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Ann Thorac Surg 2007;83:684-685
© 2007 The Society of Thoracic Surgeons

Case Reports

Effective Symptomatic Relief of Hypertrophic Pulmonary Osteoarthropathy by Video-Assisted Thoracic Surgery Truncal Vagotomy

Adrian Ooi, MRCS, Rasheed A. Saad, MRCS, Narain Moorjani, MRCS, Khalid M. Amer, FRCS(CTS)*

Wessex Cardiothoracic Centre, Southampton University Hospital, Southampton, United Kingdom

Accepted for publication June 1, 2006.

* Address correspondence to Mr Amer, Wessex Cardiothoracic Centre, Southampton University Hospital, Tremona Road, Southampton, United Kingdom SO16 6YD. (Email: khalid.amer{at}suht.swest.nhs.uk).


   Abstract
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 Abstract
Introduction
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Various modalities for the treatment of hypertrophic pulmonary osteoarthropathy (HPOA) associated with lung cancer have been suggested since 1958. Although the etiology remains speculative, unilateral vagotomy on the side of the lung cancer achieves symptomatic relief. We report a case of a 50-year-old woman with disabling HPOA and inoperable lung cancer who experienced effective pain relief and regained full mobility after video-assisted thoracoscopic surgery was used to perform truncal vagotomy. This relatively safe and simple procedure should be considered for terminal lung cancer patients with intractable HPOA.


   Introduction
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Hypertrophic pulmonary osteoarthropathy (HPOA) is a syndrome characterized by digital clubbing and periostosis of the tubular bones [1] and manifested by swollen, red, warm, and painful joints. The syndrome is most commonly seen in patients with primary lung and pleural tumors. The only effective treatment for HPOA is treatment of the underlying condition by lung tumor resection. Historically, surgical vagotomy had been effective in providing symptomatic relief when the lung tumor proved unresectable. We report the use of a video-assisted thoracic surgery (VATS) approach as the first choice for vagotomy in this cohort of patients.

A 50-year-old woman presented with dry cough and severe clubbing of the fingers and toes on both hands and feet (Fig 1). She had increasing pain in the wrists, knees, and ankles for 4 months that compromised her mobility and daily activities and left her confined to a wheelchair. She had no history of smoking or rheumatoid arthritis.


Figure 1
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  		Fig 1. Photograph shows severe bilateral digital clubbing.

 
Clinical examination found classic clubbing with red, warm, and enlarged tender terminal phalanges of both hands and feet. She had painful and limited range of movement of both knees and ankles. Results of blood tests were normal except for a raised erythrocyte sedimentation rate. A computed tomography (CT) scan revealed a T4N2M0 lesion in the left hilum, with evidence of direct mediastinal and pericardial invasion in addition to significant mediastinal lymphadenopathy and pleural effusion.

A frozen section from a bronchoscopic biopsy specimen revealed a primary pulmonary adenocarcinoma. At the same general anaesthetic session, she underwent a left VATS truncal vagotomy. Single lung ventilation was established and three conventional ports were triangulated around the scapula. Adhesions in the pleural space were carefully dissected using diathermy scissors. The left vagus nerve was approached below the aortic arch within the fatty tissue posterior to the lung hilum. It was disconnected using the snare diathermy, and the severed edges were further diathermized to ensure complete division from each other.

Postoperatively, the patient experienced instant pain relief of all joints, and she regained full range of movements within 24 hours. She was discharged on the third postoperative day and remained pain free and fully mobile 3 months after the surgery.


   Comment
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The HPOA syndrome affects individuals of all races, with equal male-to-female distribution. The most common age group corresponds with the peak of lung cancer (55 to 75 years). In adulthood, it is commonly associated with non-small cell lung cancer and mesothelioma. It is more common with large central tumors than peripheral tumors, with no predilection for cell type [2]. HPOA is also associated with pulmonary metastases from extrathoracic neoplasms such as bone and soft tissues sarcomas and, to a lesser degree, tumors of the nasopharynx, uterus, and cervix. HPOA is rare in children, usually associated with lung metastasis from osteogenic sarcoma [3].

Although the association of HPOA with chronic lung and heart diseases was first reported by Marie and colleagues in 1890, the etiology is still unknown. Vagotomy has been reported to improve HPOA, suggesting a role for reflex vagal stimulation [4, 5]. Removal of the associated lung neoplasm or correction of a cyanotic heart malformation has similar effect, suggesting that alteration of lung function plays an important role. Paraneoplastic growth factors, neurologic, hormonal, immune mechanisms, and vascular thrombi caused by platelets and antiphospholipid antibodies have all been proposed as possible causes. Martinez-Lavin and colleagues postulate the interaction between activated platelets and the platelet-derived and endothelium-derived growth factors to be the underlying cause [6].

HPOA commonly affects joints of distal interphalanges and long bones. The onset of its clinical presentation varies according to the evolution of the underlying lung cancer. These joints become tender, red, warm, swollen, and restricted in range of movement. HPOA may occasionally precede the appearance of constitutional and respiratory symptoms by several months [1].

To date, there is no known cure for HPOA associated with lung cancer. Nonsteroidal antiinflammatory drugs (NSAIDs) may be helpful in providing pain control [7]. Atropine (chemical vagotomy) and antitumor chemotherapy have been used as temporary symptomatic relief [8]. In patients with HPOA, tumor resection often results in improvement of symptoms within 2 to 4 weeks and, sometimes, complete resolution by 3 to 6 months [6]. Surgical vagotomy from a cervical, mediastinotomy, or thoracotomy approach [4, 5] has been reported with varying degrees of success. Systematic review of the literature had not identified VATS as an access for this indication. VATS has long been established as a simple and safe procedure. It is also thought to be less invasive compared with the mediastinal and cervical approaches.

In conclusion, VATS access should be considered to perform the truncal vagotomy in these highly selected patients who have symptoms of HPOA associated with inoperable lung cancer. Patient referral for pain relief by rheumatologists and chest physicians should be encouraged.


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 Abstract
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 References
 

  1. Martinez-Lavin M, Matucci-Cerinic M, Jajic I, Pineda C. Hypertrophic osteoarthropathy: consensus on its definition, classification, assessment and diagnostic criteria J Rheumatol 1993;20:1386-1387.[Medline]
  2. Sridhar KS, Lobo CF, Altman RD. Digital clubbing and lung cancer Chest 1998;114:1535-1537.[Medline]
  3. Howard CP, Telander RL, Hoffman AD, Burgert EO. Hypertrophic osteoarthropathy in association with pulmonary metastasis from osteogenic sarcoma Mayo Clin Proc 1978;53:538-541.[Medline]
  4. Yacoub MH. Vagotomy through mediastinoscopy for pulmonary osteoarthropathy Br J Dis Chest 1966;60:144-147.[Medline]
  5. Yacoub MH. Cervical vagotomy for pulmonary osteoarthropathy Br J Dis Chest 1965;59:28-31.[Medline]
  6. Martinez-Lavin M. Hypertrophic osteoarthropathy Curr Opin Rheumatol 1997;9:83-86.[Medline]
  7. Uchiyama G, Ishizuka M, Sugiura N. Hypertrophic pulmonary osteoarthropathy inactivated by antitumor chemotherapy Radiat Med 1985;3:25-28.[Medline]
  8. Lopez-Enriquez E, Morales AR, Robert F. Effect of atropine sulfate in pulmonary hypertrophic osteoarthropathy Arthritis Rheum 1980;23:822-824.[Medline]



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