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青春期前阴毛早现作为垂体大泌乳素瘤的首发表现

Premature pubarche as a first presentation of pituitary macroprolactinoma.

作者信息

Tabatabaei Fatemeh, Sharif Mohammad Reza, Nasr Esfahani Hossein, Yazdi Zahrani Razieh, Taheri Abolfazl, Meamar Rokhsareh

机构信息

Clinical Research Development Research Centre, Najafabad Branch, Islamic Azad University, Najafabad, Iran.

Infectious Diseases Research Center, Kashan University of Medical Sciences, Kashan, Iran.

出版信息

J Res Med Sci. 2020 Nov 26;25:108. doi: 10.4103/jrms.JRMS_118_20. eCollection 2020.

DOI:10.4103/jrms.JRMS_118_20
PMID:33824673
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8019103/
Abstract

Prolactinoma is a rare tumor of childhood. Clinical presentations of prolactinoma include amenorrhea, delayed puberty, and galactorrhea. For the first time, in this case, elevated prolactin levels were associated with unexpected premature pubarche. We describe an 8-year, 7-month-old boy with acne and gradual appearance of pubic hair, corresponding to tanner stage 2. Hormonal tests showed severe hyperprolactinemia (prolactin = 246.8 μg/L and pooled prolactin = 175 μg/L and macroprolactin = 5 μg/L) and mildly elevated level of dehdroepiandrostenedion sulfate (DHEAS) and testosterone. Magnetic resonance imaging (MRI) findings confirmed the presence of a pituitary macroprolactinoma, measuring 14 mm × 12 mm × 8 mm on the right side of the pituitary gland. Cabergoline therapy was commenced (0.5 mg/week) and after 3 months, no evidence of pubarche progression was observed. Prolactin level and tumor size markedly reduced. At the 9-month follow-up visit, a normal MRI was reported. This case highlights that even when facing premature pubarche, careful examination is mandatory, and if no obvious etiology is found for premature pubarche, clinicians should consider prolactinoma.

摘要

催乳素瘤是一种罕见的儿童肿瘤。催乳素瘤的临床表现包括闭经、青春期延迟和溢乳。在本病例中,催乳素水平升高首次与意外的青春期过早出现相关。我们描述了一名8岁7个月大的男孩,有痤疮且阴毛逐渐出现,对应坦纳2期。激素检测显示严重高催乳素血症(催乳素 = 246.8 μg/L,总催乳素 = 175 μg/L,大催乳素 = 5 μg/L),硫酸脱氢表雄酮(DHEAS)和睾酮水平轻度升高。磁共振成像(MRI)结果证实存在垂体大催乳素瘤,位于垂体右侧,大小为14 mm×12 mm×8 mm。开始使用卡麦角林治疗(0.5 mg/周),3个月后,未观察到阴毛生长进展的迹象。催乳素水平和肿瘤大小明显降低。在9个月的随访中,报告MRI正常。该病例强调,即使面对青春期过早出现,仔细检查也是必要的,如果未发现青春期过早出现的明显病因,临床医生应考虑催乳素瘤。

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Premature pubarche as a first presentation of pituitary macroprolactinoma.青春期前阴毛早现作为垂体大泌乳素瘤的首发表现
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[Premature pubarche is not always an innocent phenomenon].青春期阴毛过早发育并不总是一种无害的现象。
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The early dehydroepiandrosterone sulfate rise of adrenarche and the delay of pubarche indicate primary ovarian failure in Turner syndrome.肾上腺初现期硫酸脱氢表雄酮的早期升高以及阴毛初现延迟提示特纳综合征患者存在原发性卵巢功能不全。
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Girls diagnosed with premature pubarche show an exaggerated ovarian androgen synthesis from the early stages of puberty: evidence from gonadotropin-releasing hormone agonist testing.诊断为青春期早熟的女孩从青春期早期就表现出卵巢雄激素合成增加:来自促性腺激素释放激素激动剂试验的证据。
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Front Endocrinol (Lausanne). 2023 Oct 30;14:1248231. doi: 10.3389/fendo.2023.1248231. eCollection 2023.
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Gender differentials in the timing and prognostic factors of pubarche in Nigeria.尼日利亚性早熟出现时间和预后因素的性别差异。
PLoS One. 2022 Nov 21;17(11):e0277844. doi: 10.1371/journal.pone.0277844. eCollection 2022.
3
Delayed pubarche.延迟性性早熟。

本文引用的文献

1
Pediatric Pituitary Adenoma: Case Series, Review of the Literature, and a Skull Base Treatment Paradigm.小儿垂体腺瘤:病例系列、文献综述及颅底治疗模式
J Neurol Surg B Skull Base. 2018 Feb;79(1):91-114. doi: 10.1055/s-0038-1625984. Epub 2018 Jan 24.
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Pediatric prolactinoma: initial presentation, treatment, and long-term prognosis.儿童泌乳素瘤:初始表现、治疗和长期预后。
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Premature adrenarche--a common condition with variable presentation.
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早熟肾上腺初现——一种表现多样的常见病症。
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Paediatric pituitary adenomas: a decade of change.小儿垂体腺瘤:十年变迁
Horm Res Paediatr. 2014;81(3):145-55. doi: 10.1159/000357673. Epub 2014 Feb 11.
5
Macroprolactinoma: a diagnostic and therapeutic update.巨泌乳素瘤:诊断与治疗进展。
QJM. 2013 Jun;106(6):495-504. doi: 10.1093/qjmed/hcs240. Epub 2013 Jan 16.
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The steroid metabolome of adrenarche.肾上腺功能初现期的类固醇代谢组学。
J Endocrinol. 2012 Aug;214(2):133-43. doi: 10.1530/JOE-12-0183. Epub 2012 Jun 19.
7
Childhood craniopharyngioma.儿童颅咽管瘤。
Pituitary. 2013 Mar;16(1):56-67. doi: 10.1007/s11102-012-0401-0.
8
Prolactinoma in children and adolescents.儿童和青少年泌乳素瘤
Horm Res. 2009;72(4):197-205. doi: 10.1159/000236081. Epub 2009 Sep 29.
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Clinical profile and long term follow up of children and adolescents with prolactinomas.儿童和青少年催乳素瘤的临床特征及长期随访
Pituitary. 2009;12(3):186-9. doi: 10.1007/s11102-008-0149-8.
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Macroprolactinemia in childhood and adolescence: a cause of hyperprolactinemia.儿童和青少年期的巨泌乳素血症:高泌乳素血症的一个病因
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