Hypertrophic pulmonary osteoarthropathy in pulmonary metastatic disease. A case report and review of the literature

Hypertrophic pulmonary osteoarthropathy in pulmonary metastatic disease. A case report and review of the literature

Clinical Radiology (1991) 43, 268 271 Hypertrophic Pulmonary Osteoarthropathy in Pulmonary Metastatic Disease. A Case Report and Review of the Litera...

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Clinical Radiology (1991) 43, 268 271

Hypertrophic Pulmonary Osteoarthropathy in Pulmonary Metastatic Disease. A Case Report and Review of the Literature R. A. DAVIES, M. DARBY* and M. A. RICHARDS'~

Departments of Radiology, King's College Hospital, London and *St George's Hospital, London and "~Clinical Oncology Unit, Guy's Hospital, London We present a case of a woman with pulmonary metastatic disease from breast carcinoma who presented with features of pulmonary osteoarthropathy (HPOA). We document the unusual presentation with classical features of H P O A in the absence of finger clubbing and the response of the scintigraphic appearances to antitumour chemotherapy. Davies, R_A., Darby, M. & R i c h a r d s , M.A. (1991). Clinical Radiology 43, 268-271. Hypertrophic Pulmonary Osteoarthropathy in Pulmonary Metastatic Disease. A Case Report and Review of the Literature

Pulmonary metastatic disease from extrapulmonary malignancy is a rare cause of hypertrophic osteoarthropathy (HPOA) (Aufses and Aufses, 1960). We describe a case of H P O A due to pulmonary metastasis from carcinoma of the breast and discuss some unusual features manifested by the case. CASE R E P O R T A 26-year-old woman, mother of a child aged 18 months, presented with a lump in the right breast. Biopsy showed this to be a ductal carcinoma and at operation one axillary lymph node was found to be involved. The chest radiograph at this time was clear. One month later, she presented with a dry cough and ankle swelling and a repeat chest radiograph showed multiple pulmonary metastases (Fig. 1). On questioning, the patient admitted to some ankle pain on standing, and on examination there was pitting oedema of the ankles, but no joint tenderness. There was no clubbing of the fingers. A bone scintigram showed the classical appearance of HPOA, affecting the

(a)

Fig. 1 - Initial chest radiograph showing multiple pulmonary metastatic deposits. Correspondence to: Dr R. A. Davies, Prince Charles Hospital, Merthyr Tydfil CF47 9DT, Wales.

(b)

PULMONARY OSTEOARTHROPATHY IN PULMONARY METASTATIC DISEASE

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(c) Fig. 2 (a) - Bone scan showing increasedisotope activity along margins of long bones. Corresponding radiographs of (b) ankle and (c) wrist, showing periosteal new bone (arrows).

Fig. 3 - Chest radiograph showing resolution of pulmonary metastases.

distal ulnae, tibiae and fibulae and to a lesser extent the radii and femora (Fig. 2a). Radiographs of the hands, wrists and ankles confirmed the diagnosis, showing well-applied periosteal reaction on the lateral borders of the ulnae, tibiae and fibulae (Fig. 2b, e). In view of the rarity of metastatic disease, particularly from breast carcinoma, as a cause of HPOA, needle biopsy of the lung was performed to exclude a new primary lesion, or possibly secondary deposits from an unsuspected sarcoma. The cytological appearances were compatible with metastatic breast carcinoma. The patient received a course of chemotherapy, with complete resolution of the pulmonary metastases (Fig. 3) and simultaneous improvement in both the clinical and bone-scan manifestations of HPOA (Fig. 4). The patient remained in remission for 7 months, at which time pulmonary metastases returned, with recrudescenceof the HPOA both clinicallyand radiologically (Fig. 5).

DISCUSSION P u l m o n a r y metastases a c c o u n t for a very small p r o p o r tion o f cases of H P O A , the great majority being due to primary l u n g cancer, particularly s q u a m o u s carcinoma. In contrast, in a review of the literature in 1931,

Fig. 4-Bone scan contemporary with Fig. 3 showing resolution of periosteal activity.

B l u m e n s a a t found, out of 144 cases o f H P O A published at that time, only nine cases of l u n g t u m o u r . O f these only one was a p r i m a r y b r o n c h i a l carcinoma, a n o t h e r a p r i m a r y of u n c e r t a i n histology, a n d the r e m a i n i n g seven were due to metastases from extrathoracic sites, of which one was from breast c a r c i n o m a (Kruger, 1906). The great m a j o r i t y of cases (135) were due to chronic lung and pleural i n f l a m m a t i o n a n d to heart disease, p r e s u m a b l y cyanotic. Since T h o m p s o n (1904) first described a c a s e of H P O A in association with p r i m a r y bronchial malignancy, the incidence of the latter has increased dramatically, a n d with the decline in tuberculous a n d other chronic lung a n d pleura infections, it is n o w the c o m m o n e s t cause of H P O A . The earliest description of H P O A in the context of

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Yacoub et al., 1967; Serre et al., 1968; Aufses and Aufses, 1969; Tarasova et al., 1972; Brower and Teates, 1973; Sethi and Saxton, 1974; Firooznia et al., 1975; Sonoda and Krauss, 1975; Goldstraw and Walbaum, 1976; Kerst, 1977; Lokich, 1977; Howard et al., 1978; Wilson and Naidoo, 1979; Amin, 1985). Our patient was unusual in two respects. Firstly, she exhibited H P O A in the absence of finger clubbing, a rare occurrence, the first case having been recorded by Di Matteo and Godlewski in 1947, and a search of the literature over the last two years revealing only one case (Harris, 1988). Secondly, resolution of the radiological manifestations of HPOA in pulmonary metastatic disease has not previously been described, though reduction in the degree of bone scan abnormality has been shown (Lokich, 1977), and Ali et al. (1980) describe fluctuation in severity with treatment in a case of pulmonary metastatic disease, also from breast carcinoma. The scintigraphic appearance of H P O A is highly characteristic, and is a sensitive indicator of periosteal activity. In our patient, treatment of the underlying pulmonary metastatic disease led to complete resolution of the bone scan abnormality. Acknowledgements: We thank Professor Robert Rubens, Clinical Oncologist, Guy's Hospital for his permission to report this case We also thank Dr Rudi Borgstein for his help with translation from the Dutch. REFERENCES

Fig. 5 Bone scan showing recrudescence of appearances of hypertrophic pulmonary osteoarthropathy following relapse.

pulmonary metastatic disease has been attributed to Virchow (1895). However, in his 1915 review of the subject, Locke casts doubt on the accuracy of the diagnosis, pointing out that the patient described by Virchow also had acromegaly, with its attendant digital changes and tendency to osteoarthritis. Virchow did not have the benefit of Roentgen's discovery, a year later, to document the bony appearances with X-rays. Since 1895, 73 cases have been recorded in the literature; 41 of them were summarized in a review article (Yacoub et al., 1967). Only five other cases due to metastatic breast carcinoma are recorded. There is a roughly equal incidence of sarcomas (33) and carcinomas (30), with melanoma (7) and miscellaneous tumours making up the remainder. Allowing for its overwhelming prevalence, breast carcinoma is remarkably under-represented, with osteosarcoma, a much rarer tumour, featuring more than twice as often (15 cases) (data from Bariety and Coury, 1950;

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