Poor linear growth and delayed puberty are the most common presenting manifestations of juvenile primary hypothyroidism. Multicystic ovaries and pituitary mass resulting from thyrotroph cell hyperplasia as presenting features are quite rare. We report a patient with juvenile primary hypothyroidism who presented with acute abdominal pain resulting from huge multicystic ovaries, but its cause remained undiagnosed. Later she manifested with headache and vision loss resulting from a large pituitary mass of thyrotroph cell hyperplasia. These advanced manifestations of the disease and their resolution after l-thyroxine replacement therapy are discussed.