DOI: 10.12890/2024_004922
A case report of mesenteric panniculitis and fibrous dysplasia with primary metabolic syndrome
Authors
birgit.khandalavala@unmc.edu
Keywords
Mesenteric panniculitis, fibrous dysplasia, metabolic syndrome, obesityAbstract
Introduction: Mesenteric panniculitis (MP) is a rare clinically diagnosed disease resulting from fibro-inflammation in the intestinal adipose tissue. Precedent intra-abdominal inflammation or trauma is often evident. Fibrous dysplasia (FD) is a benign uncommon childhood disease of the bones, with fibrous connective tissue replacing bone tissue. FD can cause pain, disfigurement, and entrapment of critical neighbouring structures. Adult-onset craniofacial involvement is rare. Metabolic syndrome is characterized by obesity and chronic subclinical inflammation, promoting abnormal cellular fibrosis and proliferation. The concomitant incidence of MP and FD in an older female adult with metabolic syndrome is unprecedented. An aberrant metabolic contributory pathophysiology for both MP and FD could be postulated.
Case Report: A 60-year-old Caucasian woman with primary long-standing well-treated metabolic syndrome with obesity presented for revision bariatric surgery. She had previous intra-abdominal surgeries and occasional intermittent abdominal pain. The surgery was abandoned due to extensively inflamed intestinal mesentery. Radiological studies and intra-abdominal biopsy confirmed MP. She subsequently developed pronounced unilateral facial swelling and radiological studies confirmed FD. Treatment has been suboptimal for both MP and FD. She developed venous thromboembolism on tamoxifen treatment for MP. The patient currently reports abdominal pain and swelling from MP, and facial disfigurement, pain, and tinnitus from FD. Blood markers for inflammation have remained consistently high, and there is statin intolerance. Most recently liver steatosis and diabetes have developed.
Conclusion: A case of MP and FD with primary metabolic syndrome is unique. An increased awareness of such rare cases could lead to further research, and critically needed treatments to optimize outcomes.
References
- Sulbaran M, Chen FK, Farraye FA, Hashash JG. A Clinical Review of Mesenteric Panniculitis. Gastroenterol Hepatol (NY) 2023;19:211-218.
- Pinheiro F, Rgo A, ArajoFilho I. Mesenteric panniculitis in the elderly - update on diagnostic and therapeutic approach. Int J Surg Med 2016;2:127.
- Kim HY, Shim JH, Heo CY. A rare skeletal disorder, fibrous dysplasia: a review of its pathogenesis and therapeutic prospects. Int J Mol Sci 2023;24:15591.
- Dobrowolski P, Prejbisz A, Kurylowicz A, Baska A, Burchardt P, Chlebus K, et al. Metabolic syndrome—A new definition and management guidelines. Arter Hypertens 2022;26:99–121.
- Mendes PA, Ferreira DM, Temido H, Pina R, de Carvalho A. Mesenteric panniculitis in Sjögren’s syndrome: a new systemic manifestation to consider? Eur J Case Rep Intern Med 2018;5(4).
- Alkhaibary A, Alassiri AH, Alsalman M, Edrees S. Unusual presentation of fibrous dysplasia in an elderly patient. J Radiol Case Rep 2019;13:26.
- Eze VN, Halligan S. Mesenteric panniculitis: a clinical conundrum. Br J Radiol 2023;96:20211369.