Precocious Puberty and Large Multicystic Ovaries in Young Girls with Primary Hypothyroidism |
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| Affiliation: | 1. The Urogenital Sub-committee and the Surveillance Committee of Japanese Society of Chemotherapy (JSC), The Japanese Association for Infectious Diseases (JAID) and The Japanese Society for Clinical Microbiology (JSCM), Tokyo, Japan;2. The Surveillance Committee of JSC, JAID and JSCM, Tokyo, Japan;3. Department of Infection Control and Laboratory Medicine, Sapporo Medical University School of Medicine, Sapporo, Japan;4. Department of Urology, University of Occupational and Environmental Health, Kitakyushu, Japan;5. Department of Urology, Gifu University Hospital, Gifu, Japan;6. Department of Urology, School of Medicine, Fujita Health University, Toyoake, Japan;7. Blood Purification Center, Kagoshima University Hospital, Kagoshima, Japan;8. Department of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan;9. Department of Urology, Hyogo College of Medicine, Nishinomiya, Japan;10. Infection Control Research Center, Kitasato University, Tokyo, Japan;11. Department of Urology, Sapporo Medical University School of Medicine, Sapporo, Japan;12. Department of Urology, The Jikei University Katsushika Medical Center, Tokyo, Japan;13. Department of Urology, The Jikei University School of Medicine, Tokyo, Japan;14. Division of Urology, Kobe University Graduate School of Medicine, Kobe, Japan;15. Department of Urology, Hiroshima University Institute of Biomedical and Health Sciences, Hiroshima, Japan;p. University of Occupational and Environmental Health, Kitakyushu, Japan;q. Department of Urology, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan;r. Ito Urology Clinic, Kitakyushu, Japan;s. Hosobe Clinic, Tokyo, Japan;t. Hirayama Urology Clinic, Kumamoto, Japan;u. Narita Clinic, Nagoya, Japan;v. Yamaguchi Dermatology and Urology Clinic, Munakata, Japan;w. iClinic, Sendai, Japan;x. Sumii Clinic, Hiroshima, Japan;y. Kawai Urology Clinic, Kitakyushu, Japan;z. Kanokogi Clinic, Nishinomiya, Japan;11. Kawano Clinic, Fukuoka, Japan;12. Chokyu Tenma Clinic, Himeji, Japan;13. Hirajima Clinic, Okayama, Japan;14. Araki Urological Clinic, Kurashiki, Japan;15. Kaji Clinic, Fukuoka, Japan;16. Kawahara Urology Clinic, Kagoshima, Japan;17. Department of Urology, Takayama Hospital, Chikushino, Japan;18. Gifu Urological Clinic, Gifu, Japan;19. Yoshioka Urology Clinic, Nishinomiya, Japan;110. Department of Urology, Kano Hospital, Fukuoka, Japan;111. Kadena Urological Clinic, Hiroshima, Japan;112. Nishi Urology and Dermatology Clinic, Fukuoka, Japan;113. Nishimura Urology Clinic, Kitakyushu, Japan;114. Yamauchi Urological Clinic, Aichi, Japan;115. Department of Urology, Toyota Memorial Hospital, Toyota, Japan;1p. Department of Urology, Daiyukai Daiichi Hospital, Ichinomiya, Japan;1q. Ihara Clinic, Kobe, Nishinomiya, Japan;1r. Matsumura Urology Clinic, Kato, Japan;1s. Shirane Urology Clinic, Aki-gun, Japan;1t. Department of Urology, National Hospital Organization Hakodate National Hospital, Hakodate, Japan;1u. Akiyama Urology Clinic, Nishinomiya, Japan;1v. Department of Urology, Fukuoka Shin Mizumaki Hospital, Fukuoka, Japan;1w. Uenohara Clinic, Kitakyushu, Japan;1x. Munakata Suikokai General Hospital, Fukuoka, Japan;1y. Yoh Urology and Dermatology Clinic, Inazawa, Japan;1. Department of Pediatrics, İstanbul Bakırköy Maternity and Children''s Diseases Training and Research Hospital, Istanbul, Turkey;2. Division of Pediatric Nephrology, Department of Pediatrics, İstanbul Bakırköy Maternity and Children''s Diseases Training and Research Hospital, Istanbul, Turkey;3. Department of Pediatric Surgery, İstanbul Bakırköy Maternity and Children''s Diseases Training and Research Hospital, Istanbul, Turkey;1. Department of Hand Surgery, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China;2. Department of Plastic Surgery and Hand Surgery, Klinikum Rechts der Isar, Technische Universität München, München, Germany;1. Department of Pediatrics, University of Alberta, Edmonton, AB, Canada;2. Centre for the Studies of Asphyxia and Resuscitation, Neonatal Research Unit, Royal Alexandra Hospital, Edmonton, AB, Canada;3. Department of Pediatric and Adolescent Medicine, Akershus University Hospital, Lørenskog, Norway;4. Medical Library, Akershus University Hospital, Lørenskog, Norway |
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| Abstract: | ObjectiveTo describe 2 cases of primary hypothy-roidism, precocious puberty, large multicystic ovaries, possible diagnostic dilemma, unilateral oophorectomies, and subsequent response to levothyroxine replacement therapy.MethodsWe present the clinical, biochemical, radiologic, and histopathologic findings in 2 patients with rare cases of Van Wyk-Grumbach syndrome and megaovaries, who underwent unilateral oophorectomy.ResultsTwo patients, an 8-year-old girl and a 3-year-old girl (cases 1 and 2, respectively), were referred to our center. Both patients presented with precocious puberty and vaginal bleeding and had undergone unilateral oophorectomy before referral. In the first patient (case 1), the surgical intervention was a consequence of torsion of the left megaovary, necessitating emergency oophorecto-my. Oophorectomy in the second patient (case 2) was a result of initial diagnostic confusion, inasmuch as a sex-cord stromal tumor was suspected. A detailed history, physical examination, and laboratory results pointed toward primary hypothyroidism due to Hashimoto’s thy-roiditis and thyroid dysgenesis, respectively. Serial ultra-sound studies of the abdomen and pelvis revealed large multicystic ovaries, with progressive enlargement (includ-ing regrowth from an apparent ovarian “postsurgical remnant”). Both patients responded dramatically after initiation of levothyroxine replacement therapy, with no further vaginal bleeding and reversal of megaovary to normal size (in case 1).ConclusionIn a highly selected minority of children with untreated primary hypothyroidism, there is development of precocious puberty and progressively enlarging multicystic ovaries. The precise endocrine, neuroanatomic, and neurophysiologic bases for this phenomenon are unclear. Nevertheless, the entire clinicopathologic picture,including giant ovaries, dramatically reverts to normal status with the restoration of a euthyroid state by means of simple levothyroxine replacement therapy. (Endocr Pract. 2007;13:652-655) |
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