Correspondence

Pancoast's Syndrome

N Engl J Med 1998; 338:765-766March 12, 1998

Article

To the Editor:

As Arcasoy and Jett (Nov. 6 issue)1 suggest in their excellent review of Pancoast's syndrome, patients may present with supraclavicular lymph-node enlargement. The authors did not mention one shortcut to the diagnosis of this type of tumor. Fine-needle aspiration biopsy of enlarged supraclavicular lymph nodes or an ipsilateral supraclavicular fullness is a rapid, safe, and inexpensive means of diagnosis. When supraclavicular adenopathy is present, this approach can eliminate the need for more expensive and more invasive procedures.

David S. Meyers, M.D.
Oregon Medical Laboratories, Eugene, OR 97405

1 References

1. 1

   Arcasoy SM, Jett JR. Superior pulmonary sulcus tumors and Pancoast's syndrome. N Engl J Med 1997;337:1370-1376
   Full Text | Web of Science | Medline

To the Editor:

We report an unusual cause of Pancoast's syndrome in an 11-year-old boy who presented with a two-month history of low-grade fever, malaise, and right-shoulder pain radiating to the arm and forearm. He had anisocoria with right-sided miosis and ptosis, plus an erythrocyte sedimentation rate of 96 mm per hour and mild thrombocytosis.

The chest radiographs showed an opacity at the posterior part of the right-lung apex. Computed tomography (CT) and magnetic resonance imaging showed a heterogeneous mass at the right-lung apex adjacent to the C8 and T1 vertebral bodies, with possible invasion of the brachial plexus but no enlarged mediastinal lymph nodes or lytic lesions in bone. Local application of a 4 percent cocaine solution led to dilation of only the left pupil. Cytologic analysis of sputum and bronchoscopy were not diagnostic. A CT-guided transthoracic needle biopsy revealed a nonspecific inflammatory process.

The patient underwent posterior thoracotomy. A heterogeneous, infiltrating mass with undefined edges that extended to the brachial plexus was found. A partial right upper lobectomy with partial resection of the paravertebral sympathetic chain and the brachial plexus was performed. On gross examination, the tumor was firm and white, with infiltrative borders. On histologic examination, there was a spindle-cell proliferation mixed with a polymorphic inflammatory component, consisting principally of lymphocytes and plasma cells.

Immunohistochemical studies revealed a mixture of B and T lymphocytes and some CD68+ histiocytes. Among B cells, both kappa and lambda plasma cells were found. Immunohistochemically and ultrastructurally, the spindle cells had features consistent with those of myofibroblasts. Flow-cytometric analysis of DNA revealed hyperdiploidy. Postoperatively, pain and systemic symptoms disappeared and Horner's syndrome improved. Three years later, magnetic resonance imaging showed only residual changes at the right upper lobe.

Inflammatory pseudotumor (plasma-cell granuloma) is a nonneoplastic reactive inflammatory response evoked by an unknown agent or agents. The concept of the lesion as benign, however, has recently been disputed on the basis of both the clinical demonstration of recurrence and local infiltration of vital structures and cytogenetic evidence of an acquired clonal chromosomal abnormality.1,2 Also, recent data indicated the presence of aneuploidy in half the cases of inflammatory pseudotumor, which appeared to reflect a more aggressive biologic behavior.2

Although the lung is the most common site, inflammatory pseudotumors occur in other locations.3 Primary pulmonary tumors are rare in children. Of the benign tumors, the most common is the inflammatory pseudotumor.4 Hence, inflammatory pseudotumor should potentially be considered in children with Pancoast's syndrome due to superior pulmonary sulcus tumor.

Xavier Bosch, M.D., Ph.D.
Manel Vera, M.D.
Hospital Casa Maternitat, 08028 Barcelona, Spain

4 References

1. 1

   Snyder CS, Dell'Aquila M, Haghighi P, Baergen RN, Suh YK, Yi ES. Clonal changes in inflammatory pseudotumor of the lung: a case report. Cancer 1995;76:1545-1549
   CrossRef | Web of Science | Medline

2. 2

   Biselli R, Ferlini C, Fattorossi A, Boldrini R, Bosman C. Inflammatory myofibroblastic tumor (inflammatory pseudotumor): DNA flow cytometric analysis of nine pediatric cases. Cancer 1996;77:778-784
   CrossRef | Web of Science | Medline

3. 3

   Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor): a clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 1995;19:859-872
   CrossRef | Web of Science | Medline

4. 4

   Jayne D, Bridgewater B, Lawson RA. Endobronchial inflammatory pseudotumour exacerbating asthma. Postgrad Med J 1997;73:98-99
   CrossRef | Web of Science | Medline

To the Editor:

We would like to propose a further, rare addition to the list of infectious causes of Pancoast's syndrome: mycotic aneurysm. We recently admitted a 33-year-old injection-drug user who presented with right-sided chest, shoulder, and periscapular pain. Twelve days earlier she had injected heroin into her right lateral pectoralis muscle. Two days later she noted swelling of the right arm, shoulder, and neck with periscapular pain, pupil asymmetry, and hemoptysis. She went to another hospital and was found to have a temperature of 39.1°C. Blood cultures subsequently grew Staphylococcus aureus, but the patient had already been discharged from the emergency room after being given cephalexin (Keflex) for presumptive cellulitis and was lost to follow-up.

Defervescence occurred; the swelling and hemoptysis resolved; and the shoulder pain waned. One day before admission to our hospital, she began to have right-sided pleuritic chest pain accompanied by the return of periscapular and shoulder pain. She also reported recent weight loss, night sweats, myalgias, and arthralgias. On physical examination, she had a temperature of 39°C, diaphoresis, anisocoria (with miosis of the right eye), and right-sided ptosis and facial anhidrosis. There was marked tenderness to palpation of the right supraclavicular area and a firm 3-cm mass. The right radial and ulnar pulses were not palpable and were only faintly audible on Doppler ultrasonography. The patient had painful ecchymoses (Janeway lesions) on the tips of her right fourth and fifth fingers but no splinter hemorrhages. Strength and sensitivity to light touch and temperature were decreased in a right-ulnar-nerve distribution.

The patient was treated presumptively for endocarditis. Neither transthoracic nor transesophageal cardiac echocardiograms revealed valvular vegetations. Chest CT revealed an air–fluid collection in the region of the right upper lung, consistent with the presence of an extrapleural abscess, and a vascular pseudoaneurysm. A mycotic aneurysm of the right subclavian artery was confirmed by ultrasonography and transcatheter angiography. The patient underwent arterial ligation and abscess drainage and made an uneventful recovery.

Aneurysms of the subclavian artery are relatively rare. Mycotic aneurysms are even more unusual.1-3 With intravenous drug use increasing, this rare cause of Pancoast's syndrome may become more common.

Jack W. Tsao, M.D., D.Phil.
Amy B. Garlin, B.A.
University of California–San Francisco, San Francisco, CA 94143

Shelley R. Marder, M.D.
San Francisco General Hospital, San Francisco, CA 94110

3 References

1. 1

   Ho KL, Rassekh ZS. Mycotic aneurysm of the right subclavian artery: a complication of heroin addiction. Chest 1978;74:116-117
   CrossRef | Web of Science | Medline

2. 2

   Pairolero PC, Walls JT, Payne WS, Hollier LH, Fairbairn JF II. Subclavian-axillary artery aneurysms. Surgery 1981;90:757-763
   Web of Science | Medline

3. 3

   Miller CM, Sangiuolo P, Schanzer H, Haimov M, McElhinney AJ, Jacobson JH II. Infected false aneurysms of the subclavian artery: a complication in drug addicts. J Vasc Surg 1984;1:684-688
   CrossRef | Web of Science | Medline

Author/Editor Response

The authors reply:

To the Editor: We appreciate the comments of Dr. Meyers and share his view that when supraclavicular lymphadenopathy is present, fine-needle aspiration biopsy is a simple and safe diagnostic technique. As with any disease process, including malignant conditions, the easiest and safest procedure for diagnosis, which may include obtaining biopsy samples from peripheral lymph nodes or skin lesions, should be used when feasible.

Drs. Bosch and Vera and Tsao et al. expand the list of the causes of Pancoast's syndrome with their interesting reports of an inflammatory pseudotumor (plasma-cell granuloma) and a mycotic subclavian-artery aneurysm. Another reported vascular lesion mimicking Pancoast's tumor in a child is a carotid pseudoaneurysm caused by a hydatid cyst.1 These cases further emphasize the need for a definitive clinical and histologic diagnosis of superior sulcus lesions before treatment.

Selim M. Arcasoy, M.D.
University of Pittsburgh, Pittsburgh, PA 15261

James R. Jett, M.D.
Mayo Clinic, Rochester, MN 55905

1 References

1. 1

   Rong SH. Carotid pseudoaneurysm simulating Pancoast tumor. AJR Am J Roentgenol 1984;142:495-496
   Web of Science | Medline

Citing Articles (1)

Citing Articles

1. 1

   A.D. Lee, G.K. Kota, N. Shyamkumar, D.T. Abraham, S. Agarwal. (2005) Endovascular Management of an Unusual Cause of Pancoast Syndrome. European Journal of Vascular and Endovascular Surgery 30:6, 621-623
   CrossRef