Neuromyelitis Optica Spectrum Disorder Manifested by Persistent Hiccups and Severe Esophagitis in an Adolescent Patient

JPGN Reports · 3 authors, 3 centres

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TL;DR

An 18-year-old male with persistent hiccups and severe distal esophagitis was ultimately diagnosed with neuromyelitis optica spectrum disorder after brain MRI revealed a dorsal medullary lesion, and his symptoms rapidly improved with intravenous steroids.

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This case report describes an 18-year-old male admitted with an 8-day history of nausea, emesis, and intermittent hiccups. Esophagogastroduodenoscopy revealed severe distal esophagitis, and the patient was started on high-dose intravenous proton pump inhibitor and sucralfate. While nausea and emesis improved, hiccups worsened to become constant and proved refractory to metoclopramide, cyproheptadine, and baclofen. A neurological consult identified ankle contractures, clonus, and diffuse hyperreflexia, and brain MRI showed a 5.5 mm enhancing dorsal medullary lesion. Autoantibody testing and cerebrospinal fluid analysis confirmed neuromyelitis optica spectrum disorder. High-dose intravenous steroids produced rapid symptom improvement, and the patient was discharged on oral steroids with close neurology and gastroenterology follow-up. The authors suggest the esophagitis was secondary to prolonged vomiting and hiccups rather than their cause.

STUDY TYPE

case_report

SPECIALTY

neurology

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ORIGINAL PAPER

PMC10191573

Neuromyelitis Optica Spectrum Disorder Manifested by Persistent Hiccups and Severe Esophagitis in an Adolescent Patient

JPGN Reports

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ABSTRACT

Patients with persistent hiccups are often referred to gastrointestinal services. Hiccups lasting greater than 48 hours are classified as persistent, while those lasting longer than 2 months are termed intractable. The etiology of hiccups is broad and can include many organ systems. Here, we present the case of an 18-year-old male patient who presented to the emergency department with an 8-day history of nausea, emesis, and intermittent hiccups. The patient was admitted to the gastroenterology service and underwent workup including esophagogastroduodenoscopy and imaging. Esophagogastroduodenoscopy revealed severe distal esophagitis. He was started on high-dose proton pump inhibitor and sucralfate. During the course of hospital admission, the patient’s intermittent hiccups worsened to become constant. Several medical therapies led to little improvement. We initially felt that the esophagitis was the etiology of the patient’s symptoms, however, due to ongoing symptoms unresponsive to typical therapy, we obtained a neurological consult. Brain MRI revealed a dorsal medullary lesion. Subsequent work up confirmed the diagnosis of neuromyelitis optica spectrum disorder. Neuromyelitis optica spectrum disorder is a rare, serious, sometimes fatal autoimmune condition of the central nervous system. Early diagnosis and treatment are important predictors of future relapses. Our patient was started on high-dose intravenous steroids with rapid improvement in symptoms. He was discharged home on steroids, proton pump inhibitor and close neurology and gastroenterology follow up. This case outlines the importance of consideration of neurological causes of hiccups or other centrally mediated conditions with gastrointestinal manifestations, even when a concomitant gastrointestinal condition is present.

CITATION EXPORT

Priya Singh, Reinaldo Garcia, Madhura Y. Phadke. Neuromyelitis Optica Spectrum Disorder Manifested by Persistent Hiccups and Severe Esophagitis in an Adolescent Patient. JPGN Reports. (2021). https://doi.org/10.1097/PG9.0000000000000117

AUTHORS

3 · 3 CENTRES

Priya Singh, Reinaldo Garcia, Madhura Y. Phadke

From the *Northeast Ohio Medical University, Rootstown, OH
†Division of Pediatric Gastroenterology, Hepatology and Nutrition, Akron Children’s Hospital, Akron, OH
‡Pediatric Gastroenterology, Hepatology and Nutrition, Stanford University School of Medicine, Palo Alto, CA.