Clinical characteristics of preterm ovarian hyperstimulation syndrome: seven cases from China and 14 cases from the literature

Abstract

Preterm ovarian hyperstimulation syndrome (POHS) is a rare condition with only a few reported cases. The objective of this study is to investigate the clinical characteristics of 21 POHS cases (seven from China and 14 from the literature). Suspected patients (n = 7) were referred for further confirmation and data on demographics, family history, clinical manifestations (edema developed in the vulva, the hypogastric site and the upper leg), serum and sonographic assessments (ovarian cyst/cysts, gonadotropins, and estradiol levels), the natural course of patients were documented. We did not observe substantial differences in clinical manifestations among POHS patients from China relative to that from the literature. Preterm female newborns developed edema in the vulva, the hypogastric site and the upper leg at 30–39⁺⁶ weeks of post-conception age (PCA) as the major clinical manifestations. The characteristic also included high gonadotropins and estradiol, and ovarian cysts. This condition can self-resolve. Clinical manifestations of POHS patients from China are similar to those from the literature. The pathognomonic signs of POHS include vulvar, hypogastric, and upper leg edema developed at 30–39⁺⁶ PCA along with high gonadotropins and E2 values and the presence of ovarian cysts in preterm female newborns and the syndrome can self-resolve.

Chinese abstract

早产儿卵巢过度刺激综合征（POHS）是一种罕见的情况, 只有少数病例报道。本研究旨在探讨21例（国内7例, 文献14例）POHS的临床特点。对疑似患者（n = 7）进行进一步确认, 并记录患者的人口统计学、家族史、临床表现（外阴水肿、下腹部及大腿水肿）、血清及超声检查（卵巢囊肿/囊肿、促性腺激素、雌二醇水平）、自然病程等资料。我们观察到中国POHS患者的临床表现与文献中无显著差异。女性早产新生儿在怀孕后期（PCA）、孕30-39⁺⁶周发生外阴水肿、下腹部和大腿水肿为最主要的临床表现。其特征还包括高促性腺激素和雌二醇及卵巢囊肿。这些情况可以自行缓解。中国POHS患者的临床表现与文献相似。POHS的特有表现包括怀孕后期、孕30-39⁺⁶周早产女性新生儿出现外阴、下腹部、大腿水肿, 并伴有高促性腺激素、高E2水平及出现卵巢囊肿, 可自行缓解。

Keywords:

• Preterm infant
• ovarian hyperstimulation syndrome
• edema
• ovarian cyst
• estradiol

Acknowledgements

Statements of ethics: The research was conducted ethically in accordance with the World Medical Association Declaration of Helsinki, and got an ethical approval from the Ethical Institutional Review Board of the First Affiliated Hospital of Wenzhou Medical University and the Yiwu Maternity and Children Hospital. We obtained written informed consent from all patients’ guardians in this study.

Disclosure statement

No potential conflict of interest was reported by the authors.

Additional information

Funding

This work is supported by the Wenzhou Science and Technology Bureau, China [Y 20140497].