We present a case of a 46-year-old transgender male who, during his female-to-male transition, presented with a pituitary apoplexy at the emergency department of a general hospital in the Netherlands. During admission, it turned out that he also suffered from acromegaly due to a growth hormone secreting pituitary adenoma for which he was successfully treated at our university hospital. Previously, his complaints typical of acromegaly were dismissed as attributable to the gender transition. Without the apoplexy, the disease probably would have remained masked by the history of transgenderism for a much longer period of time. It is, therefore, essential to keep looking for additional explanations for new pathology and complaints that cannot typically be attributed to the gender transition in these patients.