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Interesting Cases Session

This case study discusses a 28-year-old pregnant woman who presented with a complex cystic mass in the pouch of Douglas. Further investigation revealed spontaneous ovarian hyperstimulation syndrome (OHS) associated with hypothyroidism. The impact on pregnancy and the importance of considering OHS as a diagnosis are highlighted.

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Interesting Cases Session

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  1. Interesting Cases Session Dr Abi Ezhil, Dr Joanna McNeil Queen Elizabeth University Hospital Glasgow

  2. Clinical Information • 28 year old female, G0P1 • No relevant PMHx • Viable single pregnancy – unassisted conception • Only complaint was mild backache • Referred for MRI Fig 1. US at 12 weeks identified “complex multi-loculated cystic mass in Pouch of Douglas”

  3. MRI: 14 weeks

  4. MRI: 14 weeks Fig. 2 Left ovarian/pouch of Douglas mass measuring 9.6 x 9.4 x 6cm, right ovarian mass 8.3 x 7.8 x 5cm. Thickened septae and areas of haemorrhage. Small to moderate volume free fluid. Single viable pregnancy. Reported as suspicious for malignancy. Re-discussed at MDT – not definitive of malignancy, suggested repeat imaging in 6 weeks.

  5. MRI: 20 weeks

  6. MRI: 20 weeks Fig. 3 Enlarging multi-cystic swollen ovaries; 14.4cm and 15.6cm, small volume free fluid, swollen normal ovarian tissue between the cysts. Less free fluid. Viable single pregnancy. Possibility of OHS raised.

  7. Additional History • Fatigued prior to conception, husband reported deeper voice • FHx: Sister diagnosed with hypothyroidism 7 years ago

  8. Final Diagnosis • Spontaneous ovarian hyper-stimulation associated with hypothyroidism (driven by elevated TSH levels) • MDT advised endocrine opinion and repeat imaging in 6 weeks to assess response to treatment

  9. Follow-up • Transferred care to India and returned to UK ?after birth of child • Started on Thyronorm whilst in India, changed to levothyroxine following review in May 2018

  10. Discussion • OHS can occur before or after conception due to ovarian stimulation during fertility treatment • Spontaneous OHS is much rarer is classified into 3 categories: • Type I: Associated with FSH receptor mutation • Type II Driven by high levels of beta HCG (e.g. molar or multiple pregnancies) • Type III: Related to hypothyroidism (FSH-secreting pituitary adenoma, Factor V Leiden deficiency with thrombosis)

  11. Pathophysiology of Type III • Not well understood • Two theories: • Formation of estriol instead of estradiol triggers excessive gonadotrophin release due to decreased negative feedback due to substitution of the less potent hormone • FSH receptor mutation or cross signalling with normal FSH receptors allowing TSH to directly stimulate the ovaries

  12. Impact on Pregnancy • Foetus is entirely reliant on maternal T4 for 12 weeks • In the absence of maternal T4, brain development injury is a possibility • Spectrum of reduced IQ 7-10 points to more severe neurological morbidity • Patient was counselled accordingly and TOP discussed • Possibility of cyst complications in late pregnancy (rupture, haemorrhage, torsion)

  13. Key Learning Points • Consider OHS as a diagnosis is patients with unassisted pregnancy and cystic ovarian lesions • Typically imaging reveals multiple ovarian cysts of varying sizes giving a spoke-wheel appearance to the ovaries • Always consider the implications to the pregnancy of the final diagnosis and counsel accordingly

  14. Questions?

  15. References • Korevaar TIM, Medici M, Visser TJ et al. Thyroid disease in pregnancy: new insights in diagnosis and clinical management. Nature Reviews Endocrinology 2017; 13: 610-622 • Halim B, Lubis HP, Amalia T. Ovarian hyperstimulation syndrome in spontaneous pregnancy with sacrococcygeal teratoma complicated by maternal mirror syndrome: a comorbidity. Middle East Fertility Society Journal 2016; 21 (4): 277-280 (Surgical image credit) • Sridev S, Barathan S. Case report on spontaneous ovarian hyperstimulation syndrome following natural conception associated with primary hypothyroidism. J Hum Reprod Sci 2013;6:158-61. • Hedayati Emami MH, Molaei Langroudi R, Ghazanfari Amlashi F (2012) Ovarian Hyperstimulation Syndrome and Autoimmune Primary Hypothyroidism in Two Members of a Family. J Clinic Case Reports 2:113. • Kanza RE, Gagnon S, Villeneuve H et al. Spontaneous ovarian hyperstimulation syndrome and pituitary hyperplasia mimicking macroadenoma associated with primary hypothyroidism. World J Radiol 2013 January 28; 5(1): 20-24. • Ilanchezhian S, Mohan SV, Ramachandran R. Spontaneous ovarian hyperstimulation syndrome with primary hypothyroidism: imaging a rare entitiy. Radiology Case Reports 2015: 10 (1)

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