|Year : 2014 | Volume
| Issue : 3 | Page : 157-159
Paraparesis and pleural effusion in a young patient: A road less travelled
Kaushik Ghosh1, Atri Chatterjee2, Sisir Chakraborty3, Rajdip Hazra4, Susmita Ghosh5, Sanat Kumar Jatua6
1 Departments of Medicine, Malda Medical College, Malda, India
2 Department of Neurology, Nilratan Sircar Medical College and Hospital, Kolkata, India
3 Departments of Medicine College of Medicine and Sagore Dutta Hospital, Kolkata, India
4 Departments of Anesthesiology, Nilratan Sircar Medical College and Hospital, Kolkata, India
5 Departments of Anesthesiology, Murshidabad Medical College and Hospital, Berhampore, India
6 Department of Medicine, Murshidabad Medical College and Hospital, Berhampore, West Bengal, India
|Date of Submission||09-Jul-2014|
|Date of Acceptance||02-Aug-2014|
|Date of Web Publication||17-Aug-2014|
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Intrapulmonary teratoma is a rare tumor of the lung, which is scarcely reported to have metastasis at presentation. Here, we report a case of a 14-year-old patient admitted with paraparesis and heaviness of the right side of the chest and was found to have compressive myelopathy along with pleural effusion. He had elevated alpha fetoprotein and beta human chorionic gonadotropin. Imaging revealed a heterogeneous, massive space lesion occupying the right hemithorax and collapse of D12 vertebra with altered marrow signal causing extradural spinal compression. Cytopathology from the tumor revealed immature teratoma cells. The patient was diagnosed as a case of intrapulmonary teratoma with vertebral metastasis. He was started on bleomycin, etoposide, cyclophosphamide chemotherapy and spinal irradiation, which was unsuccessful.
Keywords: Chemoradiation, intrapulmonary teratoma, vertebral metastasis
|How to cite this article:|
Ghosh K, Chatterjee A, Chakraborty S, Hazra R, Ghosh S, Jatua SK. Paraparesis and pleural effusion in a young patient: A road less travelled. Sub-Saharan Afr J Med 2014;1:157-9
|How to cite this URL:|
Ghosh K, Chatterjee A, Chakraborty S, Hazra R, Ghosh S, Jatua SK. Paraparesis and pleural effusion in a young patient: A road less travelled. Sub-Saharan Afr J Med [serial online] 2014 [cited 2020 Nov 26];1:157-9. Available from: https://www.ssajm.org/text.asp?2014/1/3/157/138951
| Introduction|| |
Teratomas are usually benign neoplasms arising from pluripotent stem cells which underwent an abnormal migration during embryonic development.  They are compromised of derivatives of at least two of the three-germ cell layers, namely endoderm, mesoderm and ectoderm.  In adults mature teratomas commonly arise from gonads, mediastinum, retroperitoneum and the sacrococcygeal region. Intrapulmonary teratomas are reported as rare occurrences predominantly affecting young patients without any sex predilection. , Majority of the tumors are benign,  though malignant cases have been reported. Intrapulmonary teratoma with metastasis is extremely rare in literature. Malignant teratomas can metastasize to the brain in approximately 6% patients.  However, vertebral metastasis with resultant neurodeficits has rarely been reported.  Benign intrapulmonary teratomas have been treated with surgery; however, there is no standard protocol for treatment of widely metastatic malignant teratomas. Reports have shown that malignant teratomas with metastasis respond poorly to combined modality treatment with surgery and chemotherapy. 
| Case report|| |
A 14-year-old boy presented to us with the right-sided heaviness of chest for 2 weeks followed by onset of weakness in both lower limbs for 1-day. Heaviness of the right side of the chest slowly progressed for the last 2 months and subsequently worsened rapidly over last 1-day. There was no prior history of hemoptysis or fever. There was no history of tuberculosis. He had received complete immunization according to the national immunization program. The patient's past medical and family history were noncontributory.
On admission, the patient had tachypnea and tachycardia. He had puffiness of face and mild pallor. He had engorged nonpulsatile jugular vein, prominent veins over the right half of chest and fullness of the right intercostal spaces. Breath sound was diminished on the right side below the second intercostal space. The vocal fremitus and percussion note over the area were dull. He also had paraparesis, power in both the lower limbs were grade 3 along with bilateral absent knee and ankle jerks and an absent plantar response. There was tenderness over L4-L5 spinous process. However, he had no sensory or autonomic deficits. Examination of testes was normal so also secondary sexual characteristics. The clinical findings suggested right-sided pleural effusion and superior vena caval syndrome associated with rapid onset compressive myelopathy.
Complete hemogram was normal except presence of normocytic, normochromic anemia. Liver function tests showed an elevated alkaline phosphatase; urea, creatinine, electrolytes, thyroid function tests were normal [Table 1]. The initial chest radiograph revealed a large mass is occupying the middle and lower zones of the right lung with associated pleural effusion.
Consequently, a contrast-enhanced computed tomography (CT) of the thorax with CT guided biopsy was performed. Tomography revealed a complex heterogeneous mass involving right hemithorax with intratumoral calcification, collapse of the ipsilateral lung and mediastinal displacement to the opposite side. The heterogeneity of the lesion was suggestive of lung teratoma [Figure 1]. CT guided aspiration was done from the mass and cytopathology revealed immature teratoma cells. The patient's serum lactate dehydrogenase, beta human chorionic gonadotrophin and alpha fetoprotein were elevated [Table 1]. Considering all these investigations, a diagnosis of pulmonary teratoma, was made. Further investigations were done to evaluate coexistent myelopathy.
Magnetic resonance imaging (MRI) of thoraco-lumbar spine revealed altered marrow signal intensity in D10 and D12 vertebral bodies with mild anterior wedging of D12 vertebra suggestive of secondary deposits [Figure 2]. There was intra spinal extension of the disease process at D9, D10 level with extradural compression of the spinal cord. CT guided aspiration from the site of the collapse was done which revealed cells similar to the lung tumor.
The patient was started on chemotherapy with bleomycin, etoposide and cyclophosphamide along with radiation to spine. The patient did not respond to the regimen of chemoradiotherapy and expired on the 60 th day of hospital stay.
|Figure 1: Computed tomography scan of the thorax showing heterogeneous intralesional attenuation and calcification suggestive of teratoma|
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|Figure 2: Magnetic resonance imaging spine showing altered marrow signals suggestive of metastasis|
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| Discussion|| |
In 1839, Mohr first reported a case of pulmonary teratoma.  Most of the patient presents in their 20-30 years of life with a variety of chest symptoms, pain, hemoptysis, cough or unexplained fever. Coughing out of hair, i.e., trichoptysis is a specific sign of invasive intrapulmonary teratoma.  Bronchiectasis is reported in a number of cases. However, about half of the patient remains asymptomatic or detected incidentally in chest radiography for an unrelated cause. Our patient presented with heaviness of chest followed by paraparesis in early second decade. Pressure effect on surrounding tissue can lead to the development of pressure symptoms such as superior venacaval syndrome. Intrapulmonary teratomas usually range from 2.8 to 3 cm in diameter and are cystic and multiloculated. In our case, it was exceptionally large tumor. Usually benign mature teratomas have largely cystic component whereas solid components predominate in immature or malignant teratomas.
Computed tomography of the chest accurately estimates the density of all elements in the tumor. MRI is important for evaluation of anatomic relation to mediastinal and hilar structures. We used MRI scan as a diagnostic tool for sudden onset compressive myelopathy of this patient. Surgical removal is the treatment of choice wherever possible. Due to the extreme involvement of thorax and metastasis into the vertebrae complete resection was not done. However, a debulking surgery and combination chemo therapy would have been a better option in such advanced cases. We report this case as, to the best of our knowledge, this is the first report of spinal metastasis in a case of intrapulmonary teratoma
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[Figure 1], [Figure 2]