Per-oral endoscopic myotomy for esophageal achalasia in a case of Allgrove syndrome

Clin J Gastroenterol. 2018 Aug;11(4):273-277. doi: 10.1007/s12328-018-0819-7. Epub 2018 Jan 30.

Abstract

Allgrove syndrome, also known as Triple A syndrome, is a rare autosomal recessive genetic disease characterized by three signs: esophageal achalasia, adrenocorticotropic hormone refractoriness, and alacrima. A 31-year-old male presented to our hospital for treatment of difficulty swallowing caused by esophageal achalasia. Because he had complicating alacrima, a neurologic disease, and a family history of consanguineous marriage, a genetic neurologic disease was suspected. Then, a mutation in the achalasia-addisonianism-alacrima syndrome gene was identified. With the diagnosis of Allgrove syndrome, a per-oral endoscopic myotomy (POEM) was performed for esophageal achalasia. After the POEM, the symptoms and the esophageal pressure findings ameliorated quickly, with no recurrence noted throughout a follow-up of more than 1 year. Here, we report the first case of POEM performed for esophageal achalasia in Allgrove syndrome.

Keywords: Alacrima; Allgrove syndrome; Dysphagia; Esophageal achalasia; Per-oral endoscopic myotomy.

Publication types

  • Case Reports

MeSH terms

  • Adrenal Insufficiency / complications*
  • Adrenal Insufficiency / diagnosis
  • Adult
  • Deglutition Disorders / etiology
  • Endoscopy / methods*
  • Esophageal Achalasia / complications*
  • Esophageal Achalasia / diagnosis
  • Esophageal Achalasia / pathology
  • Esophageal Achalasia / surgery*
  • Humans
  • Male
  • Myotomy / methods*

Supplementary concepts

  • Achalasia Addisonianism Alacrimia syndrome