Lung metastasis induced chronic compartment syndrome of the lower leg

INTRODUCTION

Compartment syndrome is a complication caused by fracture, ischemic reperfusion injury, soft tissue trauma, external compression, burn, or even bleeding disorders.¹ The syndrome occurs when the pressure within a defined compartmental space increases over a critical pressure threshold, thereby decreasing the perfusion pressure to that compartment. Pain that is out of proportion to the injury and aggravating pain with passive stretching of the muscles are the earliest and most reliable indicators of acute compartment syndrome.² Lung cancer is one of the most frequently diagnosed major cancer in the world and the most common cause of cancer mortality worldwide whose incidence continues to rise. Most lung cancers are associated with a well-known carcinogen-cigarette smoke, in addition with other genetic and environmental factors. Lung cancer most commonly metastasizes to regional lymph nodes, liver, brain, distant lymph nodes, adrenals and bone, while metastasis to the lower leg is very rare.^3,4 We present a unique case of a patient with acute atraumatic compartment syndrome of the lower leg due to a pulmonal carcinoma metastasis.

CASE REPORT

A 50 year old male with a 9 months earlier known diagnosis of metastatic lung adenocarcinoma was admitted to our emergency department with a two month history of increasingly painful left lower leg, swelling and paresthesia of the medial side of the foot. Physical examination revealed an ill-defined, tense, and tender swelling left calf. Dorsalis pedis artery and posterior tibial artery pulsations were intact, but there was numbness over the medial sole and medial tree toes. The patient complained of a foot drop 7 days ago and extreme pain developed the previous day. The patient was treated for deep vein thrombosis before admission. X-ray images of the left lower leg revealed periosteal reaction on all four cortices of both bones (Figure 1). No osteolytic lesions were noted. Pulmonary X-ray images revealed complete atelectasis of the right lung (Figure 2). The white blood cell count was 17x109 l and the C-reactive protein level was also elevated (209 mg/L). Anaemia was present (Hgb 102 g/l) with blood clotting in normal limits. Preoperative liver and renal function were within normal limits. Under the tentative diagnosis of compartment syndrome with associated compressive neuropathy, urgent operation with debridement, biopsy and fasciotomy was performed.

In the operating room two incisions and all three compartment decompression procedure was performed. Upon dissection into the compartments of the lower leg, abnormal solid tissue was evacuated. The tibia and the fibula bone surface was extremely rough. The samples for microbiology and pathology analysis ware taken. The incisions were left open in order to monitor wound condition.

For the first 24 hour the patient stated of having no pain. The patient remained afebrile. Repeated laboratory workup revealed postoperative anaemia which was corrected with two units of blood transfusions. Microbiologic examinations were all negative. On hospital day 13, the pathologic examination confirmed the diagnosis of metastatic pulmonary cancer. Tumor tissue was consisted of large, atypical cells with hyperchromatic nuclei and increased mitotic index with numerous pathological mitoses (Figure 3a).

Tumor cells were arranged into sheets, solid and abortive adenoid structures with extensively areas of necrosis. Immunohistochemical analyses (IHC) showed immunopositivity for cytokeratin 7 (Figure 3b), High molecular weight cytokeratin (Figure 3c) and TTF-1 (Figure 3d) and the diagnosis of metastatic poorly differentiated pulmonary adenocarcinoma was set.

Pulmologists were immediately consulted for confirmation of palliative treatment. During wet dressing management of the fasciotomies, abnormal tissue was noted rising above the fascia (Figure 4). The second procedure, above knee amputation, was performed on the 16-th hospital day. Drainage was removed on the second postoperative day. There were no postoperative wound healing complications. At his personal request, the patient was discharged home on the 17-th hospital day. On control visits he remained pain free.

DISCUSSION

Lung cancer is a highly aggressive disease with an overall 5 year survival rate of less than 15% and the third most common form of cancer to spread to bone. It is considered to be a fast progressing disease with a short latency period after clinical diagnosis. About 30–40% of patients with lung cancer develop bone metastases during the course of their disease.⁵ Cancer cells are capable of infiltrating and colonizing remote organs including bone, contralateral lungs and the brain simultaneously.⁶ In a retrospective study of 259 nonsmall cell lung cancer patients, the most common site of skeletal metastases was the spine in 50% of patients, followed by the ribs (27.1%), ilium (10%), sacrum (7.1%), femur (5.7%) and humerus, scapula and sternum (2.9%). No lower leg skeletal metastases were noted.⁷ The lower leg is the least affected by bone metastases with a rate under 0.1%.⁸ In the literature available to us, there were only three case reports of tibial bone metastasis due to lung cancer.^9-11 Two of them were poorly differentiated adenocarcinomas, as in our case, and one was a small cell carcinoma.

In regard to the compartment syndrome, traumatic causes are very rare.¹² There are only 7 documented cases of compartment syndrome caused by hematologic diseases in the skeletal muscle, but in only two cases it was leg compartment.¹³ Furthermore, there are only two documented solid tumor metastasis complicated with compartment syndrome. One melanoma and one breast cancer with upper arm and forearm compartment syndromes.^14,15 To our knowledge, there are no documented pulmonal bone metastases complicated with lower leg compartment. The reason may be because many patients with metastatic lung carcinoma have a very short survival period. The mean survival of patients with metastatic lung cancer is about 8-10 months.¹⁶

One limitation of the case report is lack of intracompartmental pressure measurement. As the surgical treatment was urgent and there was rapid reduction of postoperative pain, we do not think that these measurements would alter our treatment.

CONCLUSION

Althought very rare, lower leg compartment syndrome can be caused by metastatic deposits of diverse tumors as well as pulmonary adenocarcinoma. It is likely that this complication will be seen more often with the worldwide increased incidence of lung cancer. Great caution is necessary in the treatment of disseminated malignancies when hard swelling of the extremities occurs.

Funding: No funding sources

Conflict of interest: None declared

Ethical approval: Not required

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