Nakamura Jun, Hikichi Takuto, Inoue Haruhiro, Watanabe Ko, Kikuchi Hitomi, Takagi Tadayuki, Suzuki Rei, Sugimoto Mitsuru, Konno Naoki, Waragai Yuichi, Asama Hiroyuki, Takasumi Mika, Sato Yuki, Irie Hiroki, Obara Katsutoshi, Ohira Hiromasa
Department of Endoscopy, Fukushima Medical University Hospital, 1 Hikarigaoka, Fukushima, 960-1295, Japan.
Department of Gastroenterology, Fukushima Medical University School of Medicine, Fukushima, 960-1295, Japan.
Clin J Gastroenterol. 2018 Aug;11(4):273-277. doi: 10.1007/s12328-018-0819-7. Epub 2018 Jan 30.
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.
奥尔格罗夫综合征,也称为三A综合征,是一种罕见的常染色体隐性遗传病,其特征为三个体征:食管失弛缓症、促肾上腺皮质激素抵抗和无泪症。一名31岁男性因食管失弛缓症导致吞咽困难前来我院就诊。由于他伴有无泪症、神经系统疾病以及近亲结婚家族史,怀疑患有遗传性神经系统疾病。随后,在失弛缓症-阿狄森氏病-无泪症综合征基因中发现了一个突变。随着奥尔格罗夫综合征的确诊,对食管失弛缓症进行了经口内镜下肌切开术(POEM)。POEM术后,症状和食管压力检查结果迅速改善,在超过1年的随访中未发现复发。在此,我们报告首例针对奥尔格罗夫综合征食管失弛缓症进行POEM手术的病例。