Intractable hiccups due to herpetic esophagitis in an immunocompromised patient

INTRODUCTION: Herpes virus family's association with visceral lesions is well established. Herpes simplex virus presentations vary based on immune status. Intractable hiccups due to herpes simplex esophagitis, to the best of our knowledge have been described twice in the literature. We present a 68 year-old immunocompromised male with intractable hiccups for 10 months. CASE: 68 year-old male with end-stage renal disease and multiple myeloma presented with coffee ground emesis and hiccups of ten months duration. A year earlier, he received cycles of bortezomib and dexamethasone, remaining on lenalidomide. During chemotherapy, he developed pneumococcal meningitis and subsequently intractable hiccups. Preceding admission, endoscopy showed duodenitis and esophagitis. Proton-pump inhibitor therapy was initiated; however, biopsy was not performed. During admission, hiccups often occurred every few seconds while off anti-emetics, persisting despite therapy. Exam showed cachexia/temporal wasting, aphthous stomatitis and abdominal tenderness. MRI of brain/spine, CT of neck, chest, abdomen and neurological evaluation were unremarkable. Endoscopy revealed gastritis and esophagitis with mucosal tears. Biopsy revealed intra-nuclear inclusions with multi-nucleated cells, consistent with herpes virus, later confirmed as herpes simplex by immunostaining. Hiccups and emesis resolved after of 2 days of intravenous acyclovir. 21 days of treatment were completed with oral valacyclovir. He remained free of hiccups during the remaining hospital stay and follow up. This case illustrates an exceptionally rare presentation of herpetic esophagitis in an immunocompromised host. As novel immunotherapeutic/suppressive agents continue to emerge, the evolving role of herpes virus prophylaxis and diagnosis of atypical presentations in new host populations is a topic of growing importance.