Case reports Posterior mediastinal paravertebral hydatid cyst presenting as spinal compression A case report
P. Ranganadham,
I. Dinakar, C. Sundaram,
KS. Ratnakar,
and T. Vivekananda.
Introduction
suuunary
Vertebral hydatidosis is rare, occurring in l-2 percent of all cases of Echinococcosis,1-5 and reported to be the commonest (66-70%) cause of paraplegia due to hydatid disease1T2*68.Paravertebral hydatid cysts in spinal muscles, retroperitoneal, costal and mediastinal locations producing paraplegia has been reported scantily in the literature@y9. In the present case, the chest radiograph showed a posterior mediastinal lesion as the possible cause of paraplegia, which was further confirmed by computerised tomography. The successful surgical excision com-
A primary posterior mediastinal hydatic cyst presenting with spinal compression syndrome is rare. We describe the case of a 35year-old lady with a posterior mediastinal hydatid cyst who presented with progressive paraplegia. Excision of cyst was followed by systemic Albendazole therapy. She showed neurological recovery without recurrence till now. The role of Albendazole in preventing recurrence has been stressed and the relevant literature has been reviewed.
bined with medical therapy has probably vented the recurrence so far in the present
Key words: hydatid cyst, posterior num, spinal compression.
mediasti-
precase.
Case report
This 35-year-old lady presented with paresthesias and progressive paraplegia of 2 months duration associated with urinary retention for 10 days. 4 days prior to her hospitalisation she had high grade fever with chills and rigors. She had no respiratory symptoms and comes from a nonendemic area for hydatidosis. On examination we saw a poorly built, illnourished lady, lungs were clear. Nervous system examination revealed complete paraplegia (O/5) with a sensory level at D, segment. The
knee and ankle jerks were exaggerated and the plantar reflexes were both extensor. There was tenderness over the D5 spinous process. Her haematological investigations were normal. Lumbar CSF was acellular with 440 mg protein. Casoni’s test was negative. Chest radiograph (Fig. 1) showed a hemispherical mass in the posterior mediastinum with normal lung fields. Radiographs of the dorsal spine showed flattening of the right pedicle of the D, vertebra with a paravertebral shadow. Abdominal Ultrasonogram was normal. Operation - A D,, laminectomy was per-
From the department of neurosurgery & pathology, the Nizam’s Institute of Medical Sciences, Panjagutta, Hyderabad 500 482(A. P) India. Address for correspondence and reprint requests: P. Ranganadham, assistant professor, Department of Neurosurgery, Nizam’s institute of medical sciences, Panjagutta, Hyderabad 500 482 (A. P). India. Accepted 16.1.89 C&n Neurol Neurosurg 1990. Vol. 92-2
149
Fig. 2. Plain Cl” Scan at D5 level showing the para-spinal swelling (hydatid cyst encroching into the spinal canal through the intervertebral foramen. (The laminae have been removed during the laminectomy which was performed prior to excision of cyst.)
operatively there is no neurological deficit and she is able to attend to her daily activities. Fig. 1. X-ray of chest showing flattening of pedicle of D, vertebra of the right side with paravertebral shadow.
formed in the prone position, the epidural veins were engorged and no intraspinal mass lesion could be made out. Further exploration was abandoned as she developed hypotension due to blood loss. A CT scan was done which showed a large well defined, slightly hyperdense lesion in the paravertebral region extending into the spinal canal through the widened right D, intervertebral foramen (Fig. 2). At re-exploration after nibbling out a part of D, transverse process, there was a large cystic mass in the right paravertebral region extending from the posterior mediastinum and deflecting the spinal cord to the left. A complete excision of the cystic mass containing 15-20 small cysts was carried out. The pleura was found to be thickened and normal lung movements were discernible. Post-operatively there was a progressive improvement in motor power and sensations and she regained normal bladder function. She was given a complete course of Albendazole 200 mg twice daily for 3 weeks combined with physiotherapy. After about 2 months she could walk with support and there was 80 percent improvement in sensations. Histopathology showed laminated, eosinophilic acellular membraneus structures con~~ing the diagnosis of hydatid cysts. At the latest followup, 11 months post 150
Discussion
The literature on vertebral hydatidosis is very scanty and mostly in the form of case reDew in 1928’ classified spinal hyports 1-3*f*6,8*10. datid lesions causing paraplegia into five major groups viz,, primary cyst in spinal cord, intradural cyst, extradural cyst, hydatid disease of the vertebrae and paravertebral lesions, vertebral hydatidosis is the ~mmonest’*,8 (~70%). A hydatid cyst primarily in the mediastinum, retroperitoneal tissues and spinal muscles may erode the vertebra by pressure or may extend or rupture into the extradural tissues through the intervertebral foramena. It may be difficult to differentiate this type of case from the primary extradural hydatid which makes its way out of the canal. However, if the size and position of cysts and the time of onset of neurological symptoms in relation to symptoms and signs pertaining to its extraspinal location are taken into consideration, they can usually be differentiated. Mills8 while analysing 18 cases of paraplegia due to hydatid disease from the British literature quotes one patient who had a large hydatid cyst of the superior mediastinum that eroded the vertebral body. Balasubramaniam et al6 reported one case of hydatid cyst in the paraspinal muscles presenting with paraplegia. Ashok et ~1.” reported a case of dorsal vertebral hydati-
dosis with paraspinal swelling, egg shell calcification and rib expansion. In a series of 27 cases collected from two different neurosurgical centres over a period of 23 years Werner et a1.4did not have a single case of paravertebral cyst. Rakower et al.” reported primary posterior mediastinal echinococcosis with rib and vertebral erosion together with separation of neighbouring eroded ribs without any neurological symptoms and signs. In the present case, the patient presented with progressive paraparesis without any respiratory symptoms and the chest radiograph was very classical of a posterior mediastinal mass without rib erosion or separation of ribs, as reported by Rakower”. The question of whether this is primarily a posterior mediastinal cyst extending to the extradural space or a vertebral extradural cyst extending into the posterior mediastinum can be resolved in the present case, as, 1) the radiographs showed a posterior mediastinal mass with evidence of squashing of the right pedicle of D5 vertebra without destruction of the body which is a feature of primary vertebral disease’-lo, 2) computerised tomography showed only a widened intervertebral foramen and the paravertebral lesion extending into the spinal canal, with normal configuration of vertebral body consistent with the observations of Mills8 and 3) at surgery, the entire cystic mass was in the posterior mediastinum extending into the extradural space eroding the transverse process of D, vertebra and was deflecting the spinal cord to the opposite side. The absence of respiratory symptoms in the present case can be explained by the accommodative capacity of the posterior mediastinum. Relapse of vertebral hydatidosis is a very common feature after a variable interval,1,2*4T5*7,8
the earliest is described after 7 weeks and the maximum number of operations on one patient appeared to be six.6 Our patient received a complete course of Albendazole, an analogue of mebendazole, which is expected to prevent relapses. The value of mebendazole in preventing relapse has been stressed by Fiennes et al. ’
We thank Prof. Kakarla Subba Rao, Director, Nixam’s Institute of Medical Sciences for permitting to publish the case report. We thank Mr. K. Satyanarayana for secretarial assistance and Mr. Lava Kumar and Mr. Murali for the photography.
References RAYPORT ~,w~sccwF HS,ZAIMAN H. Vertebralechinococcosis. J Neurosurg 1964; 21647-59. ROBINSON RG. Hydatid disease of the spine and its neurologicaln ~mp~cations. Br J Surg 1960; 47:301-6. VENGSARKAR us, ABRAHAM J. Hydatid disease of the spine. J Post Med 1965; 11:133-6.
WBRNERLAFT,JAUNL.PIERRO,CIROCALDERON,CARLES PEREZ, PATRICIO MWICA. Vertebral hydatid disease. Clinical experience with 27 cases. J Neurosurg 1976; 44112-6. YADAV ss, NAGABHUSHWA KR. Hydatid disease of the spine, with paraplegia (Report of a case). Ind J Med Scie 1983; 37:2,30-2.
BALASUBRAhtANlAMV,RAMANUJAMB,
RAMAhGJRTHIB.
Hydatid disease of the nervous system. Neurology India 1979; 1892-5. FIENNES AGTW, THOMAS DGT. Combined medical and surgical treatment of spinal hydatid disease - a case report. J NeuroI Neurosurg Psychiatry 1982; 45927-31. MILLS n. Paraplegia due to hydatid disease. J Bone Joint Surg 1956; 3831,4:884-91. SAK-SOUK FA. Extrapleural extraosseous costal echinococcosis. Br J Rad 1979; 52:918-9. ASHOK PP,RADHAKRIsHNAN K,SRIDHARAN R. SpiIId hydatid disease. A case report. Neurology India 1987; 35:241-3. RAKOWER I,M~LWIDSKY H. Primary Mediastinal echinococcosis. Am J Med 1960; 29:73-83.
151
P. Ranganadham,
I. Dinakar, C. Sundaram,
KS. Ratnakar,
and T. Vivekananda.
Introduction
suuunary
Vertebral hydatidosis is rare, occurring in l-2 percent of all cases of Echinococcosis,1-5 and reported to be the commonest (66-70%) cause of paraplegia due to hydatid disease1T2*68.Paravertebral hydatid cysts in spinal muscles, retroperitoneal, costal and mediastinal locations producing paraplegia has been reported scantily in the literature@y9. In the present case, the chest radiograph showed a posterior mediastinal lesion as the possible cause of paraplegia, which was further confirmed by computerised tomography. The successful surgical excision com-
A primary posterior mediastinal hydatic cyst presenting with spinal compression syndrome is rare. We describe the case of a 35year-old lady with a posterior mediastinal hydatid cyst who presented with progressive paraplegia. Excision of cyst was followed by systemic Albendazole therapy. She showed neurological recovery without recurrence till now. The role of Albendazole in preventing recurrence has been stressed and the relevant literature has been reviewed.
bined with medical therapy has probably vented the recurrence so far in the present
Key words: hydatid cyst, posterior num, spinal compression.
mediasti-
precase.
Case report
This 35-year-old lady presented with paresthesias and progressive paraplegia of 2 months duration associated with urinary retention for 10 days. 4 days prior to her hospitalisation she had high grade fever with chills and rigors. She had no respiratory symptoms and comes from a nonendemic area for hydatidosis. On examination we saw a poorly built, illnourished lady, lungs were clear. Nervous system examination revealed complete paraplegia (O/5) with a sensory level at D, segment. The
knee and ankle jerks were exaggerated and the plantar reflexes were both extensor. There was tenderness over the D5 spinous process. Her haematological investigations were normal. Lumbar CSF was acellular with 440 mg protein. Casoni’s test was negative. Chest radiograph (Fig. 1) showed a hemispherical mass in the posterior mediastinum with normal lung fields. Radiographs of the dorsal spine showed flattening of the right pedicle of the D, vertebra with a paravertebral shadow. Abdominal Ultrasonogram was normal. Operation - A D,, laminectomy was per-
From the department of neurosurgery & pathology, the Nizam’s Institute of Medical Sciences, Panjagutta, Hyderabad 500 482(A. P) India. Address for correspondence and reprint requests: P. Ranganadham, assistant professor, Department of Neurosurgery, Nizam’s institute of medical sciences, Panjagutta, Hyderabad 500 482 (A. P). India. Accepted 16.1.89 C&n Neurol Neurosurg 1990. Vol. 92-2
149
Fig. 2. Plain Cl” Scan at D5 level showing the para-spinal swelling (hydatid cyst encroching into the spinal canal through the intervertebral foramen. (The laminae have been removed during the laminectomy which was performed prior to excision of cyst.)
operatively there is no neurological deficit and she is able to attend to her daily activities. Fig. 1. X-ray of chest showing flattening of pedicle of D, vertebra of the right side with paravertebral shadow.
formed in the prone position, the epidural veins were engorged and no intraspinal mass lesion could be made out. Further exploration was abandoned as she developed hypotension due to blood loss. A CT scan was done which showed a large well defined, slightly hyperdense lesion in the paravertebral region extending into the spinal canal through the widened right D, intervertebral foramen (Fig. 2). At re-exploration after nibbling out a part of D, transverse process, there was a large cystic mass in the right paravertebral region extending from the posterior mediastinum and deflecting the spinal cord to the left. A complete excision of the cystic mass containing 15-20 small cysts was carried out. The pleura was found to be thickened and normal lung movements were discernible. Post-operatively there was a progressive improvement in motor power and sensations and she regained normal bladder function. She was given a complete course of Albendazole 200 mg twice daily for 3 weeks combined with physiotherapy. After about 2 months she could walk with support and there was 80 percent improvement in sensations. Histopathology showed laminated, eosinophilic acellular membraneus structures con~~ing the diagnosis of hydatid cysts. At the latest followup, 11 months post 150
Discussion
The literature on vertebral hydatidosis is very scanty and mostly in the form of case reDew in 1928’ classified spinal hyports 1-3*f*6,8*10. datid lesions causing paraplegia into five major groups viz,, primary cyst in spinal cord, intradural cyst, extradural cyst, hydatid disease of the vertebrae and paravertebral lesions, vertebral hydatidosis is the ~mmonest’*,8 (~70%). A hydatid cyst primarily in the mediastinum, retroperitoneal tissues and spinal muscles may erode the vertebra by pressure or may extend or rupture into the extradural tissues through the intervertebral foramena. It may be difficult to differentiate this type of case from the primary extradural hydatid which makes its way out of the canal. However, if the size and position of cysts and the time of onset of neurological symptoms in relation to symptoms and signs pertaining to its extraspinal location are taken into consideration, they can usually be differentiated. Mills8 while analysing 18 cases of paraplegia due to hydatid disease from the British literature quotes one patient who had a large hydatid cyst of the superior mediastinum that eroded the vertebral body. Balasubramaniam et al6 reported one case of hydatid cyst in the paraspinal muscles presenting with paraplegia. Ashok et ~1.” reported a case of dorsal vertebral hydati-
dosis with paraspinal swelling, egg shell calcification and rib expansion. In a series of 27 cases collected from two different neurosurgical centres over a period of 23 years Werner et a1.4did not have a single case of paravertebral cyst. Rakower et al.” reported primary posterior mediastinal echinococcosis with rib and vertebral erosion together with separation of neighbouring eroded ribs without any neurological symptoms and signs. In the present case, the patient presented with progressive paraparesis without any respiratory symptoms and the chest radiograph was very classical of a posterior mediastinal mass without rib erosion or separation of ribs, as reported by Rakower”. The question of whether this is primarily a posterior mediastinal cyst extending to the extradural space or a vertebral extradural cyst extending into the posterior mediastinum can be resolved in the present case, as, 1) the radiographs showed a posterior mediastinal mass with evidence of squashing of the right pedicle of D5 vertebra without destruction of the body which is a feature of primary vertebral disease’-lo, 2) computerised tomography showed only a widened intervertebral foramen and the paravertebral lesion extending into the spinal canal, with normal configuration of vertebral body consistent with the observations of Mills8 and 3) at surgery, the entire cystic mass was in the posterior mediastinum extending into the extradural space eroding the transverse process of D, vertebra and was deflecting the spinal cord to the opposite side. The absence of respiratory symptoms in the present case can be explained by the accommodative capacity of the posterior mediastinum. Relapse of vertebral hydatidosis is a very common feature after a variable interval,1,2*4T5*7,8
the earliest is described after 7 weeks and the maximum number of operations on one patient appeared to be six.6 Our patient received a complete course of Albendazole, an analogue of mebendazole, which is expected to prevent relapses. The value of mebendazole in preventing relapse has been stressed by Fiennes et al. ’
We thank Prof. Kakarla Subba Rao, Director, Nixam’s Institute of Medical Sciences for permitting to publish the case report. We thank Mr. K. Satyanarayana for secretarial assistance and Mr. Lava Kumar and Mr. Murali for the photography.
References RAYPORT ~,w~sccwF HS,ZAIMAN H. Vertebralechinococcosis. J Neurosurg 1964; 21647-59. ROBINSON RG. Hydatid disease of the spine and its neurologicaln ~mp~cations. Br J Surg 1960; 47:301-6. VENGSARKAR us, ABRAHAM J. Hydatid disease of the spine. J Post Med 1965; 11:133-6.
WBRNERLAFT,JAUNL.PIERRO,CIROCALDERON,CARLES PEREZ, PATRICIO MWICA. Vertebral hydatid disease. Clinical experience with 27 cases. J Neurosurg 1976; 44112-6. YADAV ss, NAGABHUSHWA KR. Hydatid disease of the spine, with paraplegia (Report of a case). Ind J Med Scie 1983; 37:2,30-2.
BALASUBRAhtANlAMV,RAMANUJAMB,
RAMAhGJRTHIB.
Hydatid disease of the nervous system. Neurology India 1979; 1892-5. FIENNES AGTW, THOMAS DGT. Combined medical and surgical treatment of spinal hydatid disease - a case report. J NeuroI Neurosurg Psychiatry 1982; 45927-31. MILLS n. Paraplegia due to hydatid disease. J Bone Joint Surg 1956; 3831,4:884-91. SAK-SOUK FA. Extrapleural extraosseous costal echinococcosis. Br J Rad 1979; 52:918-9. ASHOK PP,RADHAKRIsHNAN K,SRIDHARAN R. SpiIId hydatid disease. A case report. Neurology India 1987; 35:241-3. RAKOWER I,M~LWIDSKY H. Primary Mediastinal echinococcosis. Am J Med 1960; 29:73-83.
151
