Efficacy of aromatase inhibitor therapy in a case with large cell calcifying Sertoli cell tumour-associated prepubertal gynaecomastia
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Gözde Akın Kağızmanlı
,
Özge Besci
,
Kübra Yüksek Acinikli
,
Gül Şeker
,
Elif Yaşar
,
Yeşim Öztürk
,
Korcan Demir
,
Ece Böber
and
Ayhan Abacı
Abstract
Objectives
Large cell calcifying Sertoli cell tumours (LCCSCTs) are one of the infrequent causes of prepubertal gynaecomastia. Most of these tumours are in the content of Peutz–Jeghers syndrome (PJS) or other familial syndromes (Carney complex).
Case presentation
Here, we report a long-term follow-up of an 8.5-year-old prepubertal boy with a diagnosis of PJS, who presented with bilateral gynaecomastia, advanced bone age and accelerated growth velocity, and were found to have bilateral multifocal testicular microcalcifications. As the findings were compatible with LCCSCT, anastrozole was initiated. Gynaecomastia completely regressed and growth velocity and pubertal development were appropriate for age during follow-up. Testicular lesions slightly increased in size. After four years of medication, anastrozole was discontinued but was restarted due to the recurrence of gynaecomastia after six months.
Conclusions
Testicular tumour should be investigated in a patient with PJS who presents with prepubertal gynaecomastia. When findings are consistent with LCCSCT, aromatase inhibitors may be preferred in the treatment.
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Research funding: None declared.
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Author contributions: All authors contributed to the study conception and design. G.A.K. gathered data and wrote the first draft; O.B. and K.Y.A. gathered data and followed up with the patient; G.S. and Y.O. performed a colonoscopy and treated gastroenterologic involvement; E.Y. performed testicular ultrasound; A.A. edited the paper; and K.D. and E.B. investigated the clinical disorder, made the clinical diagnosis, followed up with the patient, and critically edited the paper. All authors commented on the drafts of the manuscript. All authors read and approved the final manuscript. All authors have accepted responsibility for the entire content of this manuscript and approved its submission.
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Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.
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Ethical approval: Research has been complied with all the relevant national regulations, and institutional policies and in accordance with the World Medical Association Declaration of Helsinki and the appropriate guidelines for human studies.
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Informed consent: Written informed consent was obtained from the parents.
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Consent for publication: Parents signed informed consent regarding publishing their data. All authors read and approved the final manuscript for publication.
References
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Articles in the same Issue
- Frontmatter
- Review Article
- The efficacy and safety of dipeptidyl peptidase-4 inhibitors and glucagon-like peptide-1 agonists in pediatric patients with type 2 diabetes: a systematic review
- Original Articles
- Triple burden of malnutrition and role of anaemia in the development of complications associated with type 1 diabetes in Indian children and youth
- Effect of obesity and excessive body fat on glycaemic control in paediatric type 1 diabetes
- Role of pan immune inflammatory value in the evaluation of hepatosteatosis in children and adolescents with obesity
- Secular trends of birth weight and its associations with obesity and hypertension among Southern Chinese children and adolescents
- Insights into the implication of obesity in hypogonadism among adolescent boys
- The association between plasma carnitines and duration of diabetic ketoacidosis treatment in children with type 1 diabetes
- Cathelicidin as a marker for subclinical cardiac changes and microvascular complications in children and adolescents with type 1 diabetes
- Developing a risk assessment tool for identifying individuals at high risk for developing insulin resistance in European adolescents: the HELENA-IR score
- Hemoglobin A1C can differentiate subjects with GCK mutations among patients suspected to have MODY
- Perceptions and use of complementary and alternative medicine in patients with precocious puberty
- Case Reports
- Infection with SARS-CoV-2 may alter the half-life of desmopressin (DDAVP) in patients with central diabetes insipidus
- Heterozygous CDC73 mutation causing hyperparathyroidism in children and adolescents: a report of 2 cases
- Wolfram syndrome in a young woman with associated hypergonadotropic hypogonadism – A case report
- Continuous glucose monitoring in an infant with panhypopituitarism having hypoglycemia on growth hormone therapy
- Severe consumptive hypothyroidism in hepatic hemangioendothelioma
- Efficacy of aromatase inhibitor therapy in a case with large cell calcifying Sertoli cell tumour-associated prepubertal gynaecomastia
