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Sclerosing mesenteritis: An ergot-related complication of pergolide therapy in Parkinson's disease?

โœ Scribed by Kersi J. Bharucha; Steve M. Blevins


Publisher
John Wiley and Sons
Year
2008
Tongue
English
Weight
38 KB
Volume
23
Category
Article
ISSN
0885-3185

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โœฆ Synopsis


Pergolide mesylate, an ergot-related D1/D2 dopamine receptor agonist, was introduced in 1989 for the treatment of Parkinson's disease (PD). Following reports of pleural, cardiac, and retroperitoneal fibrosis, 1-3 it was recently withdrawn from the US market. We report a patient with PD on pergolide therapy for 11 years who developed small bowel obstruction secondary to sclerosing (retractile) mesenteritis, a rare, but serious inflammatory condition of the small bowel mesentery. It often poses a diagnostic challenge and may mimic malignancy. 4 Sclerosing mesenteritis may be idiopathic, or occur in association with inflammatory disorders including retroperitoneal fibrosis, sclerosing cholangitis, Riedel's thyroiditis, and orbital pseudotumor. 5 It has not been previously reported in the setting of chronic pergolide therapy.

The patient was diagnosed with PD at age 44, when he presented with a resting tremor in his right hand and dragging of his right foot. Following initial therapy with amantadine 200 mg/day, trihexyphenidyl 6 mg/day, and pergolide 3 mg/day, levodopa extended-release (300 mg daily) with carbidopa was added 2 years later. At age 50, he developed motor fluctuations with peak-dose dyskinesias and freezing of gait. Bilateral subthalamic deep brain stimulators were implanted at age 53, with improvement, and he was given carbidopa/l-dopa immediate release (l-dopa 300 mg/day) with pergolide 3 mg/day. Trihexyphenidyl was discontinued. At age 54, he had several bouts of intermittent diarrhea with abdominal cramping. He was not on entacapone, tolcapone, or other medications known to cause diarrhea. Following an upper gastrointestinal endoscopy, he received antibiotics and a proton-pump inhibitor for presumed Helicobacter pylori infection. The diarrhea and intermittent abdominal pain persisted, and he was admitted 4 months later with obstipation. Plain abdominal films showed dilated loops of small bowel with fluid levels, consistent with ileus, which improved with conservative management. At age 55, he was readmitted with small-bowel obstruction. An abdominal CT scan showed an 8 cm mass in the mesentery, suggestive of malignancy. At exploratory laparotomy matted loops of bowel and mesentery were resected, leaving only 70 cm of distal ileum after an end-to-end anastomosis. Histology showed diffuse mesenteric sclerosing fibrosis, lipogranulomas, calcification, and chronic inflammation with adhesions, consistent with sclerosing mesenteritis. There was no malignancy. Renal function, liver patients on pergolide who have unexplained abdominal pain, change in bowel habit, or small-bowel obstruction.


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