Shivangini Duggal, Monica Botros, Emerald Zaw, Swati Mahapatra, Jesus Guzman, Keith Garrison, Richard Mccallum
{"title":"三A综合征——贲门失弛缓症的罕见遗传原因。","authors":"Shivangini Duggal, Monica Botros, Emerald Zaw, Swati Mahapatra, Jesus Guzman, Keith Garrison, Richard Mccallum","doi":"10.14309/crj.0000000000001686","DOIUrl":null,"url":null,"abstract":"<p><p>Triple A syndrome (TAS) is a rare disorder inherited in an autosomal recessive manner. It is characterized by the triad of alacrimia, achalasia, and adrenal insufficiency. It is caused by mutations in the <i>AAAS</i> gene (achalasia-addisonianism-alacrima syndrome), which disrupts the protein ALADIN (Alacrima-Achalasia-Adrenal insufficiency Neurologic disorder protein), which plays an important role in nucleocytoplasmic transport and cellular stress response. Unlike the presented cases, most patients with TAS present in early childhood with various symptoms including dry eyes, difficulty swallowing, and recurrent infections in addition to signs of adrenal crisis such as hypotension, hypoglycemia, and hyperpigmentation. Diagnosis can be achieved by genetic testing, revealing the mutations in the <i>AAAS</i> gene. Management of TAS mainly focuses on addressing each condition separately such as prescribing artificial tears for alacrimia, glucocorticoid replacement therapy for adrenal insufficiency, and performing pneumatic dilation or surgical intervention for achalasia. Early diagnosis is crucial for improving quality of life and minimizing the morbidity and mortality linked to the syndrome.</p>","PeriodicalId":7394,"journal":{"name":"ACG Case Reports Journal","volume":"12 5","pages":"e01686"},"PeriodicalIF":0.6000,"publicationDate":"2025-04-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12026375/pdf/","citationCount":"0","resultStr":"{\"title\":\"Triple A Syndrome-A Rare Hereditary Cause of Achalasia.\",\"authors\":\"Shivangini Duggal, Monica Botros, Emerald Zaw, Swati Mahapatra, Jesus Guzman, Keith Garrison, Richard Mccallum\",\"doi\":\"10.14309/crj.0000000000001686\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Triple A syndrome (TAS) is a rare disorder inherited in an autosomal recessive manner. It is characterized by the triad of alacrimia, achalasia, and adrenal insufficiency. It is caused by mutations in the <i>AAAS</i> gene (achalasia-addisonianism-alacrima syndrome), which disrupts the protein ALADIN (Alacrima-Achalasia-Adrenal insufficiency Neurologic disorder protein), which plays an important role in nucleocytoplasmic transport and cellular stress response. Unlike the presented cases, most patients with TAS present in early childhood with various symptoms including dry eyes, difficulty swallowing, and recurrent infections in addition to signs of adrenal crisis such as hypotension, hypoglycemia, and hyperpigmentation. Diagnosis can be achieved by genetic testing, revealing the mutations in the <i>AAAS</i> gene. Management of TAS mainly focuses on addressing each condition separately such as prescribing artificial tears for alacrimia, glucocorticoid replacement therapy for adrenal insufficiency, and performing pneumatic dilation or surgical intervention for achalasia. Early diagnosis is crucial for improving quality of life and minimizing the morbidity and mortality linked to the syndrome.</p>\",\"PeriodicalId\":7394,\"journal\":{\"name\":\"ACG Case Reports Journal\",\"volume\":\"12 5\",\"pages\":\"e01686\"},\"PeriodicalIF\":0.6000,\"publicationDate\":\"2025-04-25\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12026375/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"ACG Case Reports Journal\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.14309/crj.0000000000001686\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/5/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"GASTROENTEROLOGY & HEPATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"ACG Case Reports Journal","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.14309/crj.0000000000001686","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/5/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"GASTROENTEROLOGY & HEPATOLOGY","Score":null,"Total":0}
Triple A Syndrome-A Rare Hereditary Cause of Achalasia.
Triple A syndrome (TAS) is a rare disorder inherited in an autosomal recessive manner. It is characterized by the triad of alacrimia, achalasia, and adrenal insufficiency. It is caused by mutations in the AAAS gene (achalasia-addisonianism-alacrima syndrome), which disrupts the protein ALADIN (Alacrima-Achalasia-Adrenal insufficiency Neurologic disorder protein), which plays an important role in nucleocytoplasmic transport and cellular stress response. Unlike the presented cases, most patients with TAS present in early childhood with various symptoms including dry eyes, difficulty swallowing, and recurrent infections in addition to signs of adrenal crisis such as hypotension, hypoglycemia, and hyperpigmentation. Diagnosis can be achieved by genetic testing, revealing the mutations in the AAAS gene. Management of TAS mainly focuses on addressing each condition separately such as prescribing artificial tears for alacrimia, glucocorticoid replacement therapy for adrenal insufficiency, and performing pneumatic dilation or surgical intervention for achalasia. Early diagnosis is crucial for improving quality of life and minimizing the morbidity and mortality linked to the syndrome.
期刊介绍:
ACG Case Reports Journal is a peer-reviewed, open-access publication that provides GI and hepatology fellows, private practice clinicians, and other healthcare providers an opportunity to share interesting case reports with their peers and with leaders in the field. ACG Case Reports Journal publishes case reports, images, videos and letters to the editor in all topics of gastroenterology and hepatology, including: Biliary Colon Endoscopy Esophagus Functional Bowel Disorders Inflammatory Bowel Disease Liver Nutrition and Obesity Pancreas Pathology Pediatric Small Bowel Stomach.