Ectopic hyperprolactinaemia in a woman with a mesocolic perivascular epithelioid cell tumor (“PEComa”)

Proust-Lemoine, Emmanuelle; Mitchell, Valérie; Deruelle, Philippe; Lamblin, Antoine; Néraud, B.; Leroy, Xavier; Leteurtre, Emmanuelle; Dewailly, Didier

doi:10.1016/j.ando.2008.01.004

Cited by 10 publications

(4 citation statements)

References 14 publications

(13 reference statements)

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“…Apart from these, another case investigated for hyperprolactinemia, whose pituitary imaging revealed no lesions, was operated because of a large pelvic mass originating from the mesocolon. The pathology result was a perivascular epithelioid cell tumor and PRL was found to be negative by immunohistochemical examination in this case [4]. Just like other cases of ectopic prolactinoma, prolactin levels in our patient also returned to normal after the surgery.…”

Section: Ectopic Prolactinoma 149supporting

confidence: 52%

A case of an ectopic prolactinoma

Şimşir

Kocabaş

Şahin

et al. 2011

Gynecological Endocrinology

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A 34-year-old female presented to our clinic with a 1.5 year history of secondary amenorrhea and galactorrhea. Prolactin (PRL) level was found to be 151.89 ng/ml. Pituitary imaging was reported to be normal. An examination of the patient revealed that PRL level was still high so the dose of cabergoline was further increased and subsequently, bromocriptine was added to the treatment. There was no reduction in PRL levels in controls. A scanning was performed to look for an ectopic focus. Abdominal computerized tomography revealed a heterogenous mass lesion originating from the uterus. Octreotide scintigraphy was performed and we observed an involvement consistent with the mass in the uterus. The patient underwent abdominal total hysterectomy. PRL dropped to 0.4 ng/ml the next day after the operation. The pathology result was a low-grade malignant mesenchymal tumor. Prolactin was found to be immunohistochemically negative. However, galactorrhea disappeared postoperative and PRL levels are still low. Elevated levels of PRL, resistant to bromocriptine and cabergoline, rapidly returned to normal after hysterectomy, which obviously indicates that hyperprolactinemia was associated with the myoma of the uterus.

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Section: Ectopic Prolactinoma 149supporting

confidence: 52%

A case of an ectopic prolactinoma

Şimşir

Kocabaş

Şahin

et al. 2011

Gynecological Endocrinology

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“…The predominant form in healthy subjects and in patients with prolactinomas is monomeric PRL (molecular mass 23 kDa), while dimeric or big PRL (45)(46)(47)(48)(49)(50)(51)(52)(53)(54)(55)(56)(57)(58)(59)(60), and big-big PRL or macroprolactin (150-170 kDa) correspond to less than 20% of the total PRL (13)(14)(15). When the serum of a patient with hyperprolactinemia contains mostly macroprolactin, the condition is termed macroprolactinemia (16,17).…”

Section: Prolactin Serum Isoformsmentioning

confidence: 99%

Challenges and pitfalls in the diagnosis of hyperprolactinemia

Vilar

Fleseriu

Bronstein

2014

Arq Bras Endocrinol Metab

112

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The definition of the etiology of hyperprolactinemia often represents a great challenge and an accurate diagnosis is paramount before treatment. Although prolactin levels > 200-250 ng/mL are highly suggestive of prolactinomas, they can occasionally be found in other conditions. Moreover, as much as 25% of patients with microprolactinomas may present prolactin levels < 100 ng/mL, which are found in most patients with pseudoprolactinomas, drug-induced hyperprolactinemia, or systemic diseases. On the other hand, some conditions may lead to falsely low PRL levels, particularly the so-called hook effect, that is an assay artifact caused by an extremely high level of PRL, and can be confirmed by repeating assay after a 1:100 serum sample dilution. The hook effect must be considered in all patients with large pituitary adenomas and PRL levels within the normal range or only modestly elevated (e.g., < 200 ng/mL). An overlooked hook effect may lead to incorrect diagnosis and unnecessary surgical intervention in patients with prolactinomas. Another important challenge is macroprolactinemia, a common finding that needs to be identified, as it usually requires no treatment. Although most macroprolactinemic patients are asymptomatic, many of them may present galactorrhea or menstrual disorders, as well as neuroradiological abnormalities, due to the concomitance of other diseases. Finally, physicians should be aware that pituitary incidentalomas are found in at least 10% of adult population. Arq Bras Endocrinol Metab. 2014;58(1):9-22 Keywords Hyperprolactinemia; diagnostic pitfalls; hook effect; macroprolactinemia; pituitary incidentalomas RESUMO A definição da etiologia da hiperprolactinemia muitas vezes representa um grande desafio e um diagnóstico preciso é fundamental antes do tratamento. Embora níveis de prolactina > 200-250 ng/mL sejam altamente sugestivos de prolactinomas, ocasionalmente podem ser encontrados em outras condições. Além disso, até 25% dos pacientes com microprolactinomas podem apresentar-se com níveis de prolactina < 100 ng/mL, os quais são evidenciados na maioria dos pacientes com pseudoprolactinomas, hiperprolactinemia induzida por drogas ou doenças sistêmicas. Por outro lado, deve--se atentar às condições que podem levar a valores de prolactina falsamente baixos, particularmente o chamado efeito gancho. Este último é um artefato causado por um nível extremamente elevado de PRL e que pode ser confirmado pela repetição do exame após diluição do soro a 1:100. O efeito gancho deve ser considerado em todo paciente com grandes adenomas hipofisários e níveis de prolactina dentro da faixa normal ou apenas moderadamente elevados (p. ex., < 200 ng/mL). Um efeito gancho não detectado pode levar a diagnóstico incorreto e intervenção cirúrgica desnecessária em pacientes com prolactinomas. Outro desafio importante é a macroprolactinemia, um achado comum que precisa ser identificado visto que geralmente não requer tratamento. Ainda que a maioria dos pacientes seja assintomática devido à concomitância de outr...

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“…In non-pathological states, prolactin secretion has been demonstrated in decidua, mammary tissue, ovaries, the male reproductive tract, epithelial cells, the immune system, brain, hair follicles, and skin [3]. Extra-pituitary pathological prolactin secretion has been described from perivascular epithelioid tumors [4,5], ovarian tumors [6][7][8], acute lymphoblastic and myeloid leukemias [9][10][11], colorectal adenocarcinoma [12,13], non-Hodgkin's lymphoma [14], renal cell carcinoma [15], renal epitheloid angiomyolipoma [16], cervical cancer [17], and lung cancer [18].…”

Section: Discussionmentioning

confidence: 99%

Uterine Fibroid Causing Hyperprolactinemia and Paradoxical Prolactin Rise with Dopamine Agonist: Case Report and Systematic Review

Upreti

Dray

Elston

2020

SN Compr. Clin. Med.

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Hyperprolactinemia in premenopausal women often presents with amenorrhea, symptoms of estrogen deficiency and subfertility. After exclusion of other common causes and in the context of a normal pituitary MRI scan, hyperprolactinemia is typically deemed "idiopathic." Here we present a case where excess prolactin secretion was attributed to a uterine fibroid and following myomectomy the hyperprolactinemia and its clinical sequelae resolved. Following 6 years of secondary infertility, a 41-year-old woman presented with secondary amenorrhea, expressible galactorrhea, and symptoms of estrogen deficiency. Prolactin levels were consistently raised (1300 mU/L to 2100 mU/L, reference interval < 600 mU/L), and repeated pituitary imaging demonstrated no pituitary abnormality. Prolactin levels demonstrated a paradoxical rise in response to dopamine agonist therapy (2000 mU/L to 5900 mU/L). Imaging demonstrated a uterine fibroid and the patient underwent myomectomy. Following myomectomy, prolactin levels became undetectable, galactorrhea and symptoms of estrogen deficiency resolved, menses returned, and a planned unassisted pregnancy followed. Ectopic prolactin secretion from uterine fibroids is a rare cause of hyperprolactinemia and can result in the same clinical sequelae as pituitary causes. As ectopic prolactin secretion is not under inhibitory control of dopamine, it is usually resistant to, or may rise in response to, dopamine agonist therapy. Prior to a diagnosis of "idiopathic" hyperprolactinemia, ectopic sources of prolactin should be considered.

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Ectopic hyperprolactinaemia in a woman with a mesocolic perivascular epithelioid cell tumor (“PEComa”)

Cited by 10 publications

References 14 publications

A case of an ectopic prolactinoma

A case of an ectopic prolactinoma

Challenges and pitfalls in the diagnosis of hyperprolactinemia

Uterine Fibroid Causing Hyperprolactinemia and Paradoxical Prolactin Rise with Dopamine Agonist: Case Report and Systematic Review

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