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J Thorac Cardiovasc Surg 2005;130:1724-1726
© 2005 The American Association for Thoracic Surgery
Brief Communications |
a Institute for Cardiovascular Diseases,
b Institute for Histology and Embryology
c Institute for Pathology,
d Institute for Enocrinology, UC Clinical Centre of Serbia, Serbia and Montenegro
* Address for reprints: Mladen J. Kocica, MD, UC Clinical Centre of Serbia, Institute for Cardiovascular Diseases, Clinic for Cardiac Surgery, 8th Kosta Todorovic St, 11000 Belgrade, Serbia and Montenegro (Email: kocica{at}sezampro.yu).
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Intracardiac leiomyomatosis (ICL) is a rare and dangerous form of intravenous leiomyomatosis (IVL).
1-5
The case of ICL presented here depicts some diagnostic and surgical difficulties that may arise from the rarity and complexity of this condition. Further, to our best knowledge, neither the mass nor the growth rate of the abdominal part of the presented ICL has been described in the available literature. Finally, this case and extensive literature search allowed us to clarify some important questions pertaining to this entity.
A 36-year-old white woman (multigravid, biparous), who underwent a right salpingo-oophorectomy 7 years before admission, presented with a recurrent growing pelvic mass, dyspnea, fatigue, weight loss, ascites, leg swelling, and dysmenorrhea.
Abdominal and pelvic ultrasound revealed a right-sided, uterine adnexal tumor (5 x 11 cm) penetrating the inferior vena cava (IVC) (Figure 1, a). After a clinical episode of severe dyspnea, tachycardia, and syncope, echocardiography revealed the presence of a mass in the right atrium, originating from the IVC and protruding across the tricuspid valve throughout the cardiac cycle. The patient was transferred for "palliative" cardiac surgery with a presumptive diagnosis of recurrent ovarian malignancy with intracardiac extension.
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Standard and immunohistochemical analyses (Figure 1, b-d) indicated IVL. Histologic diagnosis was confirmed using international teleconsultation facilities (Department of Pathology, Thoraxklinik, Heidelberg, Germany).
A second-stage operation was scheduled to remove abdominal and intracaval tumor remnants. During 1 month, between 2 operations, the abdominal mass doubled in size, resembling terminal gravidity, without any other clinical worsening in the patient's condition except increased leg swelling.
On median laparotomy, the right colon and small bowel were mobilized and Kocherization of duodenum was performed to gain safe access to the IVC. Vessel loops were placed around the IVC below the renal veins and inferiorly, below the orifice of the right ovarian vein. After blunt mobilization of the retroperitoneal mass, elliptical excision of the IVC around the right ovarian vein orifice allowed traction delivery of the intracaval portion of the tumor. The IVC was closed with linear suture. The entire tumor (8.5 kg, 45 x 20 x 20 cm) and the uterus were removed en bloc (Figure 1, d).
The patient's postoperative course was complicated with excessive bleeding, and relaparotomy was necessary. Hemostatic sutures were placed on the uterine cervical stump, and control of the bleeding site was fully accomplished.
The rest of the postoperative course was uneventful. Control abdominal ultrasonography and echocardiography revealed no visible intravascular tumor remnants. Nine months after the patient's discharge, no recidivant or metastatic masses were detected by ultrasonography.
Since the first description of ICL by Durck and Hormann in 1907, 113 cases have been published.
2,3
Before 1980, when Timmis and colleagues extracted an ICL,
2
all attempts to do so were fatal. A new era in treatment of IVL and ICL began in 1982, when Ariza and colleagues performed the first successful 2-stage total resection.
2
We performed a comprehensive literature search, including all available electronic and paper sources, together with the references in each publication, to identify all reported cases of ICL. A compendium of information from isolated reports is summarized in Table 1. We believe that ICL is not so rare an entity as previously thought (during the past 2 years, 16% of all ICL cases were reported). A higher index of clinical suspicion with the availability of contempory imaging tools (ie, abdominal and cardiac ultrasound, computed tomography, magnetic resonance imaging, and venography) will lead to an increased number of reports in the future.
2-5
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Because a complete removal of the tumor is mandatory,
1-5
an adequate preoperative diagnosis should provide detailed information about the tumor localization, extravascular and intravascular diameters, site of vascular entry, and patency of iliac and femoral veins.
5
The optimum and safest strategy is a single or 2-stage procedure with an abdominal approach and subsequent thoracic approach. The abdominal stage should ensure complete extravascular tumor excision and control of the vascular entry site(s). The thoracic stage, preferably with short total CPB on the beating heart, should allow easy tumor withdrawal from the right cardiac chambers and IVC.
1-5
References
This article has been cited by other articles:
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  N. Fasih, A. K. Prasad Shanbhogue, D. B. Macdonald, M. A. Fraser-Hill, D. Papadatos, A. Z. Kielar, G. P. Doherty, C. Walsh, M. McInnes, and M. Atri Leiomyomas beyond the Uterus: Unusual Locations, Rare Manifestations1 RadioGraphics, November 1, 2008; 28(7): 1931 - 1948. [Abstract] [Full Text] [PDF]
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 M. Coganow, B. M. Das, E. Chen, and J. A. Crestanello Single-stage resection of a mixed endometrial stromal sarcoma and smooth muscle tumor with intracardiac and pulmonary extension. Ann. Thorac. Surg., October 1, 2006; 82(4): 1517 - 1519. [Abstract] [Full Text] [PDF]
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