Spontaneous ovarian hyperstimulation syndrome (SOHSS) is extremely rare in naturally conceived pregnancies and is potentially a life-threatening condition. Objective of present study was to highlight the development of new and atypical findings associated with SOHSS in a woman who conceived naturally with singleton pregnancy while taking inositols and metformin. A 35 years old lady was under pressure to conceive, so she presented to us for fertility consultation and further management. Being a case of polycystic ovary syndrome (PCOS) with irregular periods, hyperandrogenism and dyslipidemia with raised blood sugar level, she was started on inositols and metformin to aid fertility and to improve endocrine and metabolic parameters. To evaluate the status of ovaries and endometrium, ultrasound (US) was done which revealed moderate amount of free fluid in pelvis, thickened endometrium and mild enlargement of ovaries with unusually small and atypical follicles. Pregnancy test and beta-human chorionic gonadotropin (βHCG) confirmed pregnancy. She was asymptomatic and was managed conservatively. Pregnancy progressed and at term, a healthy male baby was delivered. Awareness and suspicion of SOHSS in a PCOS case aided early diagnosis and timely management. Intake of type of pharmacological and non-pharmacological agents to be monitored for their impact on fertility, ovarian enlargement, severity of ovarian hyperstimulation syndrome (OHSS), clinical presentation, laboratory profile and outcome of pregnancy.