The Effects of Sampling Lateralization on Bilateral Inferior Petrosal Sinus Sampling for Pediatric Cushing's Disease-A Single Endocrinology Centre Experience and Review of the Literature

Front Endocrinol (Lausanne). 2021 Apr 19:12:650967. doi: 10.3389/fendo.2021.650967. eCollection 2021.

Abstract

Background: This study aims to analyze the diagnostic accuracy of bilateral inferior petrosal sinus sampling (BIPSS), the gold standard test for the differential diagnosis of ACTH-dependent Cushing's syndrome (CS) in a group of pediatric patients with Cushing's disease (CD).

Methods: This is a retrospective analysis which include 12 patients with hypercortisolemia and inconclusive pituitary MRI, who underwent bilateral inferior petrosal sinus sampling (BIPSS) and transsphenoidal surgery (TSS) from 2004 to 2020 in the Children's Memorial Health Institute (CMHI) Warsaw, Poland. Pituitary origin of ACTH secretion was considered if baseline central to peripheral (C/P) ACTH level ratio was ≥ 2 or C/P ratio was ≥ 3 after human corticotropin-releasing hormone (hCRH) stimulation. The diagnosis was histologically confirmed in almost all cases after TSS.

Results: The diagnostic accuracy of BIPSS reached 75% at baseline and 83.3% after CRH stimulation. The compatibility of localization of a microadenoma by BIPSS with the surgical location was 66.7%.

Conclusions: Owing to its high diagnostic effectiveness, BIPSS remains the best test to differentiate CD from EAS. The indications for the procedure should be carefully considered, because EAS in the pediatric population, unlike in adults, is extremely rare. Moreover BIPSS has only limited value for indicating tumor localization.

Keywords: BIPSS; Cushing’s disease; adenoma; hypercortisolemia; lateralization; pituitary adenoma.

MeSH terms

  • Adenoma / diagnosis
  • Adolescent
  • Adrenocorticotropic Hormone / metabolism*
  • Child
  • Corticotropin-Releasing Hormone / blood
  • Diagnosis, Differential
  • Endocrine System
  • Endocrinology / methods*
  • Female
  • Follow-Up Studies
  • Humans
  • Longitudinal Studies
  • Magnetic Resonance Imaging
  • Male
  • Petrosal Sinus Sampling / adverse effects*
  • Petrosal Sinus Sampling / methods
  • Pituitary ACTH Hypersecretion / diagnosis*
  • Pituitary Gland / diagnostic imaging
  • Pituitary Neoplasms / diagnosis*
  • Poland
  • Reproducibility of Results
  • Retrospective Studies
  • Time Factors
  • Tomography, X-Ray Computed

Substances

  • Adrenocorticotropic Hormone
  • Corticotropin-Releasing Hormone