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CASE REPORT Asymptomatic thoracic splenosis after thoracoabdominal trauma: establishing a diagnosis

Richard A. Beekmana,*, Brian Louieb, Ravinder Singha, Samih Salamac, John D. Millerd

aDepartment of Surgery, McMaster University Medical Centre, 319 MacNab St N, Hamilton, Ont., Canada L8L 1K7 bDivision of Foregut and Pulmonary Surgery, University of Southern California, Los Angeles, CA, USA cDepartment of Pathology, St Joseph’s Healthcare, Hamilton, Ont., Canada dDepartment of Thoracic Surgery, St Joseph’s Healthcare, Hamilton, Ont., Canada

Accepted 13 December 2004

Introduction Case report

Splenosis is defined as autotransplantation of viable A 58-year-old male was referred for evaluation of splenic tissue after . Cases of intracra- multiple pulmonary nodules suspicious for malig- nial,14 subcutaneous,19 pulmonary,15 pleural, ovar- nancy. Thirty-five years earlier, he had been ian, scrotal,10 hepatic, pancreatic and peritoneal20 involved in a motor vehicle crash and was diagnosed splenosis have been reported in the literature after with a fractured mandible, multiple left-sided rib traumatic disruption. Splenosis is common, and fractures, a pulmonary laceration with left haemo- when present, typically presents with numerous pneumothorax, ruptured left diaphragm with intra- lesions (up to 100).7 The majority of these lesions thoracic stomach and splenic laceration. After are found incidentally, masquerading as malignant initial assessment and stabilization he underwent lesions, but a small number have presented with and repair of the left diaphragmatic gastrointestinal or intra-peritoneal bleeding, bowel laceration. He recovered uneventfully from these obstruction,1 ureteral colic,18 haemoptysis, or neu- and was discharged. Recently, he developed rologic symptoms. The crucial issue with asympto- an upper respiratory tract infection and received a matic splenosis is to establish a diagnosis of chest radiograph (CXR) as part of his investigations. splenosis while excluding malignant lesions in an Apical and basal pleural-based nodules were iden- efficient and minimally invasive fashion. We report tified CXR (Fig. 1) and further clarified by computed a case of multiple pleural splenosis and present it in tomography (CT) scan of the chest (Fig. 2). an historical context. Attempted image guided trans-thoracic aspiration of these lesions in a peripheral hospital was aborted * Corresponding author. Tel.: +1 905 525 8029; because of fear of complications. Video assisted fax: +1 905 521 6156. thoracoscopic surgery (VATS) biopsy of the lesions E-mail address: [email protected] (R.A. Beekman). was performed. Intra-operative frozen section con-

1572-3461 # 2004 Elsevier Ltd. Open access under CC BY-NC-ND license. doi:10.1016/j.injury.2004.12.050 284 R.A. Beekman et al.

Figure 1 Pre-operative CXR demonstrating left apical pleural based lesion.

firmed a diagnosis of thoracic splenosis (Fig. 3). A post-operative tagged RBC scan demonstrated two intra-abdominal splenosis in addition to the intra- pleural basilar splenosis (Fig. 4). The patient was discharged on post operative day 2 and was asymp- tomatic at two subsequent clinic visits.

Figure 2 CT Scan thorax demonstrating pleural based Discussion lesions near apex and posterior to hilum.

The first case of thoracic splenosis was diagnosed at tion as they are not universally mentioned in the case autopsy in 1937 by Shaw and colleagues,16 in a 20- reports. When patients sustain high energy blunt or year-old male patient who succumbed to overwhelm- penetrating trauma, splenosis can cross normal ana- ing sepsis. Some time previous he had been in an tomic boundaries and develop in unusual locations. accident and sustained splenic trauma necessitating removal. Only 17 years earlier von Kuttner described the phenomenon of intra-abdominal autotransplan- tation ofsplenic tissueina single human case andinan experimental canine model. While splenosis within the abdominal cavity is relatively common, occur- rence outside the abdominal cavity is unusual. Thor- acicsplenosiscanbesuspectedifthefollowingfactors are present: a history of trauma, splenic injury, and diaphragmatic injury. On reviewing the literature we found 53 previous cases of intrathoracic splenosis in 47 articles. All cases of thoracic splenosis are left sided presumably developing from direct spread of splenic tissue via a diaphragmatic laceration rather Figure 3 Histology of apical lesion demonstrating sple- than haematogenously. Diaphragmatic lacerations nic parenchyma with overlying pleura. Note malpighian may be subclinical or forgotten at time of presenta- follicle. Asymptomatic thoracic splenosis after thoracoabdominal trauma 285

intrathoracic masses. Video assisted thoracic sur- gery (VATS) is a relatively recent adaptation of minimal access surgery to the thoracic region. Only a single previous report was found which success- fully used VATS for biopsy of thoracic splenosis.17 VATS techniques are dependent upon the creation of a working space between the parietal and visceral pleura. Trauma and inflammatory changes can oblit- erate this potential space with dense adhesions necessitating conversion to conventional open sur- gery. Thoracotomy was a frequently used diagnostic tool with indication for surgery being the need for histologic confirmation of malignancy. There are three reports of malignancy concur- rent with intra-thoracic splenosis (prevalence 5.6%) and a single report of splenosis presenting as an oesophageal tumor. The first case was a right lower lobe squamous cell carcinoma diagnosed by percutaneous needle biopsy with left multiple pleural nodules proven to be splenosis on 99mTc scanning.2 There were double reports of the next case, a 44-year-old man with a diffuse reticular- Figure 4 Post operative 99mTc scan demonstrating two nodular pattern and left pleural based mass. Thor- intra-abdominal splenosis as well as a remaining basal 99m thoracic splenosis. acic splenosis was demonstrated with Tc scinti- graphy and core biopsy, while adenocarcinoma was diagnosed by transbronchial biopsy.6,9 Finally, a right lower lobe T1N0M0 squamous cell carcinoma Of the reported cases, the median age at time of was misdiagnosed as a T1N3M0 because of ‘‘massive presentation was 45 years with a range of 15—79. All left mediastinal ’’.11 The sup- but eight cases were male, reflecting the male posed left sided lymphadenopathy was discovered predominance in the trauma population. The aver- to be benign splenosis on further investigation. A age time from injury to presentation was 21 years 48-year-old woman presented with a suspected (range 1—49). All lesions were asymptomatic and enlarging oesophageal leiomyoma just above the were found while investigations were performed for left diaphragm. At the time of thoracotomy, his- angina, vague chest pain, musculoskeletal pain, or tologic examination identified the suspected leio- when routine imaging was performed. A single myoma as a splenosis.8 These cases highlight the exception presented with haemoptysis which crucial importance of recognizing and differentiat- resolved upon removal of intrathoracic splenosis.3 ing splenosis from primary and secondary neoplas- Traumatic cases were secondary to sharp or pene- tic disease as this affects staging, treatment and trating thoraco-abdominal trauma in 35/54 (64.8%) prognosis of cancer patients. cases including gunshot and shrapnel wounds and Removal of splenosis, while necessary for diag- stabbings. A further 13/54 (24.1%) cases were sec- nosis, may be detrimental to a patient. It remains ondary to blunt trauma including falls and motor unknown if splenosis can return normal immunolo- vehicle accidents and in six cases the cause of gical function of the . A recent review trauma was not known. reported that up to 92 g of splenosis tissue was Diagnosis of thoracic splenosis is difficult and insufficient to protect patients from overwhelming requires the presence of a detailed history as well post splenectomy sepsis (OPSS).5 On the other hand, as a high index of suspicion. Once this diagnosis is studies on immunological function of a patient with entertained it is best confirmed with 99mTc scinti- previous traumatic splenectomy and 100 intra- graphy of the chest.4 Both FNA and core biopsy abdominal splenosis identified normal circulating techniques were also frequently employed to estab- antibody levels and response to S. pneumonia anti- lish a diagnosis. FNA is unfortunately often indeter- gens was within normal limits.7 In view of this minate as FNA findings in splenosis resemble those of controversy and potential benefit of splenic nodules lymphoproliferative disorders.13 we advocate removal of only a minimum of auto- When the less aggressive techniques fail, surgery transplanted tissue for diagnosis, rather than exci- must be undertaken to rule out malignancy with sion of all nodules. 286 R.A. Beekman et al.

The natural history of splenosis in any location is 2. Artinian MA, Gilliam JI. CT of intrathoracic splenosis in the fairly benign. A prospective study of 17 patients presence of bronchogenic carcinoma. J Comput Assist Tomogr 99m 1993;17:827—8. using Tc imaging demonstrated the risk of devel- 3. Cordier JF, Camondes JP, Marx P, Heinen I, Loire R. Thoracic oping abdominal and intrathoracic splenosis after splenosis presenting with hemoptysis. Chest 1992;102:626— combined splenic and diaphragmatic injury as 65 7. and 27%, respectively.12 There were no reports of 4. Hagman TF, Winer-Muram HT, Meyer CA, Jennings SG. 99m enlargement with compression of adjacent struc- Intrathoracic splenosis. Superiority of Technetium Tc heat damaged RBC imaging. Chest 2001;120:2097—8. tures in the chest causing symptoms. These obser- 5. Hansen K, Singer DB. Asplenic-hyposplenic overwhelming vations lead us to conclude that routine follow up is sepsis: postsplenectomy sepsis revisted. Pediatr Dev Pathol not necessary and patients can be discharged to 2001;4:105—21. primary care physicians and investigations per- 6. Hardin VM, Morgan AME. Thoracic splenosis. Clin Nucl Med formed as required for other conditions and risk 1994;19:438—40. 7. Hathaway JM, Harley RA, Self S, Schiffman G, Virella G. factors. Immunolgical function in post-traumatic splenosis. Clin Despite confirmation of splenosis with nuclear Immun Immunopathol 1995;74:143—50. medicine scanning, concurrent malignancy in other 8. Hietala E-M, Hermunen H, Kostiainen S. Intrathoracic sple- lesions has been reported.2,6,9,11 Investigations nosis, report of a case simulating esophageal leiomyoma. must diagnose all lesions suspected of splenosis. Scand J Thor Cardiovasc Surg 1993;27:61—3. 9. Jackson HD, Carney KJ, Knautz MA, Tenholder MF. Left upper When not all lesions can be accounted for, explora- lobe mass and diffuse reticular-nodular infiltrate. Chest tion via VATS or thoracotomy is warranted to rule out 1994;105:1864—5. concurrent malignancy. 10. Koleski FC, Turk TM, Ouwenga M, Herrell SD, Borge MA, Albala DM. Splenosis as a cause of testicular pain: laparoscopic management. J Endourol 1999;13:373—5. 11. Kwan AJ, Drum DE, Ahn C, Tow DE. Intrathoracic splenosis Conclusion mimicking metastatic lung cancer. Clin Nucl Med 1994;19:93—5. Thoracic splenosis remains a rare diagnosis but can be 12. Normand JP, Rioux M, Dumont M, Bouchard G, Letourneau L. expected based on a history of splenic trauma with Thoracic splenosis after blunt trauma: frequency and imaging diaphragmatic injury and single or multiple left sided findings. AJR 1993;161:739—41. 13. Renne G, Coci A, Biraghi T, Schmid C. Fine needle aspiration pleural based nodules. Diagnosis can be confirmed of thoracic splenosis. A case report. Acta Cytol 1999;43:492— with tagged RBC scanning and needle biopsy techni- 4. ques avoiding invasive techniques. When these tech- 14. Rickert CH, Maasjosthusmann U, Probst-Cousin S, August C, niques fail, more aggressive methods are required to Gullotta F.A unique case of cerebral spleen. Am J Surg Pathol distinguish splenosis from malignancy, including VATS 1998;22:894—6. 15. Sarda R, Sproat I, Kurtycz DF, Hafez R. Pulmonary parench- or a thoracotomy. Clinical acumen is required to ymal splenosis. Diagn Cytopathol 2001;24:352—5. differentiate malignancy from intra-thoracic spleno- 16. Shaw AF, Shafi A. Traumatic autoplastic transplantation of sis, particularly when there are coexisting patholo- splenic tissue in man with observations on the late results of gies in high risk patients. Once diagnosed, splenosis splenectomy in six cases. J Pathol Bacteriol 1937;45:215—35. requires neither excision nor frequent follow up. 17. Tzunezuka Y, Sato H. Thoracic splenosis; from a thoraco- scopic viewpoint. Eur J Cardiothorac Surg 1998;13:104—6. 18. Varma DGK. Campeau RJ, Kartchner ZA. Scinitigraphic detec- tion of splenosis causing ureteral compression and hydone- phrosis. AJR 1991;156:406. References 19. Velitchkov NG, Kjossev KT, Losanoff JE, Kavardijkova VA. Subcutaneous splenosis: a clue to diagnosis of thoracic sple- 1. Abeles DB, Bego DG. Occult gastrointestinal bleeding and nosis. JR Coll Surg Edinb 1999;44:66. due to entero-enteric intussusception caused 20. Vento JA, Peng F,Spencer RP,Ramsey WH. Massive and widely by splenosis. Surg Endosc 2003;17:1494. distributed splenosis. Clin Nucl Med 1999;24:845—6.