Aim: To report a rare case of Spontaneous subcutaneous emphysema and pneumo-mediastinum Introduction: Spontaneous subcutaneous emphysema and pneumo-mediastinum may be defined as the presence of free gas or air in the subcutaneous tissue or mediastinal structures without an apparent precipitating cause. It most commonly occurs in adolescent in good physical health group devoid of severe existing lung pathology. Case Report: In our case, a middle aged married housewife was referred to Department of Medicine, Peoples Medical University Hospital Nawabshah, Sindh, Pakistan, by her local GP for the worsening of her condition due to development of acute neck pain, difficulty in breathing, eating and swallowing (mainly for solid foods) with swelling of neck. She was not complaining of any respiratory symptoms. The chest and neck radiographs showed subcutaneous emphysema and pneumo-mediastinum, there was no any evidence of air leakage from esophagus. She was subsequently put on free fluids, light diet, antibiotics, analgesia and other supportive measures along with close observation. After three days of admission, her clinical symptoms were alleviated to a great extent. She was discharged well from hospital after four days. Conclusion: Spontaneous subcutaneous emphysema with pneumo-mediastinum is medical and surgical emergency. Diagnosis may be made by routine chest X-rays and CT scan of the chest. Prompt diagnosis and immediate management may affect the morbidity and mortality outcomes in this condition.