and Literature Review

Paraneoplastic Cushing syndrome because of corticotrophin-releasing hormone–secreting Wilms' tumor

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