Prolactinoma in childhood and adolescence—Tumour size at presentation predicts management strategy: Single centre series and a systematic review and meta-analysis

TY - JOUR

T1 - Prolactinoma in childhood and adolescence—Tumour size at presentation predicts management strategy

T2 - Single centre series and a systematic review and meta-analysis

AU - Arya, Ved Bhushan

AU - Aylwin, Simon J.B.

AU - Hulse, Tony

AU - Ajzensztejn, Michal

AU - Kalitsi, Jennifer

AU - Kalogirou, Nicolas

AU - Bodi, Istvan

AU - Thomas, Nick

AU - Hampton, Tim

AU - Kapoor, Ritika R.

AU - Buchanan, Charles R.

PY - 2021/3

Y1 - 2021/3

N2 - ObjectiveTo report the clinical presentation, management and outcomes of young patients with prolactinomas (<20 years) and conduct a systematic review and meta-analysis.Patients and DesignClinical, biochemical and radiological data (1996-2018) were collected from our centre. A systematic review and meta-analysis of published literature (1994-2019) on prolactinoma (age <20 years) were conducted. Both random and fixed effects meta-analysis were used to pool outcomes across studies.Results 1 Case seriesTwenty-two patients (14 females) were identified; median age at diagnosis 15.7 years (range 13-19); 12 patients (6 females) had a macroprolactinoma. Seven patients (macroprolactinoma-6) had associated pituitary hormone deficiencies at presentation. Five patients (4 males) underwent surgical resection due to poor response to cabergoline or apoplexy. Patients undergoing surgery had larger tumours (p < .02) and higher serum prolactin concentration (p < .005). All patients with macroprolactinoma >20 mm required surgical intervention.Results 2 Systematic review and meta-analysisWe selected 11 studies according to strict inclusion criteria describing 275 patients. Macroprolactinoma was more common in girls (78.7% [95% CI 70.5-85.9]) than boys and was more frequent than microprolactinoma (56.6% [95% CI 48.4-64.5]). In males, only 6/57 (10.5%) of tumours were microprolactinoma as compared to 102/198 (51.5%) microprolactinoma in females (risk difference −0.460; [95% CI −0.563 to −0.357]; p < .001). Surgery was first-line therapy in 18.9% patients, with another 15.4% requiring it as a second line (overall 31.3%).ConclusionsMacroprolactinoma, particularly if >20 mm, usually requires multimodal therapy including surgical intervention. While overall prolactinomas in <20 years age group are more common in females, the proportion of macroprolactinoma vs microprolactinoma is greater in males, particularly for large invasive tumours. Microprolactinoma is a rare diagnosis in adolescent males.

AB - ObjectiveTo report the clinical presentation, management and outcomes of young patients with prolactinomas (<20 years) and conduct a systematic review and meta-analysis.Patients and DesignClinical, biochemical and radiological data (1996-2018) were collected from our centre. A systematic review and meta-analysis of published literature (1994-2019) on prolactinoma (age <20 years) were conducted. Both random and fixed effects meta-analysis were used to pool outcomes across studies.Results 1 Case seriesTwenty-two patients (14 females) were identified; median age at diagnosis 15.7 years (range 13-19); 12 patients (6 females) had a macroprolactinoma. Seven patients (macroprolactinoma-6) had associated pituitary hormone deficiencies at presentation. Five patients (4 males) underwent surgical resection due to poor response to cabergoline or apoplexy. Patients undergoing surgery had larger tumours (p < .02) and higher serum prolactin concentration (p < .005). All patients with macroprolactinoma >20 mm required surgical intervention.Results 2 Systematic review and meta-analysisWe selected 11 studies according to strict inclusion criteria describing 275 patients. Macroprolactinoma was more common in girls (78.7% [95% CI 70.5-85.9]) than boys and was more frequent than microprolactinoma (56.6% [95% CI 48.4-64.5]). In males, only 6/57 (10.5%) of tumours were microprolactinoma as compared to 102/198 (51.5%) microprolactinoma in females (risk difference −0.460; [95% CI −0.563 to −0.357]; p < .001). Surgery was first-line therapy in 18.9% patients, with another 15.4% requiring it as a second line (overall 31.3%).ConclusionsMacroprolactinoma, particularly if >20 mm, usually requires multimodal therapy including surgical intervention. While overall prolactinomas in <20 years age group are more common in females, the proportion of macroprolactinoma vs microprolactinoma is greater in males, particularly for large invasive tumours. Microprolactinoma is a rare diagnosis in adolescent males.

KW - cabergoline

KW - MEN1

KW - meta-analysis

KW - pituitary adenoma

KW - prolactinoma

KW - systematic review

UR - http://www.scopus.com/inward/record.url?scp=85098090742&partnerID=8YFLogxK

U2 - 10.1111/cen.14394

DO - 10.1111/cen.14394

M3 - Article

AN - SCOPUS:85098090742

SN - 0300-0664

VL - 94

SP - 413

EP - 423

JO - Clinical endocrinology

JF - Clinical endocrinology

IS - 3

ER -