Publication:
Gynecomastia and Its Management In Boys With Partial Androgen Insensitivity Syndrome

Received Date

2024

Issued Date

2024

Resource Type

Research Article

Language

eng

File Type

application/pdf

No. of Pages/File Size

8 pages

DOI

10.1210/clinem/dgae562

Journal Title

The Journal of Clinical Endocrinology & Metabolism

Access Rights

open access

Rights

ผลงานนี้เผยแพร่ภายใต้ สัญญาอนุญาตครีเอทีฟคอมมอนส์แบบ แสดงที่มา-ไม่ใช้เพื่อการค้า-อนุญาตแบบเดียวกัน 4.0 (CC BY-NC-SA 4.0)

Rights Holder(s)

Oxford University Press on behalf of the Endocrine Society

Suggested Citation

Supitcha Patjamontri, Angela K. Lucas-Herald, Jillian Bryce, Erica van den Akker, Martine Cools, Evgenia Globa, Gil Guerra-Junior, Olaf Hiort, Paul Hofman, Paul-Martin Holterhus, Ieuan A. Hughes, Anders Juul, Anna Nordenstrom, Gianni Russo, Marianna R. Stancampiano, Sumudu N. Seneviratne, Rieko Tadokoro-Cuccaro, Ajay Thankamony, Naomi Weintrob, Natalia Zelinska, S. Faisal Ahmed (2024). Gynecomastia and Its Management In Boys With Partial Androgen Insensitivity Syndrome. The Journal of Clinical Endocrinology & Metabolism, doi:10.1210/clinem/dgae562 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/101157

Research Projects

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Thesis

Title

Gynecomastia and Its Management In Boys With Partial Androgen Insensitivity Syndrome

Author(s)

Supitcha Patjamontri
 Angela K. Lucas-Herald
 Jillian Bryce
 Erica van den Akker
 Martine Cools
 Evgenia Globa
 Gil Guerra-Junior
 Olaf Hiort
 Paul Hofman
 Paul-Martin Holterhus
 Ieuan A. Hughes
 Anders Juul
 Anna Nordenstrom
 Gianni Russo
 Marianna R. Stancampiano
 Sumudu N. Seneviratne
 Rieko Tadokoro-Cuccaro
 Ajay Thankamony
 Naomi Weintrob
 Natalia Zelinska
 S. Faisal Ahmed

Author's Affiliation

Mahidol University. Faculty of Medicine Siriraj Hospital. Division of Endocrinology and Metabolism

Abstract

Introduction: Partial androgen insensitivity syndrome (PAIS) is a rare condition that is reported to be commonly associated with gynecomastia in males. Objectives: To assess the management of gynecomastia in male PAIS. Materials and Methods: Retrospective review of males with PAIS over the age of 10 years in the I-DSD registry. Results: Of the 205 eligible cases, information was available for 57 from 13 centers. An androgen receptor gene variant was confirmed in 45 (79%) with a median age at first presentation of 1.0 year (range 0.1, 26.0). Of the 45 genetically confirmed cases, gynecomastia was present in 41 (91%) with a median age at the time of gynecomastia development of 13.5 years (11.0, 29.0). In the other 4 (9%) with no gynecomastia, the median age at last assessment was 15.7 years (10.6, 17.0). In 30 cases with information available, micropenis was present at the time of gynecomastia development in 23 (77%). Of the 35 with information available, 2 (6%) exhibited spontaneous resolution between the ages of 15 and 21 years and 25 (71%) had breast surgery at a median age of 15.7 years (14.0, 23.0). Of these 25, 9 (26%) had previously received medical therapy. The median clinician score of effectiveness for medical therapy was 3 (1, 8) compared to 10 (3, 10) for surgery (P < .0001). In 31 with information available, 13 (42%) had received psychology support. Conclusion: Gynecomastia is common in PAIS but not universal. Surgical management may be more effective than medical therapy, but there is a need for further standardized and systematic studies.

Sponsorship

Faculty of Medicine Siriraj Hospital, Mahidol University
The development of the I-DSD Registry was originally funded by the Medical Research Council UK (G1100236)
The Seventh European Union Framework Programme (201444)
The European Society for Paediatric Endocrinology.

Keyword(s)

Androgen insensitivity syndrome
 Gynecomastia
 Disorders of sex development
 I-DSD registry

Availability

URI

https://repository.li.mahidol.ac.th/handle/20.500.14594/101157

Collections

SI-Article