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DOI: 10.1148/radiol.2391040056
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(Radiology 2006;239:293-296.)
© RSNA, 2006


Diagnosis Please

Case 93: Thoracic Splenosis1

Amy H. Huang, MD and Kitt Shaffer, MD, PhD

1 From the Department of Radiology, Brigham and Women's Hospital, c/o Carole Dowd, 75 Francis St, Boston, MA 02115 (A.H.H.); and Department of Radiology, Dana-Farber Cancer Institute, Boston, Mass (K.S.). Received January 10, 2004; revision requested March 16; revision received March 23; final version accepted April 8.

Address correspondence to: A.H.H. (e-mail: acho{at}partners.org).


    HISTORY
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 HISTORY
 IMAGING FINDINGS
 DISCUSSION
 References
 
A 58-year-old man with a history of diffuse large B-cell lymphoma who underwent chemotherapy with cyclophosphamide, adriamycin, vincristine, and prednisone 2 years previously was evaluated for restaging. Medical history included basal cell carcinoma of the nose 20 years previously. Laboratory results were normal. Contrast material–enhanced computed tomography (CT) was performed in the chest and abdomen after intravenous administration of 100 mL of 300 mg/mL iohexol (Omnipaque; Amersham Health, Amersham, United Kingdom).


    IMAGING FINDINGS
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 HISTORY
 IMAGING FINDINGS
 DISCUSSION
 References
 
Contrast-enhanced CT performed through the lower thorax revealed multiple well-circumscribed homogeneously enhanced pleural-based nodules in the left lower hemithorax between 2 and 8 mm in size (Fig 1). Deformity of the left T8 and T9 ribs indicated old healed fractures (Fig 1). Transverse contrast-enhanced CT performed through the upper abdomen revealed an absent spleen (Fig 2).


Figure 1
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Figure 1a: Transverse contrast-enhanced CT images of the lower thoracic region. (a) A homogeneously enhanced, well-circumscribed, 6-mm-diameter pleural-based nodule (arrow) is seen in the left hemithorax. Deformity of the left T8 rib (arrowhead) indicates an old healed fracture. (b) A larger homogeneously enhanced pleural-based nodule (arrow) is seen in the left hemithorax at the level of the anterior diaphragm. Deformity of the adjacent T9 rib (arrowhead) indicates an old healed fracture. (c) Multiple homogeneously enhancing pleural-based nodules (arrows) ranging from 2 to 8 mm in size.

 

Figure 1
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Figure 1b: Transverse contrast-enhanced CT images of the lower thoracic region. (a) A homogeneously enhanced, well-circumscribed, 6-mm-diameter pleural-based nodule (arrow) is seen in the left hemithorax. Deformity of the left T8 rib (arrowhead) indicates an old healed fracture. (b) A larger homogeneously enhanced pleural-based nodule (arrow) is seen in the left hemithorax at the level of the anterior diaphragm. Deformity of the adjacent T9 rib (arrowhead) indicates an old healed fracture. (c) Multiple homogeneously enhancing pleural-based nodules (arrows) ranging from 2 to 8 mm in size.

 

Figure 1
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Figure 1c: Transverse contrast-enhanced CT images of the lower thoracic region. (a) A homogeneously enhanced, well-circumscribed, 6-mm-diameter pleural-based nodule (arrow) is seen in the left hemithorax. Deformity of the left T8 rib (arrowhead) indicates an old healed fracture. (b) A larger homogeneously enhanced pleural-based nodule (arrow) is seen in the left hemithorax at the level of the anterior diaphragm. Deformity of the adjacent T9 rib (arrowhead) indicates an old healed fracture. (c) Multiple homogeneously enhancing pleural-based nodules (arrows) ranging from 2 to 8 mm in size.

 

Figure 2
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Figure 2: Transverse CT image of the upper abdomen obtained after administration of oral and intravenous contrast material. The body and tail (arrow) of the pancreas and the upper pole of the left kidney (arrowhead) are visible. Note the absence of the spleen in the left upper quadrant.

 

    DISCUSSION
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 HISTORY
 IMAGING FINDINGS
 DISCUSSION
 References
 
Splenosis involves the autotransplantation of splenic tissue to abnormal locations after splenic injury. Once considered a rare condition, splenosis is now estimated to occur in up to 67% of patients with splenic rupture (1). Splenic tissue implants are found most commonly in the mesentery, peritoneum, and omentum (2). These deposits may vary in number, they can be any shape, and their diameter can range from a few millimeters to 12 cm (1,3,4). Unlike accessory spleens—which are few in number, occur near the splenopancreatic ligament, and are supplied by the splenic artery—autotransplanted splenic tissue derives its blood supply from surrounding tissues (1).

Thoracic splenosis occurs less frequently than abdominal splenosis and may be found in 18% of patients after splenic rupture (3,5). Thoracic splenosis usually represents the sequela of splenic disruption in association with a diaphragmatic tear, which allows implantation of splenic tissue on the left pleura (2). The average interval between initial trauma and discovery of thoracic splenosis is 21 years (5). Yammine et al (5) reviewed 38 cases of thoracic splenosis in the literature and reported a male-to-female ratio of 30:8, which likely reflects the higher rate of trauma in young men.

Symptoms of thoracic splenosis may include pleurisy or recurrent hemoptysis; however, most patients are asymptomatic and thoracic splenosis is discovered incidentally (2,5). Chest CT reveals a solitary pleural-based nodule or mass in 25% of cases and multiple pleural-based nodules in the remaining 75% (5). Pleural-based implants may occur on the parietal or visceral pleura, and they have an attenuation similar to that of the normal spleen (3,4). There has been one report of a subcutaneous splenic implant after chest tube placement and one report of a mediastinal splenic implant in a patient who underwent left lower lobectomy after trauma (6,7).

When necessary, the preoperative diagnosis of splenosis may be established with technetium 99m (99mTc) sulfur colloid scintigraphy of the liver and spleen (1,5). Use of indium 111 (111In)-labeled platelets and 99mTc heat-damaged erythrocytes has been advocated because of increased sensitivity and specificity for splenic sequestration and phagocytosis (5,8). Use of 99mTc sulfur colloid, 111In-labeled platelets, or 99mTc heat-damaged erythrocytes results in specific uptake of the radioactive isotope in splenic tissue.

This case of thoracic splenosis demonstrates the classic CT findings of multiple, variably sized, enhancing pleural nodules within the left hemithorax. Although this patient did not state a history of trauma at the time of initial interpretation, old healed fractures were identified in the left T8 and T9 ribs. These findings, coupled with the absence of the spleen, indicate a history of splenic injury, with resultant thoracic splenosis after diaphragmatic tear. The patient later stated that an injury sustained in a severe motorcycle accident 35 years previously necessitated splenectomy. Furthermore, review of a previously obtained CT image demonstrated stability of the pleural-based implants over a 3-year period.

In the absence of a specific history, detection of multiple enhancing unilateral pleural-based nodules, masses, or both, on CT scans is a nonspecific finding. Differential considerations include pleural metastases (most commonly arising from the lungs, breast, or melanoma), lymphoma, localized fibrous tumor of the pleura, malignant mesothelioma, and invasive thymoma (2,9). This patient, a male nonsmoker, lacked a history of primary metastases arising from the lungs, breast, or melanoma. Fibrous tumors of the pleura usually show substantial enhancement after administration of intravenous contrast material, but they are rarely multiple in nature (2). Likewise, malignant mesothelioma can have a masslike appearance, but it typically does not manifest as multiple discrete nodules (2). Absence of a mediastinal mass also makes invasive thymoma unlikely (2).

In this patient, the history of lymphoma in combination with the CT findings makes lymphoma a differential consideration. Splenectomy is a common treatment of splenic lymphoma (10), whereas pleural lymphoma—which is seen in patients with Hodgkin (11.4%–30.0%) or non-Hodgkin (3.7%–33.0%) disease—can manifest as solitary or multiple pleural nodules (11,12). However, pleural lymphoma is usually accompanied by a pleural effusion (1113) and is frequently seen in conjunction with other evidence of recurrence (14); neither finding was present in this case. In addition, while there are isolated reports of lymphoma demonstrating pronounced enhancement (15), enhancement to the degree seen in this patient (similar to the degree of enhancement expected in splenic tissue) would be unusual. Stability of the pleural nodules over a 3-year period is further evidence against a diagnosis of lymphoma.

Intrathoracic extramedullary hematopoiesis may also be considered in the differential diagnosis because patients with this condition can have unilateral or bilateral smooth, round, contrast-enhanced soft-tissue masses (16) and because many of the conditions that lead to extramedullary hematopoiesis can also cause hypersplenism and necessitate splenectomy. However, intrathoracic extramedullary hematopoiesis is uncommon, and it occurs predominantly in the posterior mediastinum in a paravertebral distribution (1719), often bilaterally. Ultimately, the left-sided distribution of pleural nodules and history of prior trauma indicate a diagnosis of thoracic splenosis in this patient.

Thoracic splenosis is an uncommon finding in clinical practice. Its frequency is likely underestimated because most splenic implants are asymptomatic and are only incidentally discovered at chest radiography or CT. Investigators agree that there is no reason to remove asymptomatic splenic tissue implants (5). In fact, if thoracic implants are the only remaining functioning splenic tissue in a patient, removal of these implants may expose the patient to an increased risk of infection with encapsulated organisms (2,20). A prospective diagnosis of thoracic splenosis helps avoid unnecessary percutaneous biopsy or open surgery. A history of thoracoabdominal trauma, splenectomy, and findings of left-sided pleural-based nodules should indicate a diagnosis of thoracic splenosis.


    FOOTNOTES
 

Part one of this case appeared 4 months previously and may contain larger images.

 


    References
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 HISTORY
 IMAGING FINDINGS
 DISCUSSION
 References
 

  1. Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises: case 29-1995—a 65-year old man with mediastenal Hodgkin's disease and a pelvic mass.
  2. White CS, Meyer CA. General case of the day: thoracic splenosis. RadioGraphics 1998;18:255–257.[Medline]
  3. Normand JP, Rioux M, Dumont M, Bouchard G, Letourneau L. Thoracic splenosis after blunt trauma: frequency and imaging findings. AJR Am J Roentgenol 1993;161:739–741.[Abstract/Free Full Text]
  4. Gentry LR, Brown JM, Lindgren RD. Splenosis: CT demonstration of heterotopic autotransplantation of splenic tissue. J Comput Assist Tomogr 1982;6:1184–1187.[Medline]
  5. Yammine JN, Yatim A, Barbari A. Radionuclide imaging in thoracic splenosis and a review of the literature. Clin Nucl Med 2003;28:121–123.[CrossRef][Medline]
  6. Madjar S, Weissberg D. Thoracic splenosis. Thorax 1994;49:1020–1022.[Abstract/Free Full Text]
  7. Renne G, Coci A, Biraghi T, et al. Fine needle aspiration of thoracic splenosis: a case report. Acta Cytol 1999;43:492–494.[Medline]
  8. Armas RR. Clinical studies with spleen-specific radiolabelled agents. Semin Nucl Med 1985;15:260–275.[CrossRef][Medline]
  9. Wold PB, Farrell MA. Pleural nodularity in a patient with pyrexia of unknown origin. Chest 2002;122:718–720.[Free Full Text]
  10. Grosskreutz C, Troy K, Cuttner J. Primary splenic lymphoma: report of 10 cases using the REAL classification. Cancer Invest 2002;20:749–753.[CrossRef][Medline]
  11. Bonomo L, Feragalli B, Sacco R, Merlino B, Storto ML. Malignant pleural disease. Eur J Radiol 2000;34:98–118.[CrossRef][Medline]
  12. Schmutz GR, Fisch-Ponsot C, Regent D, Sylvestre J. Computed tomography (CT) and magnetic resonance imaging (MRI) of pleural masses. Crit Rev Diagn Imaging 1993;34:309–383.[Medline]
  13. Malatskey A, Fields S, Libson E. CT appearance of primary pleural lymphoma. Comput Med Imaging Graph 1989;13:165–167.[CrossRef][Medline]
  14. North LB, Libshitz HI, Lorigan JG. Thoracic lymphoma. Radiol Clin North Am 1990;28:745–762.[Medline]
  15. Pombo F, Rodriguez E, Caruncho MV, Villalva C, Crespo C. CT attenuation values and enhancing characteristics of thoracoabdominal lymphomatous adenopathies. J Comput Assist Tomogr 1994;18:59–62.[Medline]
  16. De Backer AI, Zachee P, Vanschoubroeck IJ, Mortele KJ, Ros PR, Kockx MM. Extramedullary paraspinal hematopoiesis in hereditary spherocytosis. JBR-BTR 2002;85:206–208.
  17. Martin J, Palacio A, Petit J, Martin C. Fatty transformation of thoracic extramedullary hematopoiesis following splenectomy: CT features. J Comput Assist Tomogr 1990;14:477–478.[Medline]
  18. Dunnick NR. Image interpretation session: 1999—extramedullary hematopoiesis in a patient with beta thalassemia. RadioGraphics 2000;20:266–268.[Medline]
  19. Scott WW Jr, Fishman EK. Extramedullary hematopoiesis mimicking the appearance of carcinomatosis or peritoneal mesothelioma: computed tomography demonstration. Gastrointest Radiol 1990;15:82–83.[CrossRef][Medline]
  20. Katranci N, Parildar M, Goksel T, Savas R, Alper H. Quiz case of the month: posttraumatic intrathoracic splenosis. Eur Radiol 1998;8:151–152.[CrossRef][Medline]
Congratulations to the 163 individuals and two resident groups who submitted the most likely diagnosis (splenosis) for Diagnosis\ Please, Case 93. The names and locations of the individuals and resident groups, as submitted, are as follows:

Individual Responses

Hisashi Abe, Osaka, Japan
Gholamali Afshang, MD, Tinley Park, Ill
Oguz Akin, MD, New York, NY
Albert J. Alter, Madison, Wis
Nabil Ammouri, MD, Zahle, Lebanon
Dr Louisa Azizi, Paris, France
Sanjay Bhat, Temple, Tex
Mark A. Bisesi, Bloomington, Ind
Dr Adrian Brady, FFRRCSI, Cork, Ireland
Bill Breidahl, Perth, Australia
Eric L. Bressler, MD, Minnetonka, Minn
Jorge Brito, MD, Coimbra, Portugal
Daniel F. Broderick, MD, Jacksonville, Fla
Lynn S. Broderick, MD, Madison, Wis
Ghislain Brousseau, Charlesbourg, Quebec, Canada
Dr Andrea Bruscagnin, Venezia, Italy
Michael P. Buetow, MD, Okemos, Mich
Peter Buetow, MD, Bellingham, Wash
Stephen J. Buetow, MD, Evans, Ga
Derek A. Burdeny, MD, Omaha, Neb
Jeffrey J. Bush, MD, Eustis, Fla
Ozgur Cakmak, Antalya, Turkey
Paula Campos, MD, Cascais, Portugal
Christopher Chu, Cabarita, New South Wales, Australia
Jay M. Colby, MD, Stonington, Conn
John E. Connolly, Jr, MD, Lincolnwood, Ill
Neal R. Conti, MD, Seattle, Wash
Mark A. Cooper, FRACR, Melbourne, Australia
Theresa M. Corrigan, Louisville, Ky
Anil Kumar Dasyam, Pittsburgh, Pa
Marc G. de Baets, MD, Lugano, Switzerland
Dr Gerard de Geer, MD, Geneva, Switzerland
Andrew Deibler, MD, Winston-Salem, NC
Thaworn Dendumrongsup, MD, Songkla, Thailand
Bart D'herde, Hasselt, Belgium
Nam Ky Do, Duluth, Ga
Morgan G. Dunne, MD, San Antonio, Tex
Brett Elicker, San Francisco, Calif
Seyed Emamian, MD, PhD, Rockville, Md
Robert E. Epstein, MD, Belle Mead, NJ
Ana Sofia Ferreira, MD, Aveiro, Portugal
Nancy E. Fitzgerald, MD, Pearland, Tex
Francis Flaherty, MD, Ridgefield, Conn
Sean M. Flynn, Bloomington, Ind
Ángeles Franco, Madrid, Spain
Matthew A. Frick, MD, Rochester, Minn
Akira Fujikawa, MD, Tokyo, Japan
Ann S. Fulcher, MD, Richmond, Va
Bill Gallmann, MD, Shreveport, La
Gilles Genin, MD, Annecy, France
Vidisha Ghole, MD, Dallas, Tex
Alvaro Gomez Naar, Salta, Argentina
Eric Goodman, MD, San Diego, Calif
Christopher Govea, MD, Houston, Tex
Thomas Grant, DO, Chicago, Ill
Navraj S. Grewal, MD, Elmhurst, Ill
D. Joseph Grunz, MD, Ladue, Mo
Flavius Guglielmo, MD, Basking Ridge, NJ
Pramod Gupta, MD, Arlington, Tex
Irith Hadas-Halpern, Jerusalem, Israel
Ferris M. Hall, MD, Boston, Mass
Howard Harvin, Scottsdale, Ariz
Maureen Heldmann, MD, Shreveport, La
Raúl Hernández Muñiz, Madrid, Spain
Ronald J. Homer, MD, Weston, Conn
Alberto Iaia, MD, Wilmington, Del
Joao Rodrigues Inacio, Lisbon, Portugal
William Jackson, MD, Tacoma, Wash
Kiriakos Kalampoukas, Kozani, Greece
Nurettin Katranci, MD, Antalya, Turkey
Craig D. Kesack, Doylestown, Pa
Jay Kikut, MD, Burlington, Vt
Takuji Kiryu, MD, Gifu, Japan
Riwa Kishimoto, Chiba, Japan
Steven A. Klein, MD, Shrewsbury, Mass
Yoshihisa Kurosaki, MD, Tokyo, Japan
Stefanos Lachanis, MD, Athens, Greece
Alexis Lacout, MD, Paris, France
Mario Laguna, West Allis, Wis
James F. Lally, MD, Newark, Del
Iñigo Lecumberri, Bilbao, Spain
Raisa Lev, Orange, Calif
Peter Leyman, MD, Aalst, Belgium
John T. Lim, MD, Newport Coast, Calif
Alexander P. LoRusso, MD, Santa Fe, NM
Patricia Lowry, MD, Richmond, Va
Matthias Maier, MD, Basel, Switzerland
Stephen Manghisi, MD, Closter, NJ
N. B. S. Mani, MD, Nassau, Bahamas
Michael B. Martin, MD, Austin, Tex
Maria Jesús Martínez, Valencia, Spain
Frank McKowne, MD, Vancouver, Wash
Edward Menges, Aptos, Calif
Koen Mermuys, MD, Heverlee, Belgium
Ur Metser, MD, Tel-Aviv, Israel
Jonathan Meyer, MD, Chicago, Ill
Michael P. Meyers, MD, FRCPC, Winnipeg, Manitoba, Canada
Manabu Minami, MD, Ibaraki, Japan
Ari Mintz, MD, Lake Forest, Ill
Mansour Mirfakhraee, MD, Shreveport, La
Dr Thomas Moser, Strasbourg, France
Tammam Nehme, East Wenatchee, Wash
Karl F. R. Neufang, MD, Euskirchen, Germany
Mizuki Nishino, MD, Boston, Mass
Hiroshi Nobusawa, Tokyo, Japan
Edward S. Oh, Tucson, Ariz
Michael T. O'Loughlin, MD, West Hartford, Conn
Sanford M. Ornstein, MD, Phoenix, Ariz
Ann B. Owen, MD, Murfreesboro, Tenn
David M. Panicek, MD, New York, NY
Rosaleen B. Parsons, MD, Philadelphia, Pa
Narendrakumar P. Patel, MD, Newburgh, NY
Suresh K. Patel, MD, Chicago, Ill
Ernesto Oscar Pearson, MD, Córdoba, Argentina
Juan Carlos Pernas, MD, Barcelona, Spain
David Pham, MD, Toronto, Ontario, Canada
Ivan Pilate, MD, Mechelen, Belgium
Sandra Polin, Washington, DC
Tom Powers, MD, Nashville, Tenn
Thomas A. Predey, MD, Lemont, Ill
Shawn P. Quillin, MD, Charlotte, NC
Ilangovan Rajapandian, Harrow, Middlesex, United Kingdom
Dr Anuradha T. Rao, Houston, Tex
Daniel Rappaport, MD, FRCPC, Toronto, Ontario, Canada
Ryan Rebello, MD, Dundas, Ontario, Canada
Enrique Remartinez Escobar, MD, Melilla, Spain
Matt Rheinboldt, Nashville, Tenn
Mathieu Rodallec, Paris, France
Leon Rubinsztain, Atlanta, Ga
Kris Saadeh, Mount Pleasant, SC
Eloísa Santos Armentia, Vigo, Spain
Dr Robert Sauer, St Poelten, Austria
Anthony J. Scuderi, MD, Johnstown, Pa
Mustafa Secil, MD, Izmir, Turkey
Akram Shaaban, MD, Salt Lake City, Utah
Robert H. Sherrier, MD, Boulder, Colo
Taro Shimono, MD, Osaka, Japan
Grady Shue, Bethesda, Md
James D. Sprinkle, Jr, MD, Spotsylvania, Va
Paul Stark, MD, La Jolla, Calif
Glenn Strome, MD, San Francisco, Calif
Kouichi Sugiyama, Hamamatsu, Japan
Norio Takahashi, MD, Fukui, Japan
Dr Francisco Tardáguila, Vigo, Spain
Douglas L. Teich, MD, Brookline, Mass
Philippe Thoma, MD, Brussels, Belgium
D. Dean Thornton, MD, Birmingham, Ala
Eugene Tong, MD, Austin, Tex
Meriç Tüzün, Ankara, Turkey
Hiroyuki Ueda, Kyoto, Japan
Piet Vanhoenacker, MD, Moorsel, Belgium
Christopher Vittore, MD, Rockford, Ill
Chan Kam Wai, Chai Wan, Hong Kong
Yukari Wakabayashi, MD, Tokyo, Japan
Thomas Waslen, Saskatoon, Saskatchewan, Canada
Lori Watumull, MD, Dallas, Tex
William G. Way, Jr, Raleigh, NC
Steven T. Welch, MD, Kansas City, Mo
Ensar Yekeler, Istanbul, Turkey
Satoru Yoshida, MD, Muroran City, Japan
Kaneko You, MD, Gifu, Japan
Joe Yut, Olathe, Kan
Yu Zhang, San Francisco, Calif

Resident group responses

Hospital of the University of Pennsylvania Radiology Residents, Philadelphia, Pa
Oregon Health & Science University Radiology Residents, Portland, Ore




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