post-traumatic paramediastinal lung cysts. Pulmonary abscesses have been observed in patients with the acquired immunodeficiency syndrome. 11 •12 In contrast to the findings in this case, these patients present with prolonged systemic signs and symptoms and do not have a history of recent blunt chest trauma. The clinical course of traumatic pulmonary and paramediastinal cysts is usually benign, requiring only supportive therapy; however, in-hospital observation is recommended because deterioration due to pulmonary contusion and hypoxia may occur. Superinfection occurs rarely, and prophylactic antibiotics are not indicated. 1 If formation of an abscess does occur, specific antibiotic therapy should be instituted. Hemoptysis may persist for several days, but the cavity usually heals over the course of 2 to 16 weeks. Conservative management should be continued ifthe lesion continues to decrease in size at six weeks after injury in adults or three to four months after injury in children. 13 If the cysts become infected or expand, opening and fenestration, transthoracic suction drainage, or lobectomy may be indicated. 3 Several concepts illustrated by this case merit emphasis: (1) It may be difficult to detect the subtle but pertinent abnormalities on initial emergency-room chest roentgenograms. (2) The minor clinical but major radiographic findings must be contrasted with the status of most patients with a ruptured diaphragm, who usually have other injuries and are seriously ill. (3) Absence of bowel sounds upon chest auscultation is an indication for further imaging studies, if the patient's clinical condition permits. Remembering these key points will facilitate appropriate diagnosis and prevent unnecessary surgery. REFERENCES

1 Cocblin DL, Shaw MR. Traumatic lung cysts following minor blunt chest trauma. Clin Radiol1978; 29:151-54 2 Ganske JG, Dennis DL, Vanderveer JB. Traumatic lung cyst: case report and literature review. J Trauma 1981; 21:493-96 3 Stulz P, Schmitt HE, Hasse J, Gradel E. Traumatic pulmonary pseudocysts and paramediastinal air cyst: two rare complications of blunt chest trauma. J Trauma 1984; 24:850-53 4 Elyaderani MK, Gabriele OF. Traumatic paramediastinal air cysts. Br J Radiol1979; 52:458-60 5 Hyde I. Traumatic para-mediastinal air cysts. Br J Radiol1971;

44:380-83 6 Ravin CE, Smith GW, Lester PD, McCloud TC, Putman CE. Post-traumatic pneumatocele in the inferior pulmonary ligament. Radiology 1976; 121:39-41 7 Fagan CJ, Swischuk LE. Traumatic lung and paramediastinal pneumatoceles. Radiology 1976; 120:11-8 8 Williams JR, Bonte FJ. Pulmonary damage in nonpenetrating chest injuries. Radio! Clin N Am 1963; 1:439-48 9 Khan FA, Phillips W. Khan A. Unusual unilateral chest trauma without rib fractures leading to pulmonary laceration requiring pneumonectomy. Chest 1974; 66:211-14 10 Fagan CJ. Traumatic lung cyst. AJR 1966; 97:186-94 11 Burt AD, Scott G, Shiach CR, Isles CG. Acquired immunodeficiency syndrome in a patient with no known risk factors: a pathological study. J Clin Pathol1984;37:471-74 12 Samies JH, Hathaway BN, Echols RM, Veazy JM, Pilon VA. Lung abscess due to Corynebacterium equi. Am J Med 1986;

80:685-88 13 Joynt GH, Jaffe F. Solitary pulmonary hematoma. J Thorac Cardiovasc Surg 1963; 45:741-44 244

Pulmonary Sequestration

Neonate

In the

~~m P1ccfone, Jr., M.D.;* and 'hael E. Burt, M.D., Ph.D., F.C.C.P.t

Pulmonary sequestration is a congenital anomaly resulting in nonfunctiooing lung tissue in either the "estralobar" or "intralobar" position. Both forms derive their blood supply from the systemic circulation. Large pulmonary sequestrations can present in the newborn with poteatially fatal respiratory distress. Surgical resection is the treatment of choice. (Chat 1990; 91:244-46)

P

ulmonary sequestration is a congenital anomaly resulting in a mass of nonfunctioning lung tissue. Sequestrations can occur as "extralobar," with their own pleural investments, or, more commonly, as "intralobar," in which the nonfunctioning mass is surrounded by normal lung tissue. Both forms derive their blood supply from the systemic circulation and are most commonly associated with the lower lobes. 14 Large pulmonary sequestrations can present with potentially fatal respiratory distress in the newborn...a Presented is an extralobar sequestration in a newborn occurring in the right upper thorax, a condition not previously described in the literature to our knowledge. CASE REPORT

A 2,420-g male product of a 30-week gestation was delivered to a 42-ye&l"'!d, gravida 4, para 2, diabetic mother. The pregnancy was remarkable for a prenatal sonogram that revealed polyhydraminose and a cystic mass in the right thorax. No medications were taken during pregnancy. Newborn physical examination results were normal except for low birth weight. The infant did well, and chest roentgenogram (Fig 1 and 2) as well as a repeated sonogram conftrmed a cystic mass in the right upper thorax. Barium swallow demonstrated tracheal and esophageal deviation to the left with no invasion (Fig

3). Exploration was done through a right posterolateral thoracotomy,

which revealed normal-appearing right upper, middle, and lower lobes that were compressed by a large, pink, nonaerating mass that 6lled the upper half of the thorax. The mass had Its own pleural investment separate from the adjacent normal lobes and tented the azygos vein and mediastinal pleura laterally and superiorly. An open biopsy was performed with a frozen section diagnosis of normal, nonaerated lung. Dissection was carried down the right pulmonary artery with identification of all branches to the upper and middle lobes. There were no branches of the pulmonary artery feeding the mass. There was an identifiable pedicle leading to the mass that contained three large arteries of systemic origin. This was divided and the mass removed. The right upper, middle, and lower lobes were allowed to expand, 6lling the thorax in their normal anatomic position. The remainder of the operation and the hospital course were unremarkable. Pathologic examination specimen revealed a 21-g mass measuring 5.5 x 3 x 2.5 em. There was a small area of nonpatent cartilage, 0.2 *Adjunct Attending, Rush-Presbyterian-St. Luke's Medical Center, Chicago. tAssistant ProfessorofSurgery, Cornell University Medical College; Assistant Attending Surgeon, Memorial Sloan-Kettering Cancer Center (case performed), New York. Pulmonary Sequealndlon in the Neonldll (Piccione, Burt)

FIGURE

thorax.

1. Chest roentgenogram demonstrating mass in right upper

em at maximum diameter. The remainder of the specimen consisted of multilobulated cystic tissue composed of lung parenchyma. The histology of the mass with its separate pleura, systemic blood supply and lack of a patent tracheobronchial connection is consistent with the diagnosis extralobar pulmonary sequestration.

derived from the Latin "sequestrare," to separate, and was first applied to this anomaly by Pryce in 1946.• Usually the mass of lung tissue derives its blood supply through one or more anomalous systemic arteries and has its own rudimentary bronchial system, which does not communicate with the normal tracheobronchial tree, therefore lacking any pulmonary function. Pulmonary sequestration occurs in two types: intralobar and extralobar. Intralobar sequestrations represent a mass of nonfunctioning parenchyma occurring within the visceral pleura of the normal functioning lobes. The mass receives arterial supply through anomalous vessels originating from the aorta, and the venous drainage is into the pulmonary veins. The rudimentary bronchial system within the mass typically does not communicate with the tracheobronchial tree. Despite this, the sequestered intralobar tissue can aerate with surrounding lung parenchyma, probably through the pores ofKohn.'- 7 With this intercommunication, repeated infection of the sequestered tissue occurs and accounts for the presenting complaint in many of these patients. In extralobar sequestrations the anomalous lung tissue resides within a distinct visceral pleural investment maintaining complete anatomic and functional separation from adjacent normal lung. The arterial supply is from anomalous systemic vessels; however, the venous drainage is also

DISCUSSION

Pulmonary sequestration is a result of a disturbance in normal embryonic lung development producing a mass of nonfunctioning lung tissue. The term "sequestration" was

FIGURE

2. Lateral roentgenogram demonstrating mass.

FIGURE 3. Barium swallow demonstrating extrinsic compression of the esophagus with deviation to the left.

CHEST I 97 I 1 I JANUARY. 1990

245

systemic, usually via the azygos or hemiazygos systems. Bronchial intercommunications are very rare. Therefore, extralobar sequestration rarely presents with pulmonary infections but rather from mass effects on adjacent normal structures. As with intralobar sequestrations, the left lower lobe is the most frequent site of occurrence. u.s Pulmonary sequestrations are uncommon, with a reported incidence of 1.1 to 1.8 percent of all pulmonary resections. • Intralobar sequestrations are more prevalent than extralobar, possibly due to the greater propensity of the intralobar type to cause symptoms. In Savics review of the literature he found 400 reported cases of intralobar with 133 extralobar and six patients presenting with both types simultaneously. Greater than 90 percent of sequestrations occur on the left, with 97.75 percent of reported cases involving the lower lobes. 8 Although a case of intralobar sequestration has been recently reported in the right upper lobe, there have to date been no reported cases of extralobar sequestrations occurring in the right upper thorax. • The majority of extralobar sequestrations are found during the first year of life as incidental findings on chest roentgenograms. However, infants can present with respiratory distress if the sequestration is large enough to impair ventilation. 4 •10 Diagnosis by chest roentgenogram can be difficult, and ultrasound may help to assess the mass, diaphragm, and relationship of the mass to the mediastinum. Males are affected more than females with approximately 4:1 ratio. In contrast, intralobar sequestrations present later, with few recognized during the first year and some not until early adulthood. Males and females are equally affected. z.• Angiography can be helpful in defining anomalous blood supply, and upper GI contrast studies should be undertaken on all patients with suspected sequestration due to the small but de&nite incidence of coexisting enteric duplications. Hesults of bronchoscopic study are usually normal but may show compressive changes. The treatment of symptomatic or large asymptomatic sequestrations is surgical resection. While segmental resection will be sufficient in some patients with the intralobar type,lobectomy will be necessary in the majority. Extralobar sequestrations with their separate pleural investments can usually be removed sparing normal lung tissue. In all resections, care should be taken to identify and control all anomalous blood vessels, thereby preventing potentially fatal hemorrhage. Pulmonary sequestrations are congenital anomalies that can present life-threatening and respiratory distress in a newborn. The clinician must be aware of this condition and its therapy. REFERENCES

1 Carter R. Pulmonary sequestration. Ann Thorac Surg 1969; 7:68-88 2 Buntain WL, Woolley M, Mahour GH, Isaacs H, Payne V. Pulmonary sequestration in children: a twenty-five-year experience. Surgery 1977; 81:413-20 3 DeParedes CG, Pierce WS, Johnson DG, Waldhausen JA. Pulmonary sequestration in infants and children: a 20-year experience and review of the literature. J Pediatr Surg 1970; 5:136-48 4 Bliek AJ, Mulholland DJ. Extralobar lung sequestration associated with fatal neonatal respiratory distress. Thorax 1971; 26:125-

30 5 Horowitz RN. Extralobar sequestration of lung in newborn infant. Am J Dis Child 1965; 110:195-98 6 Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases. J Pathol 1946; 58:457-68 7 Ryckman FC, Rosenkrantz KG. Thoracic surgical problems in infancy and childhood. Surg Clin North Am 1985; 65:1423-54 8 Savic B, Birtel BS, Tholen W, Funke HD, Knoche R. Lung sequestration: report of seven cases and review of 540 published cases. Thorax 1979; 34:96-101 9 Hoelfel JC. Pulmonary sequestration of the upper lobe in children. Radiology 1986; 160:513-14 10 Werthammer Jw. Hatten HP, Blake WB. Upper thoracic extralobar pulmonary sequestration presenting with respiratory distress in a newborn. Pediatr Radiol1980; 9:11~17

Acute Respiratory Failure In Multiple Sclerosis* Ichiro Kuwahira, M.D.; 7etsuri Kondo, M.D.; YtJBU!P Ohta, M.D., F.C.C.P.; and Hajtme Yamabayaahi, M.D.

A 23-yeiiN)Id woman developed acute respiratory failure in the course of multiple sclerosis. The lack of bulbar dysfunction, the presence of quadriplegia, and the bilateral diaphragmatic weakness indicated the presence of spinal cord lesious involving pyramidal tracts bilaterally. Magnetic resonance imaging revealed a cervical demyelinating lesion between Cl and C3. (Chat 1990; 97:!46-48)

R multiple sclerosiS failure. is a condition rarely associated with (MS). However, in some instances, espira~ory

it may lead rapidly to death.•.. In the literature, the lesions relevant to respiratory failure were only estimated by clinical features or proved by the autopsy &ndings. ,.. We present a case in which the lesion leading to respiratory failure was identi&ed by magnetic resonance imaging (MRI). CASE REPORT

A 23-year-old woman was admitted to the hospital on Nov 20, 1986. Six months earlier, she had suddenly developed visual disturbance. She rapidly improved and was asymptomatic after two months. In September 1986, she experienced visual disturbance again, followed by painful tonic seizure and quadriplegia. Her symptoms progressed, and she developed a respiratory arrest. On admission, the neurologic examination showed clear consciousness, atrophy of the optic nerves, quadriplegia, and hyperreftexia, with positive bilateral Babinski's sign. There were no bulbar or sensory abnormalities. A CSF examination result was normal. Serum antimyeline antibody was positive. A chest x-ray film showed elevation of the diaphragms bilaterally and plate-like atelectasis at the lung bases. The cr findings of the slcull and spine were negative. MRI in the '1'2-weighted spin-echo image demonstrated a lesion with high signal intensity in the cervical *From the Department of Medicine, Tolcai University School of Medicine, Isehara, Kanagawa, Japan. Reprint requests: Dr. Kuwahira, Max-Planck-Institute fur e:tperlmenteUe Medlzin, Hennann-Rein-Strasae 3, Gofflftgen, \lim GermanyD-3400 Acute Respiratory Failure In MS (Kuwehlra et a/)

Pulmonary sequestration in the neonate.

Pulmonary sequestration is a congenital anomaly resulting in nonfunctioning lung tissue in either the "extralobar" or "intralobar" position. Both form...
843KB Sizes 0 Downloads 0 Views