Complexities of complete androgen insensitivity syndrome: insights from a case report and literature review

J Int Med Res. 2024 Nov;52(11):3000605241300058. doi: 10.1177/03000605241300058.

Abstract

Complete androgen insensitivity syndrome (CAIS) presents significant challenges in the accurate diagnosis and personalized management of individuals with a 46, XY karyotype who exhibit a female phenotype due to complete insensitivity to androgens. This retrospective case report analyzes the clinical data, genetic testing, hormonal profiling, and imaging studies of a patient who was initially misdiagnosed during hernioplasty and later misidentified as having Mayer-Rokitansky-Küster-Hauser syndrome. The report details the establishment of the correct diagnosis and implementation of a personalized management strategy that postponed gonadectomy until post-puberty. This approach included continuous monitoring and tailored estrogen replacement therapy, which facilitated informed patient decisions and comprehensive feminization while preventing the long-term consequences of estrogen deficiency. Supported by a literature review, this case report emphasizes the necessity of a multidisciplinary approach to managing CAIS, highlighting the importance of heightened awareness, accurate diagnostics, and personalized therapeutic plans to ensure holistic, patient-centered care.

Keywords: 46; Androgen receptor insensitivity; Complete androgen insensitivity syndrome (CAIS); Estrogen replacement therapy; Gonadectomy; Mayer-Rokitansky-Küster-Hauser syndrome; Sexual development disorders; XY karyotype.

Publication types

  • Case Reports
  • Review

MeSH terms

  • 46, XX Disorders of Sex Development / diagnosis
  • 46, XX Disorders of Sex Development / genetics
  • Androgen-Insensitivity Syndrome* / diagnosis
  • Androgen-Insensitivity Syndrome* / genetics
  • Congenital Abnormalities / diagnosis
  • Congenital Abnormalities / genetics
  • Congenital Abnormalities / pathology
  • Female
  • Humans
  • Mullerian Ducts / abnormalities
  • Mullerian Ducts / surgery

Supplementary concepts

  • Mullerian aplasia