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Eur J Cardiothorac Surg 2002;21:596-597
© 2002 Elsevier Science NL

Letter to the Editor

Multiple aortic valve papillary fibroelastoma: do not miss the other one

Gilles D. Touatia*, Doron Carmia, Henri Sevestreb, Henri Poulaina

a Department of Cardiovascular Surgery, Centre Hospitalier Universitaire d'Amiens, Hôpital Sud, 80054 Amiens Cedex 01, France
b Department of Pathology, Centre Hospitalier Universitaire d'Amiens, Hôpital Sud, 80054 Amiens Cedex 01, France

Received 5 October 2001; received in revised form 5 December 2001; accepted 5 December 2001.

* Corresponding author. Tel.: +33-3-22-45-59-25; fax: +33-3-22-45-53-31
e-mail: gtouati.hms{at}invivo.edu

Key Words: Papillary fibroelastoma • Aortic valve • Nodule of semilunar valve • Lambl's excrescences

About 36% of the 410 cases of papillary fibroelastoma reported in the literature to date are located on the aortic valve. Multiple sites are rare (7.5%), essentially involving separate valves or in the left and right cavities [1]. Only one published case has reported multiple sites on the same aortic valve [2].

Three of the eight patients diagnosed with papillary fibroelastoma in our surgical unit presented double sites on the same valve.

The first patient was a 49-year-old man hospitalized for a complete right hemiplegia. Transesophageal echocardiography (TEE) demonstrated a tumor, 12 mm in diameter, on the non-coronary cusp. Two small growths, with a long axis of 2 mm, on the right coronary leaflet were not revealed by TEE. Histological examination confirmed the diagnosis of three papillary fibroelastomas.

The second patient was a 64-year-old man hospitalized for transient monoparesis. TEE showed an echogenic mass, 7 mm in diameter, attached to the left anterior sigmoid valve. At surgery, we found a non-pedunculated tumor on the left coronary leaflet and enlargement of the nodule of semilunar valve (NSV) on the non-coronary cusp. Histological examination confirmed the diagnosis of typical papillary fibroelastoma for the primary tumor and for the fragment removed from the NSV.

The third case was a 40-year-old woman with an aneurysm of the ascending aorta. At surgery, close examination of the aortic leaflets revealed two growths located on the nodules of the left and the right coronary leaflets. The valve was preserved. Histological examination confirmed a typical fibroelastoma for the larger tumor, and Lambl's excrescence for the smaller tumor.

Based on these extremely unusual findings, several points merit discussion.

Multiple papillary fibroelastomas are rarely reported: only 31 cases have been reported among the 410 published cases of fibroelastoma. In most cases, multiple tumor sites were intraventricular or involving separate cardiac leaflets [3]. Only one case of multiple tumor sites on the aortic valve has been published in the literature [2].

The true incidence of multiple papillary fibroelastomas may be underestimated, as the diagnosis may be missed on TEE. In one of the three cases described here, the diagnosis was incidental, as the tumors were discovered during surgery.

It is not sufficient to treat only the tumor visible macroscopically, as smaller tumors on another aortic cusp may be missed. No cases of recurrence of papillary fibroelastoma after surgical excision, detected by TEE [4], have been reported in the literature, but the recurrence of embolic events has never been estimated; each valve must therefore be carefully examined to exclude any ‘hidden tumors’. Excrescences on valve leaflets or hypertrophic NSV may also need to be resected. The diagnosis can be confirmed by histological examination.

The etiology of papillary fibroelastoma is unknown [4], but our third case presented a surprising finding: coexistence of a papillary fibroelastoma and Lambl's excrescence on the same valve, which constitutes an additional clinical, not histological, argument in support of the hypothesis that these lesions correspond to different stages of the same tumor [5].

References

  1. Grinda J.M., Couetil J.P., Chauvaud S., D'Attelis N., Berrebi A., Fabiani J.N., Deloche A., Carpentier A. Cardiac valve papillary fibroelastoma: surgical excision for revealed or potential embolization. J Thorac Cardiovasc Surg 1999;117(1):106-110.[Abstract/Free Full Text]
  2. Ohushi S., Izumoto H., Kamata J., Sato Y., Kawase T., Ishibashi K., Mukaida M., Nasu M., Eishi K., Kawazoe K., Nakamura S. Aortic valve papillary fibroelastoma in a patient with mitral valve regurgitation. Kyobu Geka 1999;52:1124-1127.[Medline]
  3. Sun J.P., Asher C.R., Yang X.S., Cheng G.G., Scalia G.M., Massed A.G., Griffin B.P., Ratliff N.B., Stewart W.J., Thomas J.D. Clinical and echocardiographic characteristics of papillary fibroelastomas: a retrospective and prospective study in 162 patients. Circulation 2001;103(22):2687-2693.[Abstract/Free Full Text]
  4. Howard R.A., Aldea G.S., Shapira O.M., Kasznica J.M., Davidoff R. Papillary fibroelastoma: increasing recognition of a surgical disease. Ann Thorac Surg 1999;68(5):1881-1885.[Abstract/Free Full Text]
  5. Boone S.A., Campagna M., Walley V.M. Lambl's excrescences and papillary fibroelastomas: are they different?. Can J Cardiol 1992;8(4):372-376.[Medline]




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