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Olfactory neuroblastoma causing ectopic ACTH syndrome.

Kazuo Kanno1, Yuki Morokuma, Toru Tateno

  • 1Division of Endocrinology and Metabolism, Musashino Red Cross Hospital, Tokyo.

Endocrine Journal
|January 18, 2006
PubMed
Summary

This study reports a rare case of ectopic ACTH syndrome caused by olfactory neuroblastoma. Surgical removal of sinus tumors resolved Cushingoid symptoms, highlighting this tumor type as a potential cause of ACTH hypersecretion.

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Area of Science:

  • Endocrinology
  • Oncology
  • Neurosurgery

Background:

  • Cushing's syndrome is characterized by hypercortisolemia, often due to pituitary adenomas or adrenal tumors.
  • Ectopic ACTH syndrome, a less common cause, arises from non-pituitary tumors producing ACTH.
  • Olfactory neuroblastoma is a rare malignancy typically affecting the nasal cavity.

Observation:

  • A patient presented with Cushingoid appearance, hypertension, and hypokalemia, initially suspected as Cushing's disease.
  • Despite negative pituitary imaging, invasive testing confirmed ACTH-dependent Cushing's syndrome.
  • Olfactory neuroblastoma tumors in the ethmoid and maxillary sinuses were identified as the source of ectopic ACTH production.

Findings:

  • Tumor tissues showed positive ACTH and POMC immunoreactivity, confirming ectopic hormone production.

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  • Surgical resection of the olfactory neuroblastomas led to the resolution of hypercortisolemia.
  • Recurrence of symptoms correlated with tumor regrowth, resolving again after subsequent resection.

    • Implications:

    • Olfactory neuroblastoma should be considered in the differential diagnosis of ectopic ACTH syndrome.
    • This case underscores the importance of thorough investigation for ectopic sources of ACTH in atypical Cushing's syndrome presentations.
    • Successful management relies on accurate diagnosis and surgical intervention for sinonasal tumors causing ectopic ACTH syndrome.