Case – A tale of two boys: An atypical cause of pubertal precocity

Akilan, Kosalan; Janssen, Karmon; Lorenzo, Armando J.; Zu’bi, Fadi; Scherz, Hal C.; Koyle, Martin A.

doi:10.5489/cuaj.6270

Cited by 1 publication

(3 citation statements)

References 19 publications

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“…Occasionally, it can progress into gonadotropin-dependent precocious puberty (GDPP) following surgical removal of the mass. We report a case of delayed presentation of LCT with advanced signs of puberty which rapidly progressed to GDPP within a month in contrast to reported cases in the literature [ 1 , 2 ].…”

Section: Introductionmentioning

confidence: 58%

“…A Leydig cell tumor (LCT) is a sex cord-stromal tumor and accounts for 1–3% of all testicular tumors in adults and 4–9% in prepubertal children [ 2 ]. Even though it can occur at any age, among children, it is most commonly present in the prepubertal age between 5 and 10 years and is benign [ 3 ].…”

Section: Discussionmentioning

confidence: 99%

“…In most instances, following treatment, the signs of puberty have regressed, but occasionally, following surgery, some have entered gonadotropin-dependent precocious puberty (GDPP). A review conducted in 2015 reported 8 cases [ 1 ], two cases in 2018 [ 4 ], and only one case 2020 [ 2 ] presenting with GDPP following surgery. GDPP was detected as early as 3 months after surgery, and in some cases, it was detected as late as 1 year after surgery in contrast to our case where GDPP was detected just 1 month after surgery.…”

Section: Discussionmentioning

confidence: 99%

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A Matter of Time: Delayed Presentation and Rapid Progression from Gonadotropin-Independent to Gonadotropin-Dependent Precocious Puberty following Successful Treatment for a Leydig Cell Tumor

Naotunna

Siriwardana

Lakmini

et al. 2022

Case Reports in Pediatrics

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Leydig cell tumors, most often benign, are a rare cause of isosexual gonadotropin-independent precocious puberty in boys due to secretion of testosterone. Very rarely do these tumors produce estrogen, causing gynecomastia. Testicular sparing surgery is the mainstay of treatment currently although radical orchidectomy was the choice in the past. Following surgery, clinical signs improve along with a revision of biochemical changes. Occasionally, it has been reported few children are progressed to gonadotropin-dependent precocious puberty (GDPP) after initial clinical and biochemical recovery. Gonadotropin receptor analogs have been successful on them to halt the progression of puberty, and growth hormone administration has been used to optimize the adult height. Here, we report a case of a 10-year-old boy who presented very late due to failure in recognition of features of puberty due to a Leydig cell tumor. Even though he underwent successful radical orchidectomy, just within 1 month following surgery, he entered GDPP in contrast to the published cases where it was earliest detected at 3 months.

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Section: Introductionmentioning

confidence: 58%

Section: Discussionmentioning

confidence: 99%