Ann Thorac Surg 2003;76:1744-1746
© 2003 The Society of Thoracic Surgeons
Case report
Hypertrophic osteoarthropathy associated with esophageal cancer
Masaru Morita, MD, PhDa*,
Yoshihisa Sakaguchi, MD, PhDb,
Sosei Kuma, MDb,
Kiyoshi Kajiyama, MD, PhDb,
Kenji Sugio, MD, PhDa,
Kosei Yasumoto, MD, PhDa
a Second Department of Surgery, School of Medicine, University of Occupational and Environmental Health, Kitakyusyu, Japan
b Department of Surgery, Fukuoka-Higashi National Hospital, Koga City, Fukuoka, Japan
Accepted for publication April 3, 2003.
* Address reprint requests to Dr Morita, Second Department of Surgery, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyusyu 807-8555, Japan
e-mail: m-morita{at}med.uoeh-u.ac.jp
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Abstract
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Hypertrophic osteoarthropathy is extremely rare in patients with esophageal cancer. We herein describe a 65-year-old Japanese man whose esophageal cancer was diagnosed while examining his symptoms of hypertrophic osteoarthropathy. Esophagectomy and postoperative chemoradiation improved the typical radiologic findings of hypertrophic osteoarthropathy as well as the clinical symptoms. Furthermore, the patient has demonstrated a disease-free survival of more than 3 years.
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Introduction
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Hypertrophic osteoarthropathy is a relatively common condition in patients with primary malignant tumors in the lung and pleura [1, 2]. However it is extremely rare in patients with esophageal cancer and successful treatment has never yet been reported in patients suffering from hypertrophic osteoarthropathy with esophageal cancer. We herein describe a patient for whom esophagectomy and postoperative chemoradiation resulted in a long-term survival.
A 65-year-old Japanese man was admitted to Fukuoka-Higashi National Hospital on April 16, 1999. Collagen disease was suspected on admission as he was suffering from fever and severe pain in both his knee and ankle joints. The patient was confined to bed almost all day owing to these symptoms. On computed tomography, which was performed to investigate the cause of the unknown fever, a thickening of the esophageal wall was visualized. We therefore considered that the patient might have esophageal cancer even though he had never felt any dysphasia or retrosternal pain.
The patient had a fever of more than 38°C. Physical examination showed clubbing of the fingers and swelling as well as tenderness in the bilateral knee and ankle joints. The white blood cell count was 5,300 and the C-reactive protein level was increased to 7.43 mg/dL. Liver and renal function was normal. Neither rheumatoid factor nor antinuclear antibody was detectable. The serum hormone levels, such as growth hormone, parathyroid hormone, calcitonin, estradiol, estriol, and follicle-stimulating hormone were all within the normal range.
An esophagogram revealed a huge pedunculated tumor with ulceration in the lower esophagus (Fig 1)
and poorly differentiated squamous cell carcinoma was histologically diagnosed based on an analysis of biopsy specimens. A skeletal survey revealed a thick unilaminar periosteal reaction especially in the distal part of bilateral tibias and fibulas. Bone scintigraphy showed bilaterally symmetrical and diffuse accumulation of radioisotope (RI) in the distal end of bone of the extremities (Fig 2, A).
These radiologic findings were compatible with hypertrophic osteoarthropathy.
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Fig 1. In this esophagogram, a huge pedunculated tumor with ulceration is visualized in the lower esophagus. The length of the tumor is 9 cm.
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Fig 2. (A) Before treatment, bone scintigraphy shows a bilaterally symmetrical and diffuse accumulation of radioisotope (RI) in the distal end of bone of the extremities. (B) Scintigraphy performed 15 days after surgery already shows an improvement in the accumulation of RI in the bone. (C) Six months after surgery the abnormal accumulation of RI in the bone scintigraphy findings has almost completely disappeared.
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On July 27, 1999, an esophagctomy and reconstruction were performed. The tumor invaded the adventitia; however neither lymph node metastasis nor pulmonary metastasis was recognized. Postoperative radiation (Total 60 Gy) was performed on the mediastinum in combination with chemotherapy consisiting of cisplatin (total 138 mg) and 5-Fluorocil (total 10.35 g).
The patient recovered well from both surgery and the symptoms of hypertrophic osteoarthropathy including fever as well as pain, and the swelling in the lower extremities disappeared within 7 days after operation. The accumulation of RI on bone scintigraphy also improved 15 days after surgery (Fig 2, B). A periosteal reaction observed on plain x-ray film as well as an abnormal accumulation of RI on bone scintigraphy almost completely disappeared by 6 months after surgery (Fig 2, C). Thirty-seven months after surgery the patient is doing well with neither a recurrence of esophageal cancer nor any symptoms of hypertrophic osteoarthropathy.
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Comment
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Peirce and colleagues [3] reviewed 7 cases of esophageal cancer associated with hypertrophic osteoarthropathy. Since then an additional 5 cases were reported [4–8]. In the current case the removal of the esophageal cancer remarkably reversed the hypertrophic osteoarthropathy and neither lung metastasis nor pleural involvement was recognized. These facts strongly support that hypertrophic osteoarthropathy must be caused by primary esophageal cancer. Therefore this case is the 13th case of esophageal cancer associated with hypertrophic osteoarthropathy reported in the literature. This case may be of diagnostic use to surgeons who generally think of lung cancer, not esophageal cancer, as being associated with hypertrophic osteoarthropathy.
In the case of primary nonsmall cell lung cancer clubbing and hypertrophic osteoarthropathy is a presenting symptom in 1% of all patients and on careful review 10% to 20% of all patients have various features of this disorder. Hypertrophic osteoarthropathy is sometimes an early manifestation of an occult disease in a curative stage of such cases [1, 2]. In the case described here the opportunity to detect the primary lesion was not due to the symptoms of esophageal cancer but to those of hypertrophic osteoarthropathy.
Successful treatment of the primary lung cancer by either surgery, radiation therapy, or chemotherapy has been reported to ameliorate symptoms and the response is often a prompt one [2]. Barber and associates [6] described a patient who underwent an esophagectomy for squamous cell carcinoma of the esophagus associated with hypertrophic osteoarthropathy and improvement of the osteoarthropathy occurred rapidly after operation. However the patient died 4 months after an esophagectomy owing to metastases of esophageal cancer. Successful treatment resulting in long-term survival and well-controlled hypertrophic osteoarthropathy has never been reported in cases of esophageal cancer associated with hypertrophic osteoarthropathy as the malignant tumor was too far advanced to cure. In our case the patient was completely free of symptoms of esophageal cancer and the disease was discovered owing to the symptoms of hypertrophic osteoarthropathy even though the primary tumor was huge. Furthermore surgery and postoperative chemoradiation have been proved to be effective not only for the control of the malignant lesions but also for improving the symptoms of hypertrophic osteoarthropathy. This improvement was recognized in the radiologic findings as well as the clinical symptoms shortly after the operation. Furthermore the patient has demonstrated a more than 3-year disease-free interval without any symptoms of hypertrophic osteoarthropathy.
We conclude that esophageal cancer may potentially be one of the underlying diseases of hypertrophic osteoarthropathy even though this combination may be extremely rare and that radical treatment for malignant lesion was found to be effective in the control of hypertrophic osteoarthropathy.
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References
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- Campanella N., Moraca A., Pergolini M., et al. Paraneoplastic syndromes in 68 cases of respectable non-small cell lung carcinoma: can they help in early detection?. Med Oncol 1999;16:129-133.[Medline]
- Burstein H.J., Janicek M.J., Skarin A.T. Hypertrophic osteoarthropathy. J Clin Oncol 1997;15:2759-2760.[Free Full Text]
- Peirce T.H., Weir D.G. Hypertrophic osteoarthropathy associated with a non-metastasising carcinoma of the oesophagus. J Irish Med Assoc 1973;66:160-162.[Medline]
- Carroll K.B., Doyle L. A common factor in hypertrophic osteoarthropathy. Thorax 1974;29:262-264.[Abstract/Free Full Text]
- Rothschild B.M., Sebes J.I. Hypertrophic osteoarthropathy associated with carcinoma of the esophagus. Am J Gastroenterol 1980;73:503-507.[Medline]
- Barber P.V., Lechler R. Hypertrophic osteoarthropathy: two unusual causes. Postgrad Med J 1983;59:254-255.[Abstract/Free Full Text]
- Polkey M.I., Cook G.R., Thomson A.D., Taylor N.F. Clubbing associated with oesophageal adenocarcinoma. Postgrad Med J 1991;67:1015-1017.[Abstract/Free Full Text]
- Bhargava S.K., Pasricha P., Dwivedi S., Shiv V.K., Taneja K., Mehrotra G. Carcinoma oesophagus—an unusual cause of hypertrophic osteoarthropathy. Materia Medica Polona 1992;81:52-54.
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Abstract
Introduction
