Case Report
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World J Gastroenterol. Mar 14, 2013; 19(10): 1657-1660
Published online Mar 14, 2013. doi: 10.3748/wjg.v19.i10.1657
PEComa of the colon resistant to sirolimus but responsive to doxorubicin/ifosfamide
Wolfgang Scheppach, Nikolaus Reissmann, Thomas Sprinz, Ekkehard Schippers, Bjoern Schoettker, Justus G Mueller
Wolfgang Scheppach, Nikolaus Reissmann, Department of Medicine, Juliusspital Wuerzburg, D-97070 Wuerzburg, Germany
Thomas Sprinz, Ekkehard Schippers, Department of Surgery, Juliusspital Wuerzburg, D-97070 Wuerzburg, Germany
Bjoern Schoettker, Onkologische Schwerpunktpraxis, D-97070 Wuerzburg, Germany
Justus G Mueller, Department of Pathology, University of Wuerzburg, 97070 Würzburg, Germany
Author contributions: Scheppach W and Reismann N diagnosed and treated the patient in hospital; Sprinz T and Schippers E operated on the patient; Schoettker B treated the outpatient; Mueller JG analysed the tumor specimens histologically; all authors contributed significantly to the acquisition, analysis and interpretation of data; Scheppach W drafted the article; all coauthors revised it critically and finally approved it for publication.
Correspondence to: Wolfgang Scheppach, MD, Department of Medicine, Juliusspital Wuerzburg, Juliuspromenade 19, D-97070 Wuerzburg, Germany. gastroenterologie@juliusspital.de
Telephone: +49-931-3931701   Fax: +49-931-3931702
Received: November 17, 2012
Revised: December 11, 2012
Accepted: January 17, 2013
Published online: March 14, 2013
Processing time: 117 Days and 19.7 Hours
Abstract

A 23-year-old male presented with a three-week-history of crampy abdominal pain and melaena. Colonoscopy revealed a friable mass filling the entire lumen of the cecum; histologically, it was classified as perivascular epithelioid cell tumor (PEComa). An magnetic resonance imaging scan showed, in addition to the primary tumor, two large mesenteric lymph node metastases and four metastatic lesions in the liver. The patient underwent right hemicolectomy and left hemihepatectomy combined with wedge resections of metastases in the right lobe of the liver, the resection status was R0. Subsequently, the patient was treated with sirolimus. After 4 mo of adjuvant mammalian target of rapamycin inhibition he developed two new liver metastases and a local pelvic recurrence. The visible tumor formations were again excised surgically, this time the resection status was R2 with regard to the pelvic recurrence. The patient was treated with 12 cycles of doxorubicin and ifosfamide under which the disease was stable for 9 mo. The clinical course was then determined by rapid tumor growth in the pelvic cavity. Second line chemotherapy with gemcitabine and docetaxel was ineffective, and the patient died 23 mo after the onset of disease. This case report adds evidence that, in malignant PEComa, the mainstay of treatment is curative surgery. If not achievable, the effects of adjuvant or palliative chemotherapy are unpredictable.

Keywords: Perivascular epithelioid cell tumor; Colon; Liver metastases; Mammalian target of rapamycin inhibitor; Sirolimus; Chemotherapy; Doxorubicin; Ifosfamide