Ruptured Intrathoracic Parathyroid Adenoma

Ruptured Intrathoracic Parathyroid Adenoma* Gil H. Santos, M .D ., F .G.G.P.;oo Ghe-Lu Tseng, M.D.;t and Robert W . M. Frater , M .D .:

A case is presented wbere bemorrbage into tbe mediastinum from a ruptured intratboracic parathyroid adenoma mimicked a dissecting aortic bematoma witb acute circulatory coUapse demanding emergency volume replacement.

occurrence of hyperparathyroidism due to an T heintrathoracically located parathyroid gland is very

Papillary muscle embo lus

Incomplete rupture of Posteromedial papillary muscle

FIGURE 2. Diagram showing rupture of necrotic papillary muscle with embolization of fragment of myocardium to coronary artery. LA, Left atrium; and LV, left ventricle . embolus in the present patient obviously represents a complication, rather than a cause, of the acute myocardial infarction. REFERENCES

2

3

4

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Roberts WC, Buja LM: The frequency and significance of coronary arterial thrombi and other observations in fatal acute myocardial infarction: A study of 107 necropsy patients . Am J Med 52 :425-443, 1972 Glotzer OJ, Shaw RS, Scannell JG : Calcific coronary emboli following open valvuloplasty for aortic stenosis. J Thorac Card iovasc Surg 43:435-440 ,1962 Morrow AG, Austen WG : A technique for preventing calcific emboli following aortic valvuloplasty . Surg Gynecol Obstet 114:634-635, 1962 Buchbinder NA, Roberts WC : Left-sided valvular active infective endocarditis : A study of 45 necropsy patients. Am J Med 53 :20-35, 1972 Friedman RM, Roberts WC : Myocardial embolus: A complication of mitral valvulotomy. N Engl J Med 272 :251252, 1965

844 SANTOS, TSENG, FRATER

rare. Parathyroid tumors in the thoracic outlet which are removable by a low cervical incision are also considered as mediastinal tumors by some authorities, who state that the incidence of such tumors can be as high as 21 percent. 1 According to Edwards et al, 2 10 percent of parathyroid tumors are intrathoracic but can be removed through a cervical approach. The true incidence is more accurately reflected by the Mayo series reported by Scholz et al," in which intrathoracic adenomas removed by stern otomy or found at autopsy constituted only 1.4 percent (14) of 1,000 cases. Although incidences as high as 21 percent! and 10 percent- have been reported for intrathoracic parathyroid adenoma, most of these adenomas are, in fact, removable through the cervical approach and should not be considered as mediastinal tumors. Using the previously mentioned criteria, the incidence should, therefore, be in the region of 1 to 2 percent. CASE REPORT

A 47-year-old woman came to the emergency room with sudden onset of retrostemal pain radiating to both arms and to the back between the scapulae . This was accompanied by dizziness and diaphores is. Chest x-ray film demonstrated a wide upper mediastinum (Fig 1) compatible with aortic dissection. The medical history revealed that the patient underwent a vagotomy and pyloroplasty four months previously for pyloric obstruction due to duodenal ulcer. Abnormal biochemical findings at that time included a serum calcium level of 12.3 mg/l00 ml and a serum phosphate level of 2.7 mg/l00 mI. The results of serum parathyroid hormone assay were reported as 994 pg/ml (normal, 500 pg/ml). SkuIl x-ray films revealed a calvarium with a salt-and-pepper appearance. Hyperparathyroidism was suspected, and the neck was explored surgically . Four normal glands were found, and apart from a biopsy from each gland , they were left undisturbed. Postoperatively the patient's serum calcium level remained elevated, but she refused further work-up . She was then discharged to be followed as an outpatient and was doing well until the present adm ission. On physical examination the patient wa~ in acute distress, pale and diaphoretic. The blood °From the Department of Surgery, Albert Einstein College of Medicine, Bronx, NY. • • Instructor in Thoracic Surgery . tchief Resident in Thoracic Surgery . :Professor of Surgery . Reprint requests: Dr . Santos, 1825 Eastchester Road , Bronx

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FIGURE 3. Pathologic section from parathyroid adenoma (H and E, X 150) .

FIGURE 1. Admission chest x-ray film showing widened mediastinum. pressure was 90/70 mm H/I;, and the pulse rate was 110 beats per minute . The pulses in the neck and extremities were all present and equal. The lungs were clear, the heart sounds were not distant , and no murmurs were heard . The abdomen was soft and flat. Laboratory data included the following values: hematocrit reading, 19 percent; hemoglobin, 6.3 gm/ 100 ml, white blood cell count , 7,900/cu mm; platelet count, 210,OOO/cu mm; and prothrombin time, 12.9/11.9 seconds. Chest x-ray films showed marked widening of the mediastinum . The findings on electrocardiogram were within normal limits. With a strong suspicion of dissecting aortic aneurysm, a retrograde transfemoral aortic angiogram was performed, which demonstrated no evidence of abnormality of the thoracic aorta . Hospital Course

During the immediate admission period, the hypovolemia was corrected with 3 units of blood. In order to establish a diagnosis, an anterior mediastinotomy was done through the second right intercostal space. Several hundred cubic centimeters of blood, consisting mainly of clotted blood, was pressing against the chest wall and came out in gushes through the incision as soon as the chest wall was opened. A

FIGURE 2. Parathyroid gland weighing 40

CHEST, 68: 6, DECEMBER, 1975

gin .

mass was identified digitally, and a needle biopsy was done. Examination of frozen sections demonstrated this mass to be compatible with parathyroid carcinoma. A median sternotomy was done, and a large parathyroid gland in the ante rior mediastinum below the innominate vein was removed. The venous return communicated with the innominate vein. The arterial supply emanated from behind the innominate vein, but it was not possible to follow it up to its origin. The gland was very well circumscribed, but it had ruptured, and clots were present in its substance (Fig 2). There was no apparent invasion of surrounding structures. The gland measured 8 X 6 X 4 em and weighed 40 gin after removal of the clots. The final pathologic diagnosis was parathyroid adenoma (Fig 3); and because of its uninvolvement with other structures, it was thought to have been benign . The postoperative course was uneventful. The serum calcium level dropped to 7.9 mg/loo ml but returned to 9 mg/loo ml on the seventh postoperative day. The patient was discharged on the ninth postoperative day and has been doing well since then . DISCUSSION

Berry et al' were the first to report a case of a parathyroid tumor presenting with acute bleeding into the mediastinum, simulating an acute dissecting aneurysm. The tumor was found to be an adenoma located between the right carotid sheath and the esophagus at the thoracic inlet. Capps" in 1934 presented a case where a 22-gm parathyroid gland at the thoracic inlet bled into the mediastinum, the loose connective tissue of the neck, and the subcutaneous tissue, with progressive swelling and purple discoloration of the neck and upper anterior portion of the chest. The case reported by Berry et al 4 also had the unusual feature of being accompanied by symptoms of superior vena cava obstruction. Probably the difference between the clinical presentation of bleeding from a ruptured parathyroid gland depends upon the amount and the rapidity of blood loss. It will also depend upon the location of the gland. The object of this report is, to our knowledge, the only case where hypotension due to massive bleeding had to be counteracted by volume replacement. The gland was inside the mediastinum below the innominate vein, and the bleeding actually occurred inside the mediastinum.

RUPTURED INTRATHORACIC PARATHYROID ADENOMA 845

REFERENCES

2 3 4 5

Nathaniels EK, Nathaniels AM, Wang CA : Mediastinal parathyroid tumors : A clinical and pathological study of 84 cases. Ann Surg 171: 165, 1970 Edwards EA, Malone PD, Collins JJ Jr : Operative Anatomy of the Thorax. Philadelphia, Lea and Febiger, 1972, p82 Scholz DA, Purnell DC, Woolner LB, et al : Mediastinal hyperfunctioning parathyroid tumors : Review of 14 cases. Ann Surg 178 :173-178,1973 Berry BE, Carpenter PC, Fulton RE, et al : Mediastinal hemorrhage from parathyroid adenoma simulating dissecting aneurysm . Arch Surg 108:740-741 , 1974 Capps RB: Multiple parathyroid tumors with massive mediastinal and subcutaneous hemorrhage: A case report. Am J Med Sci 188:800-805, 1934

Lung Tumor in a Patient with Congenital Unilateral Hypoplasia of the Pulmonary Artery· Adalberto Mancebo, M.D., and Adam Wanner, M.D., F.C.C.P.

The occurrence of an undifferentiated carcinoma in the affected lung of a cigarette smoker with asymptomatic congenital hypoplasia of the right pulmonary artery is reported.

membered occasional shortness of breath while doing exercises in school. The patient denied other respiratory symptoms . At age 28 years, she was told that her heart was "pushed to the right." She admitted to smoking one pack of cigarettes a day for the last ten years . A previous chest roentgenogram taken nine months prior to the present admission did not show any lesions. Physical examination revealed a slightly obese, normally developed woman in no acute distre ss. Abnormal physical findings were limited to slight hyperresonance to percussion and distant breath sounds over the right hem ithorax . Chest roentgenograms (conventional posteroanterior and lateral projections) revealed volume loss of the right lung with mediastinal shift to the same side and hyperlucency of the left lung . The pulmonary artery segment was not identified on the right side. The heart was not enlarged and had an unremarkable configuration. An illdefined nodular lesion measuring 1.5 X 1 ern was noted in the anterior segment of the right upper lobe. No hilar lymphadenopathy was detectable ( Fig 1) . Whole-chest tomograms did not show any calcification or cavitation of the lung lesion, nor were satellite lesions or other pulmonary infiltrates seen . A lung scan showed no uptake of lliImtechnetium-labeled macroaggregated albumin in the right lung . In contrast, the left lung had a homogeneous uptake and appeared slightly enlarged. Xenon ventilation scans revealed a delayed washout on the affected side. A pulmonary angiogram confirmed the presence of a markedly hypoplastic right pulmonary artery with a prom inent pulmonary arterial tree and lower-lobe varix on the left (Fig 2) . Bronchoscopy revealed normal tracheobronchial anatomy with diffuse inBammatory changes of the mucosa. No endobronchial lesion was seen . Because of the enlarged bronchial arterial circulation known to be present in patients with

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ep orts of congenital aplasia or hypoplasia of a main branch of the pulmonary artery have appeared in the medical literature since 1868.' Dotter and Steinberg- in 1949 reported a case of hypoplastic pulmonary artery secondary to widespread unilateral lung disease. In 1953, Swyer and James' reported a patient with congenital abnormality of the pulmonary artery and unilateral emphysema. Since then, a number of similar cases have been described, particularly in the British literature.t-" Congenital unilateral hypoplasia of the pulmonary artery was first documented in life by angiography in 1952 by Madoff et al 6 and later was also observed by others.' -" This syndrome has been associated with tetralogy of Fallot, varices of pulmonary veins draining the opposite lung, and unilateral pulmonary edema. " The occurrence of a malignant neoplasm in the hypoplastic lung has, to our knowledge, not yet been reported. The purpose of this report is to present such a case. CASE REPORT

A 49-year-old asymptomatic black woman was admitted to Mount Sinai Medical Center, Miami Beach, Fla, because of an abnormal preemployment chest roentgenogram. She had an uncomplicated birth and developmental history but re°From the Division of Pulmonary Disease, Department of Medicine, Mount Sinai Medical Center, Miami Beach, Fla . Reprint requests: Dr. Wanner, 4300 Alton Road, Miami Beach

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846 MANCEBO, WANNER

FIGURE 1. Chest roentgenogram (posteroanterior projection) showing loss of volume of right lung and displacement of mediastinal structures towards right with overinBation of left lung . Coin lesion in right midlung field is well seen .

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