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Resolution of Cyclicity After Pasireotide LAR in a Patient With Cushing Disease.

Authors
  • Machado, Márcio Carlos1, 2, 3
  • Cescato, Valter Angelo Sperling4
  • Fragoso, Maria Candida Barisson Villares...5
  • Bronstein, Marcello Delano1, 2
  • 1 Neuroendocrine Unit, Division of Endocrinology and Metabolism, University of Sao Paulo Medical School, Sao Paulo, SP, Brazil. , (Brazil)
  • 2 Laboratory of Cellular and Molecular Endocrinology/LIM25, University of Sao Paulo Medical School, Sao Paulo, SP, Brazil. , (Brazil)
  • 3 Endocrinology Service, AC Camargo Cancer Center, Sao Paulo, SP, Brazil. , (Brazil)
  • 4 Neurosurgery Service, University of Sao Paulo Medical School, Sao Paulo, SP, Brazil. , (Brazil)
  • 5 Adrenal Unit, Division of Endocrinology and Metabolism, University of Sao Paulo Medical School, Sao Paulo, SP, Brazil. , (Brazil)
Type
Published Article
Journal
AACE Clinical Case Reports
Publisher
American Association of Clinical Endocrinologists
Publication Date
Jan 01, 2021
Volume
7
Issue
4
Pages
277–281
Identifiers
DOI: 10.1016/j.aace.2021.02.007
PMID: 34307853
Source
Medline
Keywords
Language
English
License
Unknown

Abstract

The cyclicity (CIC) of cortisol spontaneously occurs in a minority of patients with Cushing syndrome (CS). When it arises, diagnostic and therapeutic approaches become more challenging. This study aimed to report a patient with Cushing disease (CD) who achieved normalization of cortisol and CIC pattern with pasireotide long-acting release (pasi/LAR). A 43-year-old female patient related an 8-month history of CS. An 8-mm pituitary nodule depicted by magnetic resonance imaging, serum cortisol suppression of >50% after 8 mg of dexamethasone therapy, and the absence of other lesions were compatible with a CD diagnosis. The patient presented with a CIC pattern with 1 episode before and 17 episodes after an unsuccessful pituitary surgery. Medical treatment with cabergoline alone up to 3.5 mg/wk and a combined treatment with ketoconazole 400 mg/d did not improve CIC CS. Pasi/LAR was initiated at a dose of 20 mg/mo. A few days after the first dose, the patient experienced symptoms suggestive of adrenal insufficiency. The medication and dose were maintained for 24 months. During this period, there was a normalization of UFC levels and progressive clinical improvement. Additionally, new episodes of CIC were not observed. A CD patient with a challenging issue of CIC was reported. The condition was not controlled after pituitary surgery and by the combined treatment with cabergoline and ketoconazole, although hypercortisolism was abated by the continuous use of pasi/LAR. To our knowledge, this is the first report as regards the use of this medication to control CIC in a patient with CD. © 2021 AACE. Published by Elsevier Inc.

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