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Double unilateral intrathoracic meningocele
Double unilateral intrathoracic meningocele Report 0/ a case A case of intrathoracic meningocele with the unusual featur es of two cysts or sacs is reported. This is only the fourth case in which there was more than one sac on the sam e side of the thorax.
John H. Blewett, Jr., M.D.,* and John T. Szypulski, M.D .,** Harrisburg, Fa.
Intrathoracic meningocele is still a rare entity. Thus far, only seventy cases have been reported in the literature since Pohl' described the first case in 1933. It is even more uncommon for two intrathoracic meningoceles to occur simultaneously, and it is for this reason an additional case is reported. Case report G. M., a 46-year-old white man , was admitted to the Harrisburg Polyclinic Hospital with a growing mass in the right chest. The pat ient had had a lesion in the right thorax 11 years prior to admission but had remained asymptomatic until the present admission. Physical examination revealed several small neurofibromas of the forehead and back. The cardiorespiratory system was normal. Chest x-ray films disclosed that the homogeneous density which had been present in the upper third of the right lung for 11 years had increased in size during that interval (Figs. 1 and 2) . A preoperative diagnosis of posterior mediastinal tumor was made and a right posterolateral thor acotomy was performed. A cyst measuring 7 em. in diameter was found in the vertebral gutter at the level of the fifth dorsal vertebrae. A small cyst (3 crn.) was found in the sixth intercostal space in the vertebral gutter (Fig. 3) . Both cysts were covered with parietal pleura and were found From the Polyclinic Hospital, Harrisburg, Pa . 17105. Rece ived for publication Oct . 5, 1973. "Resident in Surg ery . ""Chief, Department of Thoracic Surgery.
to communicate with the spinal canal (F ig. 4) . The large cyst was excised and the small One plicated. H istologic examination confirmed the diagnosis of men ingocele . The patient had an uneventful postoperative course and returned to work 2 months after the operation.
Discussion
In reviewing previously reported cases, we found that more than two thirds were associated with neurofibromatosis. Nanson," who reviewed the subject extensively, recognized a syndrome of neurofibromatosis, kyphoscoliosis, and intrathoracic meningocele. Symptoms seem to be lacking. There may be vague discomfort, but most often these cysts do not cause symptoms unless
Fig. 1. Admission chest x-ray film shows a large mass in the right thorax.
481
The Journal of
482
Blewett and Szypulski
Thoracic and Cardiovascular Surgery
Fig. 2. Chest x-ray film taken 11 years prior to admission .
Fig. 3. A 7 ern. cystIs overlying a 3 ern. cyst at the level of the fifth thoracic vertebra.
Fig. 4. Parietal pleura covering cysts which communicate with the spinal canal.
they become massive. A definite etiologic factor for the development of an intrathoracic meningocele remains to be found. A developmental weakness, either congenital or acquired, of the bone or dura has been suggested. Meningoceles may present at any age but are most common in the 30 to 50 year age
range in both sexes. Miles and colleaguesnoted that 52 per cent have occurred on the right side and 40 per cent on the left side. In only 8 previous cases have there been more than one meningocele, and in only 3 were the lesions on the same side. Thus our patient had an extremely unusual type of intrathoracic meningocele.
Volume 67 Number 3
Double unilateral intrathoracic meningocele
483
Morch, 1974
Diagnosis of this entity should be suspected if an intrathoracic mass is found in the posterior mediastinum and is accompanied by the above-mentioned threefold syndrome. Air or oil myelography may be used to verify the diagnosis. Surgery is the last resort for a diagnosis and is indicated for symptomatic or enlarging meningoceles. Treatment consists of open thoracotomy and excision with care taken to suture the pedicle securely to prevent the escape of spinal fluid postoperatively.
3 4 5 6 7
REFERENCES Pohl, R.: Meningokele in Brustraum unter dem Bilde eines intrathorakalin Rundschattens, Rontgenprafis 5: 747, 1933. 2 Nanson, E. M.: Thoracic Meningocele Associ-
8
ated With Neurofibromatosis, J. THORAC. SURG. 33: 650, 1957. Miles, J., Pennybacker, J., and Sheldon, P.: Intrathoracic Meningocele, J. Neurol. Neurosurg. Psychiatry 32: 99, 1969. Hilton, H. D., and McCarthy, H. H.: Intrathoracic Meningocele, J. THORAC. SURG. 37: 261, 1959. Baker, J., and Curtis, G. M.: Intrathoracic Meningocele, West. J. Surg. 61: 209, 1953. YaDeau, R. E., Clagett, O. T., and Divertie, M. B.: Intrathoracic Meningocele, J. THORAC. CARDIOVASC. SURG. 49: 202, 1965. Welch, C. S., Ettinger, A., and Hecht, P. L.: Recklinghausen's Neurofibromatosis Associated With Intrathoracic Meningocele: Report of a Case, N. Engl. J. Med. 238: 622, 1948. Winemann, R., Hallman, G., Babar, D., and Greenberg, S. D.: Intrathoracic Meningocele: Case Report and Review of the Literature, J. THORAC. CARDIOVASC. SURG. 46: 40, 1963.
John H. Blewett, Jr., M.D.,* and John T. Szypulski, M.D .,** Harrisburg, Fa.
Intrathoracic meningocele is still a rare entity. Thus far, only seventy cases have been reported in the literature since Pohl' described the first case in 1933. It is even more uncommon for two intrathoracic meningoceles to occur simultaneously, and it is for this reason an additional case is reported. Case report G. M., a 46-year-old white man , was admitted to the Harrisburg Polyclinic Hospital with a growing mass in the right chest. The pat ient had had a lesion in the right thorax 11 years prior to admission but had remained asymptomatic until the present admission. Physical examination revealed several small neurofibromas of the forehead and back. The cardiorespiratory system was normal. Chest x-ray films disclosed that the homogeneous density which had been present in the upper third of the right lung for 11 years had increased in size during that interval (Figs. 1 and 2) . A preoperative diagnosis of posterior mediastinal tumor was made and a right posterolateral thor acotomy was performed. A cyst measuring 7 em. in diameter was found in the vertebral gutter at the level of the fifth dorsal vertebrae. A small cyst (3 crn.) was found in the sixth intercostal space in the vertebral gutter (Fig. 3) . Both cysts were covered with parietal pleura and were found From the Polyclinic Hospital, Harrisburg, Pa . 17105. Rece ived for publication Oct . 5, 1973. "Resident in Surg ery . ""Chief, Department of Thoracic Surgery.
to communicate with the spinal canal (F ig. 4) . The large cyst was excised and the small One plicated. H istologic examination confirmed the diagnosis of men ingocele . The patient had an uneventful postoperative course and returned to work 2 months after the operation.
Discussion
In reviewing previously reported cases, we found that more than two thirds were associated with neurofibromatosis. Nanson," who reviewed the subject extensively, recognized a syndrome of neurofibromatosis, kyphoscoliosis, and intrathoracic meningocele. Symptoms seem to be lacking. There may be vague discomfort, but most often these cysts do not cause symptoms unless
Fig. 1. Admission chest x-ray film shows a large mass in the right thorax.
481
The Journal of
482
Blewett and Szypulski
Thoracic and Cardiovascular Surgery
Fig. 2. Chest x-ray film taken 11 years prior to admission .
Fig. 3. A 7 ern. cystIs overlying a 3 ern. cyst at the level of the fifth thoracic vertebra.
Fig. 4. Parietal pleura covering cysts which communicate with the spinal canal.
they become massive. A definite etiologic factor for the development of an intrathoracic meningocele remains to be found. A developmental weakness, either congenital or acquired, of the bone or dura has been suggested. Meningoceles may present at any age but are most common in the 30 to 50 year age
range in both sexes. Miles and colleaguesnoted that 52 per cent have occurred on the right side and 40 per cent on the left side. In only 8 previous cases have there been more than one meningocele, and in only 3 were the lesions on the same side. Thus our patient had an extremely unusual type of intrathoracic meningocele.
Volume 67 Number 3
Double unilateral intrathoracic meningocele
483
Morch, 1974
Diagnosis of this entity should be suspected if an intrathoracic mass is found in the posterior mediastinum and is accompanied by the above-mentioned threefold syndrome. Air or oil myelography may be used to verify the diagnosis. Surgery is the last resort for a diagnosis and is indicated for symptomatic or enlarging meningoceles. Treatment consists of open thoracotomy and excision with care taken to suture the pedicle securely to prevent the escape of spinal fluid postoperatively.
3 4 5 6 7
REFERENCES Pohl, R.: Meningokele in Brustraum unter dem Bilde eines intrathorakalin Rundschattens, Rontgenprafis 5: 747, 1933. 2 Nanson, E. M.: Thoracic Meningocele Associ-
8
ated With Neurofibromatosis, J. THORAC. SURG. 33: 650, 1957. Miles, J., Pennybacker, J., and Sheldon, P.: Intrathoracic Meningocele, J. Neurol. Neurosurg. Psychiatry 32: 99, 1969. Hilton, H. D., and McCarthy, H. H.: Intrathoracic Meningocele, J. THORAC. SURG. 37: 261, 1959. Baker, J., and Curtis, G. M.: Intrathoracic Meningocele, West. J. Surg. 61: 209, 1953. YaDeau, R. E., Clagett, O. T., and Divertie, M. B.: Intrathoracic Meningocele, J. THORAC. CARDIOVASC. SURG. 49: 202, 1965. Welch, C. S., Ettinger, A., and Hecht, P. L.: Recklinghausen's Neurofibromatosis Associated With Intrathoracic Meningocele: Report of a Case, N. Engl. J. Med. 238: 622, 1948. Winemann, R., Hallman, G., Babar, D., and Greenberg, S. D.: Intrathoracic Meningocele: Case Report and Review of the Literature, J. THORAC. CARDIOVASC. SURG. 46: 40, 1963.