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J Thorac Cardiovasc Surg 1996;111:1103-1104
© 1996 Mosby, Inc.

BRIEF COMMUNICATIONS

SPLENOTHORACIC FISTULA COMPLICATING PRIMARY SPLENIC HYDATIDOSIS

Madrid, Spain

Received for publication July 10, 1995 Accepted for publication August 7, 1995. Hydatid disease (HD), which is caused by infestation with the parasite Echinococcus granulosus, may occur in almost any organ of the host. The spleen is the third most frequently affected organ (after liver and intrathoracic locations), accounting for 1.7% to 4% of all cases of HD. ¹

Splenic hydatid cysts may rupture intraabdominally. In contrast, intrathoracic rupture is extremely rare. ² We present the case of a patient with primary splenic HD complicated with pleuropulmonary extension through a splenothoracic fistula. To our knowledge, this combination has not been previously reported.

A 46-year-old woman was admitted to the hospital because of pain in the left side of her chest and a mild fever of 1 month's duration. The patient reported a history of contact with dogs. She had been well until 6 months before admission, when she had a bout of left lumbar pain that improved spontaneously. One month before her admission, fever, pain in the left side of her chest, and cough developed. Left lower lobe pneumonia was diagnosed, and the patient took oral erythromycin without clinical improvement.

At admission, results of physical examination were normal except for signs of left pleural effusion. The patient's temperature was 37.6° C. Laboratory data included leukocyte count, 10,400 cells/mm³ (80% neutrophils, 3% eosinophils); sedimentation rate, 92 mm/hr, and hematocrit, 34.7%. A chest roentgenogram disclosed a left lower lobe consolidation and small left pleural effusion. Later radiographs showed progression of the pleural effusion. Thoracentesis showed slight bloody pleural fluid with hydatid membranes and scolices. The Echinococcus indirect hemaglutination titer was 1/2560, and latex fixation was 1/64. Ultrasonographic examination of the abdomen reveled an enlarged spleen with a large multilocular cyst and a unilocular cyst. A computed tomographic scan of the abdomen confirmed the ultrasonographic findings (Fig. 1). A computed tomographic scan of the thorax (Fig. 2) showed a left pleural effusion. At the level of the left lower lobe, a multilocular structure was observed; this suggested a diaphragmatic cyst with lung and/or subdiaphragmatic involvement.

View larger version (118K): [in this window] [in a new window]   Fig. 1. Abdominal computed tomographic scan reveals splenomegaly with a large multilocular hydatid cyst and a unilocular hydatid cyst in the spleen.

View larger version (106K): [in this window] [in a new window]   Fig. 2. Computed tomographic scan of lungs shows a multilocular cyst at the level of left lower portion of the lungs. This lesion may correspond to a diaphragmatic cyst with lung and/or subdiaphragmatic involvement.

Pathologic examination found the pleural fluid and the lung specimen to contain daughter cysts and scolices. Postoperative recovery was uneventful, and the tubes were removed when they no longer drained. Mebendazole (500 mg every 8 hours) was administered for a month. There has been no recurrence for more than a year.

Splenic HD may be primary (more frequent ³) or secondary, related to ruptured intraabdominal cysts. In primary infestation, the parasite has passed through hepatic and pulmonary filters.

Splenothoracic hydatidosis is a rare occurrence, usually occurring as a result of multivisceral intraabdominal hydatidosis. ³ Recently, von Sinner and colleagues ⁴ described a man with secondary intrathoracic hydatidosis. Previously, the patient had had diffuse intraabdominal HD (involving the spleen). Unlike that patient, our patient had solitary splenic hydatidosis and had shown no clinical evidence of disease.

Splenic hydatid cysts may produce serious complications, with rare reports of rupture into the pleura ² and into the bronchial system. ² Because the hydatid fluid is highly antigenic, rupture into the pleural cavity results in chest pain, cough, implantation of the cyst in the cavity, and sometimes anaphylactic shock.

In our case, Echinococcus affected the thorax by extension of splenic HD across the diaphragm. Most likely, the growing cyst had previously eroded the pleura, resulting in the formation of a splenopleural fistula and later parenchymal involvement. There was a pleural effusion as well as multiple pleural hydatidosis caused by the development of the fistula.

Surgery remains the treatment of choice for HD. In associated hydatidosis of the thorax and abdomen, the operation must be carried out with thoracoabdominal access. When there are gross adhesions between the diaphragm and the liver (or spleen), however, it may be wise to primarily address surgical treatment of the chest lesion ⁵ with abdominal cyst evacuation.

Footnotes

From the Departments of Internal Medicine,^a Respiratory Medicine,^b and Family and Communitary Medicine,^c Hospital La Paz, Paseo de la Castellana, Madrid, Spain.

J THORAC CARDIOVASC SURG 1996;111:1103-4

References

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2. Ríos Mozo M, Fernández Sanz S. Hidatidosis esplénica. Rev Clin Esp 1958;60:67-72.
   
3. Muñoz Sánchez JA, Conthe Gutiérrez P, Arnalich Fernández F, et al. Hidatidosis en un hospital general. I. Análisis epidemiológico de 1056 casos. Med Clin 1982;78:421-6.
   
4. Von Sinner WN, Rifai A, te Strake L, Sieck J. Magnetic resonance imaging of thoracic hydatid disease. Acta Radiol 1990;31:59-62.[Medline]
   
5. Borrs S, Shaw J. Hepato-bronchial fistula caused by hydatid disease. Thorax 1981;35:25-8.
   

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