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Intrathoracic splenosis presenting as persistent chest pain
© Fukuhara et al.; licensee BioMed Central Ltd. 2012
Received: 14 May 2012
Accepted: 3 September 2012
Published: 7 September 2012
Thoracic splenosis is a rare entity resulting from splenic and diaphragmatic injury. Patients remain asymptomatic, and surgical intervention is not indicated in the majority of cases. We report a case of a 50-year-old male with a history of splenectomy due to a gunshot wound 30 years previously who presented with vague, progressively worsening chest pain. He was found to have a large intrathoracic splenosis. Unique features of our patient include the presence of symptoms, the significant interval growth of the splenic tissue, and the unprecedented size of the mass, which we believe to be the largest among those previously described.
Thoracic splenosis is a rare, benign condition involving autotransplantation of splenic tissue into the pleural cavity via trauma or surgery. It is usually incidentally detected, asymptomatic, and therapy is not indicated. Due to the resemblance to malignancy on diagnostic imaging studies, the majority of patients have undergone extensive workups or invasive procedures in the past . It has been suggested that removal of thoracic splenic tissue in patients without functional abdominal splenic tissue may render the patient asplenic, increasing the risk of infection , although this concept is still controversial. We herein describe a case of symptomatic intrathoracic splenosis in a 50-year-old man. To our knowledge, thoracic splenosis has not been associated with symptoms with the exception of three previously reported cases that involved pleuritic chest pain and recurrent hemoptysis [3–5].
Discussion and conclusion
Thoracic splenosis, first reported by Shaw and Shafi in 1937 , describes the autotransplantation of splenic tissue into the pleural cavity after splenectomy for traumatic or iatrogenic injury, resulting in multiple nodular implants on the left pleura. Autotransplanted spleens differ from accessory spleens by blood supply, local perforator arteries versus splenic artery respectively . Thoracic splenosis occurs less frequently than does abdominal splenosis and may be found in 18% of patients after splenic rupture. However, its frequency is likely underestimated because most splenic implants are asymptomatic and are only incidentally discovered during chest X-ray or CT. The average interval between initial trauma and diagnosis of thoracic splenosis is 18.8 years . The splenic implants are sessile or pedunculated reddish blue nodules ranging from a few millimeters to 7 cm in diameter . To our knowledge, the size of the present case is the largest among those in previously reported intrathoracic splenosis. The current diagnostic modality of choice for splenosis is noninvasive nuclear scintigraphy. 99mTc sulfur colloid scintigraphy was first used to diagnose splenosis based on the ability of the radiolabeled colloid to localize in the reticuloendothelial system. However, scintigraphy using 99mTc heat-damaged erythrocytes or indium 111-labeled platelets is more sensitive and specific for splenic uptake, making these tests the current diagnostic tools of choice . It is suggested that implanted splenic tissue offers some degree of protection against bacterial infection, lowering the frequency of postsplenectomy sepsis , although the degree of immunoprotection offered by this tissue remains unclear. Many of the human data are in the form of case reports documenting failure of splenic tissue to protect against septicemia , possibly because of the small amount of tissue as well as poor vascularization. A review of the English literature revealed only three symptomatic patients with intrathoracic splenosis: two who presented with hemoptysis and one who presented with pleuritic chest pain [3–5]. We postulate that the pain in our patient was referred pain due to a mass effect and irritation of the pericardium, parietal pleura, and diaphragm mediated by the phrenic nerve. In addition, the mass was obviously growing as shown in the chest X-ray, and this mass effect can explain the recent exacerbation of the chest pain. The natural history of intrathoracic splenosis is essentially benign. However in our case, the autotransplanted tissue increased in size significantly. The diagnosis of thoracic splenosis can be established noninvasively with several diagnostic modalities, and surgical intervention is not indicated unless the patient is symptomatic. In cases of symptomatic thoracic splenosis refractory to non-operative treatment, surgery is beneficial.
Written informed consent was obtained from the patient for publication of this case report. A copy of written consent is available for review by the Editors-in Chief of this journal.
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