Rationale: Inhaled iloprost has been used as a selective pulmonary vasodilator in the treatment of pulmonary arterial hypertension; however, it is unclear whether it is an effective treatment for exercise-induced pulmonary hypertension (PH). Patient concerns: A 66-year-old Japanese woman with systemic sclerosis-associated interstitial lung disease presented with dyspnea, worsening during exertion. Diagnosis: Overlap of exercise-induced PH and group 3 PH. Interventions: Inhaled iloprost was administrated. Outcomes: Exercise echocardiography revealed that inhaled iloprost therapy improved her exercise-induced PH. Conclusion: Exercise echocardiography and inhaled iloprost revealed that the patient’s PH included both group 3 and group 1 PH. This case suggests that a combination of exercise echocardiography and inhaled iloprost may be useful in distinguishing patients with systemic sclerosis-associated interstitial lung disease in whom group 1 and group 3 PH overlap. In addition, this method might also be useful for determining the therapeutic effects of iloprost.

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