Pseudoprecocious puberty and gynaecomastia as presenting features of Peutz-Jeghers syndrome

BMJ Case Rep. 2025 Jan 21;18(1):e262022. doi: 10.1136/bcr-2024-262022.

Abstract

A boy in mid-childhood presented with right-sided gynaecomastia, which was excised. He represented and, on review by endocrinology, Tanner staging showed stage 2 left-sided glandular breast tissue and some features of virilisation. His testicular volumes remained prepubertal (3 mL). Testicular ultrasound demonstrated irregular echogenic foci bilaterally, and mucosal freckling was noted. A genetic panel confirmed a de novo STK11 variant, pathogenic for Peutz-Jeghers syndrome (PJS). Aromatase inhibition was chosen in preference to left-sided mastectomy. His growth velocity and bone age stabilised, and left-sided breast budding reduced in size. He continues to have a good cosmetic outcome with preserved height potential 5 years later. In this case, to date, treatment with an aromatase inhibitor has negated the need for further surgery. Pseudopuberty in boys with PJS can lead to a diagnostic and treatment challenge. We present a recent case and review the evolving treatment standards and diagnostic strategies.

Keywords: Drugs: endocrine system; Endocrine system; Gynecomastia; Paediatrics (drugs and medicines).

Publication types

  • Case Reports

MeSH terms

  • AMP-Activated Protein Kinase Kinases
  • Aromatase Inhibitors / therapeutic use
  • Child
  • Gynecomastia* / diagnosis
  • Gynecomastia* / etiology
  • Humans
  • Male
  • Peutz-Jeghers Syndrome* / complications
  • Peutz-Jeghers Syndrome* / diagnosis
  • Protein Serine-Threonine Kinases / genetics
  • Puberty
  • Puberty, Precocious / diagnosis
  • Puberty, Precocious / etiology

Substances

  • STK11 protein, human
  • AMP-Activated Protein Kinase Kinases
  • Aromatase Inhibitors
  • Protein Serine-Threonine Kinases