Antiphospholipid Antibody Syndrome an Unusual Case Presentation
Joann Lohr, Emily Wright, Thomas Panke.
Trihealth, Cincinnati, OH, USA.
OBJECTIVE- To increase awareness of antiphospholipid antibody syndromes by describing an atypical case of antiphospholipid antibody syndrome presenting as ischemic toxic megacolon progressing to complete rectal occlusion in a patient who had not had any prior pelvic surgery
METHODS - Case report and retrospective review of literature of antiphospholipid antibodies and Asherson's syndrome.
RESULTS- A 66 year old G2P2 smoker with a history of HTN, bipolar disorder, COPD, TIA and PVD presented to the emergency room with a three day history of abdominal pain, nausea, and vomiting. Physical exam and imaging suggested toxic megacolon which warranted exploratory laparotomy. At that time a subtotal abdominal colectomy and Brooke Ileostomy were performed. Pathology was consistent for toxic megacolon with complete transmural devitalization and dilatation. Features of acute transmural ischemic colitis involving rare mesenteric intravascular thrombosis of medium sized vessels were present. Hypercoagulable profile revealed a mildly elevated lupus anticoagulant which was indicative of antiphospholipid antibody syndrome.
The patient was anticoagulated for one year. After which she returned to the operating room for ileostomy reversal. Intraoperative pathology showed acute patchy inflammation and mucosal ulceration which prompted resection of her remaining sigmoid colon. Her rectum was completely occluded preventing ostomy takedown.
CONCLUSIONS- Antiphospholipid antibody syndrome (APS) is an autoimmune disorder involving antibodies directed towards phospholipids found throughout the vasculature. Presence of antiphospholipid antibodies increases the tendency of blood to form clots which can obstruct vasculature and cause organ ischemia. APS can easily be missed as part of a differential diagnosis. Although more commonly involving renal and pulmonary vascular occlusions, any organ system including the skin may be affected. As in the case of this patient, true toxic megacolon may not be the result of inflammatory bowel syndrome, but may instead be secondary to APS. Asherson's syndrome, the most severe form of APS can be rapidly progressive and have fatal consequences including a risk of DIC and tissue necrosis in the GI tract. APS must therefore be considered in a broad differential diagnosis when there is a possibility of a hypercoagulable state and unusual disease presentations.
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