NEUROMUSCULAR AND VASCULAR HAMARTOMA SECONDARY TO ENDOMETRIOSIS AT APPENDICEAL TIP MISDIAGNOSED AS APPENDICEAL NEOPLASM: AN EXTREMELY RARE CASE REPORT WITH LITERATURE REVIEW

Abstract

Background: Endometriosis of the appendix is reported to be 1 to 3 % of the total endometriosis. Neuromuscular and vascular hamartoma (NMVH) is a rare lesion with only 27 cases reported in intestine since its initial description in 1982 and only one reported case in the appendix until now. We present a 52-year-old Omani female patient complaining of chronic interm Case presentation: itted abdominal pain. On CT scan (computed tomography scan), there was appendiceal lesion near the tip suggestive of mucinous cystadenoma. Diagnostic laparoscopy with appendectomy was conducted. There was a well-dened lesion with central fatty area at appendiceal tip focally merge with the outer muscle layer and mostly embedded in the mesoappendix. Microscopic examination showed focal area of endometriosis at the outer longitudinal muscle layer of the appendix. This focally merge with a well-dened mass like lesion that is embedded in the mesoappendix. The lesion composed of benign random arranged smooth muscle bers, scattered nerve bundles, some of which showed ganglionic cells including degenerated one, adipose tissue, thin and thick-walled small blood vessels. Features were consistent with NMVH that appeared secondary to the nearby endometriosis. Conclusion: Endometriosis of bowel should be suspected in female with longstanding intermitted abdominal pain. NMVH is extremely rare in appendix, thus clinical and radiological suggestion of such diagnosis is difcult. Secondary neuromuscular hamartoma is reported in association with Crohn's disease as a consequence of the chronic inammation and thus could be suspected secondary to endometriosis