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Cryptogenic Multifocal Ulcerous Stenosing Enteritis (CMUSE): A Tale of Three Decades
Ayaskanta Singh1, Manoj Kumar Sahu1, Manas Kumar Panigrahi2
1Department of Gastroenterology and Hepatobiliary Sciences, Siksha O. Anusandhan University Institute of Medical Sciences and SUM Hospital, Bhubaneswar, India.
ACG Case Reports Journal
|March 24, 2017
Summary
This case study details a patient with cryptogenic multifocal ulcerous stenosis enteritis (CMUSE) who experienced significant improvement with hormonal therapy. This suggests a potential new treatment avenue for this rare intestinal condition.
Area of Science:
- Gastroenterology
- Pathology
- Endocrinology
Background:
- Cryptogenic multifocal ulcerous stenosis enteritis (CMUSE) is a rare gastrointestinal disorder.
- Diagnosis can be challenging, often requiring extensive investigation and leading to delayed treatment.
- Previous treatments for CMUSE have had limited success.
Observation:
- A patient presented with a 35-year history of recurrent ileus and melena, experiencing multiple misdiagnoses and surgeries.
- Resected intestinal tissue showed features consistent with CMUSE, alongside notable angioectatic lesions.
- The patient exhibited a dramatic positive response to hormonal therapy.
Findings:
- This is the first reported case of CMUSE demonstrating a significant response to hormonal therapy.
- The observed angioectatic lesions suggest a potential vasculopathic component in this CMUSE variant.
- Hormonal therapy with ethinylestradiol and norethisterone proved effective.
Implications:
- Hormonal therapy may represent a novel treatment strategy for specific variants of CMUSE.
- Understanding the role of vasculopathy in CMUSE pathogenesis could lead to targeted therapies.
- This case expands the known clinical spectrum and therapeutic options for CMUSE.
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