TY - JOUR
T1 - A CASE REPORT OF MESENTERIC PANNICULITIS AND FIBROUS DYSPLASIA WITH PRIMARY METABOLIC SYNDROME
AU - Khandalavala, Birgit
AU - Cuppett, Ryder
N1 - Publisher Copyright:
© 2024 SMC Media Srl. All rights reserved.
PY - 2024
Y1 - 2024
N2 - 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.
AB - 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.
KW - fibrous dysplasia
KW - Mesenteric panniculitis
KW - metabolic syndrome
KW - obesity
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U2 - 10.12890/2024_004922
DO - 10.12890/2024_004922
M3 - Article
C2 - 39651389
AN - SCOPUS:85211037833
SN - 2284-2594
VL - 11
JO - European Journal of Case Reports in Internal Medicine
JF - European Journal of Case Reports in Internal Medicine
IS - 12
ER -