Hypertrophic pulmonary osteoarthropathy (HPOA) associated with primary lung cancer is reported in a fifty-four-year-old man. Symptoms of HPOA were makedly improved following lung resection for adenocarcinoma of the lung

Ayabe Hiroyoshi

Hara Shinsuke

Hisano Hiroshi

Kawahara Katsunobu

Oka Tadayuki

Tagawa Yutaka

Tomita Masaotomita

Tsuji Hiroharu

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Acta Med. Nagasaki 36:89-93Hypertrophic Pulmonary Osteoarthropathy Associated with          Primary Adenocarcinoma of the LungHiroyoshi AYABE, Hiroshi HISANO, Tadayuki OKA Hiroharu TSUJI, Shinsuke HARA, Yutaka TAGAWA   Katsunobu KAWAHARA and Masao TOMITA  The First Department of Surgery, Nagasaki University School of MedicineReceived for Publication, April 3, 1991ABSTRACT : Hypertrophic pulmonary osteoarthropathy (HPOA) associated with primary lung cancer is reported in a fifty-four-year-old man. Symptoms of HPOA were makedly improved following lung resection for adenocarcinoma of the lung.       INTRODUCTION  Hypertrophic pulmonary osteoarthropathy (HPOA) was described independently by Bam berger" and Marie". It is clinically character-ized by clubbing of the fingers and toes, painful swelling of the joints and arthralgia. It is most commonly associated with malignant neo-plasms. Primary lung cancer accounts for 80 per cent of cases, pleural tumors 10 per cent and other intrathoracic tumors 5 per cent". The cause of HPOA remains unknown.  We report a patient with HPOA whose symp-toms were markedly improved following lung resection for primary bronchogenic carcinoma.        CASE REPORT  A fifty-four-year-old man presented a three months history of bilateral knee and ankle pain and an abnormal shadow on a chest roentgeno-gram. He had no respiratory symptoms. He had smoked 20 cigarettes daily for 33 years before admission. On physical examination, the patient appeared well. There were marked clubbing of the toes and fingers (Fig. 1) and periarticular swelling of the foot ankles. Therewere no cervical lymphoadenopathy. Chest was clear.  Radiographs of the chest showed an ill-defined 6 cm mass lesion in the right upper lung field (Fig. 2). Bronchoscopy showed no endo-bronchial lesions, and cytological examination of a bronchial brushing and a sputum specimen gave negative results. Laboratory investigations gave results within normal limits except for mild anemia and elevation of c-reactive protein and erythrocyte sedimentation rate.  Computed tomographic scan of the chest revealed a large mass lesion with spiculation in contact with visceral pleura in the posteri-or segment of the right upper lobe (Fig. 3). Radiographs of the bones of legs showed sub-periosteal bone deposition at the lower ends of the femur, tibia and fibura (Fig. 4). An isotopic bone scan demonstrated pericortical linear concentration of the nuclide along the bilateral femoral and tibial shafts together with icreased uptake in juxta-articular bones. The results of the computed tomographic scan of the brain and liver were negative.  Thoracotomy was performed under the di-agnosis of lung cancer and revealed a large mass lesion in the right upper lobe which invaded to visceral pleura. There were no hilar andFig. 1. Marked clubbing of toesFig. 4. Preoperative roentgenograph of right femur,       tibia and fibula showing subperiosteal new        bone formationFig. 2. Preoperative chast radiograph demonstrat-      ing a large mass shadow in the right upper       lung fieldFig. 5. Histological section of primary tumor       showing undifferentiated adenocarcinoma       (Hematoxylin and eosin stain 20X5)Fig. 3. Preoperative computed tomographic scan       showing irregular round shadowFig. 7. a.        Fig. 6. a. Fig. 6. a. Fig. 6. a) Preoperative bone scintigraphs of legs       showing subperiosteal new bone formation      b)Postoperative bone scan demonstrating       disapperance of bone deposition                  Fig. 7. b. Fig. 7. Photographs of digits. a) preoperative       marked clubbing of fingers b) postoperative       finding one year and four months after        operationmediastinal lymph node enlargement and pleural effusion. Intraoperative needle aspira-tion biopsy confirmed an adenocarcinoma and a right upper lobectomy with mediastinal lymph node dissection was performed.  The lesion was a firm, subpleural irregular tumor of 5.2 X 5.0 X 3.0cm. Histology of the tumor showed a poorly differentiated adenocar-cinoma (Fig. 5). There was no metastasis in hilar and mediastinal lymph nodes (p-T2NoMo, Stage I).  His symptoms of arthralgia of knee and ankle joints improved markedly just after awakening from general anesthesia. His postoperative course was uncomplicated. No adjuvant chem-othrapy or radiotherapy was given. He is well now without disease and complains no arthral-gia two years after the pulmonary resection. A bone X-Ray examined one year after operationrevealed a disappearance of subperiosteal new bone formation and an isotopic bone scan showed normalization of nuclide uptake of femoral and tibial shafts (Fig. 6).  A clubbing of his fingers and toes gradually improved after operation and his extremities are almost in normal apperance (Fig. 7).         DISCUSSION  Hypertrophic pulmonary osteoarthropathy is most commonly associated with primary lung carcinoma. It has also been associated with lung metastasis'), pulmonary infections, achalasia and liver diseases'. HPOA is said to occur in 0.7 per cent to 12 per cent of patients with bronchogenic carcinomas'. The pathophysiolo-gy of HPOA is characterized by a significant increase in peripheral blood flow in the distalhalf of the extremities. However, the cause of this phenomenon is not known. There are several theories explaining this symptom. First, some kind of reflex through vagal nerve is thought to be the cause of HPOA, since vagotomy7' or atropine improved this symptoms. The vagal nerve distributes widely over the visceral pleura and the pulmonary neoplasms are often large and in contact with pleural surface. Second, some authors stressed the endocrine substance as a cause of HPOA be-cause a relativley high incidence of gyneco-mastia and over excretion of growth hormon, estrogen or gonadotropin were frequently ob-served in the patients with HPOA9' 9'. Third, vasodilatic substances such as prostagran-dine or bradykinine excreted form primary pulmonary neoplasms increase the blood flow of the extrimities. In the present case the hormonal assay was not performed and no endocrine disorders were observed. Therefore, the cause of HPOA of this patient was unknown, but some kind of neurogenic reflex may be suggested since his joint pain disappeared soon after lung resection.  In the histologic type of the patients with lung cancer associated with HPOA, squamous cell carcinoma and undifferentiated carcinoma were more frequent than adenocarcinomalo> However, oat cell carcinoma is rare in the pa-tient with HPOA.  The symptoms are characterized by arthral-gia, painful swelling of the limbs and clubbing of the fingers and toes. These symptoms appear early in the course of the disease, but do not relate to the extent of lung cancer"  Radiographic features of HPOA are sub-periosteal new bone formation at the lower ends of the tibia, fibula, radius and ulna. However, these changes are uncommon at the finger, pelvic bone or vertebrum. The isotope scan of bones demonstrated high nuclide accu-mulation at the long bone"- 13). Ali1) reported that these findings of high uptake of isotope were observed also at the skull and mandibulum at a rate of 40-60 per cent. To differentiate findings of high isotopic uptake between bone metastais and HPOA is important in the management of the patient with lung cancer. In the latter, the distribution of high uptake iseven, diffuse and symmetric. On the other hand, in the former, high uptake is irregular, localized and asymmetric and commonly distributed at the vertebrum, rib or pelvic bone. On the clinical stand point, the differential diagnosis between two the pathologies is not difficult.  The symptoms of HPOA such as joint pain are commonly improved after resection of lung neoplasms15, 16) as shown in the present case. However, there were several reports that symptoms decreased after simple thoracotomy, vagotomy, chemotherapy or indomethacin treat-ment. Relief of joint pain was gained without documented tumor response in some reports. This patient is free of disease and symptoms of HPOA two years after lung resection. As the estimation of the cause of HPOA, whether reappearance of symptoms of HPOA are observed or not is interesting when recurrence of lung cancer occurs. Because recurrence in the lung will cause the symptoms of HPOA, metastasis in extrapulmonary sites, symptoms will not appear, if the theory of neurogenic theory is true. If the endocrine theory is correct, symptoms of HPOA may appear regardless of the recurrent sites of cancer.          REFERENCES 1) Bamberger E : A case reprot (German), Wien KIM    Wochenschr 2 : 226, 1889. 2) Marie P : De l'osteoarthropie hypertrophiante    pneumonique, Rev Med 10: 1, 1890. 3) Coury C : Hippocratic fingers and hypertrophic    osteoarthropathy, BrJDis Chest 54 : 202-209,1960. 4) Amin R : Hypertrophic osteoarthropathy in    association with pulmonary metastasis from   carcinoma of bladder, Urology 26: 581-582, 1985. 5) Epstein 0, Ajdukiewicz AB, Dick R, Sherlock    S : Hypertrophic hepatic osteoarthropathy, Am   J Med 67: 88-97, 1979. 6) Lavi Y, Paladugu RR, Benfield JR : Hypertrophic    pulmonary osteoarthropathy in experimental    canine lung cancer, J Thorac Cardiovasc Surg   84: 373-376, 1982. 7) Flavel G : Reversal of pulmonary hypertrophic    osteoarthropathy by vagotomy, Lancetl :260-262,   1956. 8) Ginsberg J, Brown JB : Increased estrogen excre-   tion in hypertrophic pulmonary osteoarthro-   pathy, Lancet 2 : 1274-1276, 1961. 9) Steiner H, Dahlback 0, Waldenstorn J : Ectopic   growth hormone production and osteoarhtro-   pathy in carcinoma of the bronchus, Lancet, 1   783-785, 1968. 10) Yacoub MH : Relation between the histology of    bronchial carcinoma and hypertrophic pulmo-   nary osteoarthropathy, Thorax 20: 537-542, 1965. 11) Honma H : The cause of pulmonary hyper-  trophic osteoarthropathy, Jpn j Clin Med, 33:   392-397, 1975. 12) Greenfield GB: The various roentgen ap-   pearance of pulmonary hypertrophic osteoar-  thropathy, Am J Roent and Nucl Med 101 : 927-  934, 1967. 13) Freeman MH, Tonkin AK: Manifestation of    hypertrophic pulmonary osteoarthropathy in    patients with carcinoma of the lung, Radiology  120: 363-365, 1976.14) All A : Distribution of hypertrophic pulmonary   osteoarthropathy AJR 134: 771-780, 1980. 15) Yamamoto H, Abe 0, Yoneyama T, Ishikawa    S, Honma H : A case of bronchial carcinoma    associated with clubbing of fingers and toes, Jpn   J Thorac Surg 23: 177-183, 1970. 16) Yokota N, Shinohara Y, Fujimoto S, Yamamoto    Y, Tanaka K : A case of pulmonary hypertrophic    osteoarthropathy accompanied with marked    improvement of arthropathy and laboratory    findings after resection of lung cancer, Int Med,   63 : 361-364, 1989.

Hypertrophic Pulmonary Osteoarthropathy Associated with Primary Adenocarcinoma of the Lung

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Hypertrophic Pulmonary Osteoarthropathy Associated with Primary Adenocarcinoma of the Lung

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