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Hypothyroidism is usually associated with delayed puberty and occasionally may present with isosexual precocious puberty. In girls, this may present with breast development, multicystic ovaries and vaginal bleeding. This entity characterized by ovarian hyper stimulation leading to early puberty secondary to hypothyroidism is known as Van Wyke Grumbach syndrome. In contrast to the early puberty caused by other causes, precocious puberty of hypothyroidism is characterized by short stature and delayed bone age. Awareness about this condition and the treatment of this condition with levothyroxine will lead to avoidance of surgery and unnecessary intervention. We present the case of a six-year-old girl who presented with precocious puberty and pituitary hyperplasia. This case highlights the need for the professionals to familiarize themselves about uncommon complications of untreated hypothyroidism.