Foetal exposure to mitotane/Op'DDD: Post‐natal study of four children

Magkou, Dimitra; Cao, Christine Do; Bouvattier, Claire; Douillard, Claire; Marcellus, C. De; Cazabat, Laure; Gérard, Maxime; Raffin-Sanson, Marie-Laure; Young, Jacques

doi:10.1111/cen.13854

Cited by 7 publications

(6 citation statements)

References 31 publications

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“…Their normal educational and vocational development thus extends the findings of Magkou et al. ( 51 ), showing normal neurocognitive development throughout early childhood after exposure to mitotane in pregnancy.…”

Section: Discussionsupporting

confidence: 86%

“…In contrast, our patient's sons, as well as the other live births, reported in Table 2, showed no neurologic impairments. Their normal educational and vocational development thus extends the findings of Magkou et al (51), showing normal neurocognitive development throughout early childhood after exposure to mitotane in pregnancy.…”

Section: Fertility During Mitotane Treatmentsupporting

confidence: 85%

“…In all the other live births, mitotane was either stopped before conception or in the first trimester. There was no regular evaluation of o,p’-DDD-plasma levels throughout pregnancy in most of the presented cases and there are inconsistent findings regarding the extent to which mitotane is transferred from the mother to the fetus ( 26 , 49 , 51 ).…”

Section: Discussionmentioning

confidence: 88%

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Safe long-term therapy of Cushing’s syndrome over 37 years with mitotane

Seibold,

Hönemann,

Tönjes

et al. 2024

Front. Endocrinol.

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While suggested, surgery is not always possible as a first-line treatment of Cushing’s Disease (CD). In such cases, patients require medical therapy in order to prevent complications resulting from hypercortisolism. Although there has been a wide expansion in pharmacological options in recent years, mitotane was the agent of choice for treating hypercortisolism decades ago. Due to the introduction of other therapies, long-term experience with mitotane remains limited. Here, we report the case of a woman with CD who was treated with mitotane for 37 years. During the treatment period, biochemical and clinical disease control was achieved and the patient had two uncomplicated pregnancies. Drug-related side effects remained moderate and could be controlled by several dose adjustments. Our case highlights the ability of mitotane to allow an effective control of hypercortisolism and to represent a safe treatment option in special situations where CD requires an alternative therapeutic approach. Furthermore, we provide a literature review of the long-term use of mitotane and reported cases of pregnancy in the context of mitotane therapy.

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Section: Discussionsupporting

confidence: 86%

Section: Fertility During Mitotane Treatmentsupporting

confidence: 85%

Section: Discussionmentioning

confidence: 88%

See 1 more Smart Citation

Safe long-term therapy of Cushing’s syndrome over 37 years with mitotane

Seibold,

Hönemann,

Tönjes

et al. 2024

Front. Endocrinol.

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“…Hormones were measured as previously described in the accredited (COFRAC; www.cofrac.fr) clinical endocrinology laboratory in each centre (Lahlou et al, 2004;Magkou et al, 2018). In brief, total testosterone and estradiol, were measured by RIA, LH and FSH, were measured by a time-resolved fluorometric assay, SHBG was measured using the Immulite 2000 immunoassay, and inhibin B and AMH were measured using a solid-phase sandwich assay.…”

Section: Hormonal Assaysmentioning

confidence: 99%

Structural analysis of the impact of a novel androgen receptor gene mutation in two adult patients with mild androgen insensitivity syndrome

et al. 2020

Self Cite

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Androgen receptor gene (AR) mutations are responsible for androgen insensitivity syndrome (AIS) presenting with a clinical phenotype that ranges from gynaecomastia and/ or infertility in mild AIS (MAIS) to complete testicular feminisation in complete AIS. We report a novel AR gene mutation in two unrelated adult patients with MAIS and we studied its functional impact using 3D modelling. Patient 1, referred for infertility, presented with gynaecomastia, mild hypospadias and bilateral testicular hypotrophy contrasting with high testosterone levels, an elevated FSH, an elevated androgen sensitivity index (ASI) and oligoasthenoteratospermia. In vitro fertilisation and intracytoplasmic sperm injection resulted in a successful twin pregnancy. Patient 2 referred for a decrease in athletic performance had surgically treated gynaecomastia, oligoasthenospermia, high testosterone levels and an elevated ASI. Despite his impaired spermogram, he fathered two children without assisted reproductive technology. AR gene sequencing in the two patients revealed a common novel missense mutation, Ala699Thr, in exon 4 within the ligand-binding domain. 3D modelling studies showed that this mutation may impact dimer stability upon ligand binding or may affect allosteric changes upon dimerisation. This study illustrates the value of structural analysis for the functional study of mutations and expands the database of AR gene mutations.

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“…Currently, there are only five published case reports of women with ACC on mitotane who have become pregnant Table 1. [3][4][5][6][7] As a result, there is scant data regarding pregnancy outcomes, neonatal morbidity and safety of mitotane as well as progression of disease; thus, counseling patients poses a significant challenge, especially in the post-Dobbs era. We attempt to provide insight on the medical and ethical dilemmas for patients similar to ours.…”

Section: Introductionmentioning

confidence: 99%

Fertility and reproductive outcomes in woman with adrenocortical carcinoma on mitotane: a case report

Salina

2023

MOJWH

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Adrenocortical carcinoma (ACC) is a rare disease with an incidence of 1 in 1.7 million. Due to its rare occurrence, there is sparse data on fertility or reproductive outcomes for patients with ACC. Our case demonstrates a nulligravida woman with recurrent metastatic ACC on daily mitotane that was diagnosed with an incidental pregnancy. Mitotane crosses the placenta, but the teratogenic effects are unknown. Currently, there are only five case reports published of women with ACC conceived while on mitotane. We present a case of pregnancy in a woman with metastatic ACC who was treated with mitotane.

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Foetal exposure to mitotane/Op'DDD: Post‐natal study of four children

Cited by 7 publications

References 31 publications

Safe long-term therapy of Cushing’s syndrome over 37 years with mitotane

Safe long-term therapy of Cushing’s syndrome over 37 years with mitotane

Structural analysis of the impact of a novel androgen receptor gene mutation in two adult patients with mild androgen insensitivity syndrome

Fertility and reproductive outcomes in woman with adrenocortical carcinoma on mitotane: a case report

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