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An isolated chest wall metastasis from cervical squamous cell carcinoma after 19 years of disease-free survival and no local recurrence: A case report and review of literature.

Byline: Ying-Yi. Chen, Chih-Hsin. Wang, Wen-Chuan. Tsai, Mu-Hsien. Yu, Shih-Chun. Lee, Hung. Chang, Tsai-Wang. Huang

Chest wall tumors can arise from a wide variety of benign and malignant etiologies, and therefore it can be challenging for clinicians to diagnose and manage them. Isolated metastatic squamous cell carcinoma from cervical cancer is extremely rare. We report a metastatic chest wall tumor from cervical cancer arising after 19 years of disease-free survival. Wide resection of the metastatic chest wall tumor, followed by a polypropylene mesh and free flap reconstruction with adequate coverage was successful. The patient continues to be a disease-free at 2 years.

Introduction

Carcinoma of the uterine cervix is the most common gynecological malignancy in developing countries and 15-61% of women with cervical cancer will develop distant metastases within the first 2 years of completing treatment. [sup][1] In patients who develop distant metastases, the most frequently observed metastatic sites are the lung (21%), para-aortic nodes (11%), abdominal cavity (8%), and supraclavicular nodes (7%). Bone metastases occurred in 16% of patients, predominantly involving the lumbar and thoracic spine. [sup][2] A huge chest wall tumor caused by distant metastasis from cervical cancer is uncommon. Tumors of the chest wall vary from benign to malignant and can arise from any soft tissue or bony structure. The most common primary malignant chest wall tumor is a sarcoma, arising from soft, cartilaginous, or bony tissues. [sup][3] Primary soft tissue neoplasms of the chest wall include peripheral nerve tumors, lipomas, liposarcomas, hemangiomas, elastofibromas, lymphomas, desmoid tumors, and malignant fibrous histiocytomas. [sup][4] Most metastases to the chest wall are from adjacent structures, commonly the breast, lung, pleura, and mediastinum. [sup][5] Distant metastatic chest wall tumors from soft tissue carcinoma are rare. In this report, we report a rare case of metastatic squamous cell carcinoma (SCC) arising from the cervix, with successful management using chest wall resection following reconstruction with a polypropylene mesh and free-flap coverage.

Case Report

A 67-year-old woman was referred to our department for a protruding mass in the left anterior chest wall [Figure 1]a that had persisted for 2 years. The patient denied history of any medical diseases except for SCC of the cervix, Stage 1B1. She had undergone radical hysterectomy 19 years previously. Postoperative adjuvant chemotherapy (cisplatin and 5-fluorouracil) and radiotherapy had been administered. The patient had experienced long-term, disease-free survival with regular follow-up until 10 years ago.{Figure 1}

Two years before this admission, she developed a painful chest wall mass. She initially ignored its development and then felt shame for the progressively enlarging mass because of its weird outward appearance until the pain in the left anterior chest wall became intolerable. Computed tomography (CT) of the chest revealed a confluent soft tissue mass in the left anterior wall [Figure 1]b. Sonography-guided needle biopsy for this chest wall tumor revealed SCC pathology. After comprehensive evaluation, including positron emission tomography (PET) scans and physical examination, there were no other metastases or local recurrences of the previously treated cervical cancer.

The patient underwent chest wall resection including partial resection of the sternum and lung because of partial sternal destruction and direct invasion of the anterior segment of the left upper lobe of the lung. A mass measuring 10 cm x 8 cm was removed leaving a clear surgical margin [Figure 2]. The chest wall defect was reconstructed using a polypropylene mesh, followed by a free-flap (an anterolateral flap from the right thigh) coverage [Figure 3]. Pathological examination of the resected specimen was compatible with metastatic SCC arising from the cervix [Figure 4]. No sequela or complication of sternum resection and free flap reconstruction were noted. After 2 years of follow-up, with physical examination and chest X-rays every 3-6 months, there was no evidence of local recurrence nor distal metastases [Figure 5].{Figure 2}{Figure 3}{Figure 4}{Figure 5}

Discussion

More than 50% of chest wall tumors are malignant, typically involving direct invasion or metastases from adjacent thoracic tumors. Metastatic chest wall tumors arising from soft tissue sarcomas or carcinomas are rare. [sup][5] Some inflammatory conditions, such as abscesses and inflammation, can also present as a chest wall mass. Due to the wide variation in the histology and origin of chest wall tumors, the differential diagnosis can be challenging. The integration of imaging information, location, and clinical information are required to make a precise diagnosis.

In our case, the patient developed a large mass in the left anterior chest wall. CT scans of the chest revealed bony involvement of the sternum and pleural invasion. With malignant tumors, this usually arises from bony involvement and is frequently a sign of invasion. Although the patient had a history of cervical cancer, she had experienced disease-free survival for >5 years without evidence of relapse. Biopsy of this tumor was necessary for pathological identification to aid clinicians in arranging subsequent examinations for surgical indications. Based on the preoperative diagnosis of a solitarily recurrent cervical SCC metastasis in the left chest wall, the surgical indications, risks, and complications, including the safe surgical margin of wide excision and consultation with a plastic surgeon for reconstruction with a polypropylene mesh and free-flap coverage, were fully disclosed to the patient and her family before surgery.

Squamous cell carcinoma of the cervix is a common form of gynecologic malignancy. As a result of the implementation of active screening programs, most cases are identified at Stage I. Carcinomas of the cervix usually spread in an orderly and predictable fashion. The earliest and most common metastases are by direct extension to contiguous structures, including the vagina, peritoneum, urinary bladder, ureter, rectum, and paracervical tissues. [sup][6] However, distant metastates involving the lung, liver, bone marrow, and gastrointestinal tract have also been reported. [sup][7] Up to 50% of patients with Stage IV SCC can present with distant metastases. [sup][4] The gastrointestinal tract is involved in approximately 8% of cases, most commonly in the rectosigmoid colon as a result of local extension. Isolated metastases to the chest wall from cervical SCC are rare. In this case, the patient had experienced 19 years of disease-free survival after her previous operation and adjuvant therapy. Disease relapse presented as a chest wall mass without local recurrence or the other distant metastases. PET and CT scans showed no abnormality except the chest wall lesion.

The chest wall tumor likely originated from an isolated skin metastasis at left chest wall and progressed. The mechanism of metastasis can be understood as a series of steps providing three basic patterns of distribution: Mechanical (arising from anatomical proximity and lymphatic draining), site-specific (arising from the selective attachment of tumor cells to a specific organ), and nonselective (independent of these distribution factors). [sup][8] It has been well-documented that metastases from internal carcinomas can spread to the skin via the vascular channels and present as 1-3 cm dermal nodules at a distance from the primary site. [sup][9] Cutaneous metastatic lymphangitis is rarely reported in association with cervical cancer and may be related to radiotherapy as described in some cases of breast cancer. [sup][9] The proposed mechanism of cutaneous metastasis is radiation-induced endothelial cell damage leading to tumor cell trapping. [sup][10],[11] It is usually confined to the irradiation fields, appearing months to years after treatment completion. [sup][10] Another probable mechanism is alteration of the lymphatic flow. [sup][9],[12] In most cases, it probably results from the obstruction of deep lymphatic pathways by the tumor with shunting of the lymphatic flow to the cutaneous lymphatics. [sup][12],[13],[14] In cases of cutaneous metastasis in patients with breast cancer, local changes within the vasculature of the irradiated area are thought to contribute to the trapping, growth, and survival of tumor cells. [sup][10] However, radiation has not been shown to increase the incidence of metastasis. [sup][10] In our case, the metastasis could be attributable to a retrograde spread of tumor secondary to lymphatic obstruction [sup][13] or to a hematogenous spread. [sup][14]

Cervical cancer rarely metastasizes to the skin, occurring in <2% of patients. [sup][15],[16],[17],[18] Patients with cutaneous metastatic disease may present at the time of diagnosis or up to 10 years later. [sup][12],[13],[14] The presence of metastatic disease in the skin signals a poor prognosis and the average reported survival is 8.5 months. [sup][8],[12],[19] Until date, no effective treatment has been identified for such lesions and care is palliative, using radiation, chemotherapy, and surgery, alone or in combination. [sup][11],[12] The most common sites of skin metastases from various solid malignancies are the anterior chest and abdominal walls. [sup][20],[21],[22] In our case, there was no local recurrence of SCC of the cervix and no other distant metastases identified by gynecological examinations and whole body PET scan. After discussion with the patient and her family, cosmetic concerns and quality of life were primary concerns. Therefore, they agreed to perform wide excision of chest wall tumor combined with reconstruction with a polypropylene mesh and free flap coverage. The extent of safe margin was more than 2 cm, which was confirmed intraoperatively by frozen section.

Since the first known chest wall reconstruction in the 18 century by Tansini, [sup][23] the advent of antibiotics, improvements in anesthesia and critical care, and developments in ablative and reconstructive surgery have allowed extensive chest wall resection to be performed with acceptable morbidity and mortality. Malignancies of the chest wall tend to be aggressive and often infiltrate deeply, thus necessitating extensive ablative surgery to ensure tumor-free margins. [sup][24] The aim of reconstruction is to restore reliable and durable soft-tissue coverage to protect the intra thoracic structures; avoid disruption of respiratory functions, and provide, if possible, acceptable esthetic outcomes. [sup][25] Chest wall reconstruction following extensive resection is greatly facilitated by the use of vascularized flaps and prosthetic material. Communication between the ablative and reconstructive teams is important to ensure appropriate preoperative planning regarding the extent of resection, keeping in mind the various reconstructive options including .management of the pleural cavity, skeletal support, and soft tissue coverage. [sup][26],[27] A thorough understanding of the respiratory mechanics and local thoracoabdominal anatomy is crucial for managing these complex defects. The first option is the use of loco-regional muscle and myocutaneous flaps as they permit immediate single-stage chest wall reconstruction. [sup][25],[28],[29] Losken et al . [sup][26] presented a reconstructive algorithm for plastic surgery following extensive chest wall resection according to their clinical experience in 200 patients with chest wall resections. However, all of their procedures used regionally pedicled flaps and the authors thought flap reconstruction was required more often to cover large anterior defects, with regional flaps predominating. For cases in which the resections are too wide for loco-regional muscle flaps or involve the supporting skeletal elements and/or the vital intra thoracic structures, the reconstructive problem becomes more complex and therefore, distant, reliable, well-vascularized flap coverage is mandatory. [sup][30] Di Candia et al . [sup][31] analyzed a single plastic surgeon's (Charles M. Malata) experience with five central composite chest wall reconstructions using the anterolateral thigh (ALT) free flap to outline its indications and benefits. However, the ALT free flap has some drawbacks in this setting. First, it involves microsurgery with all its attendant risks in older patients. [sup][32] Despite its bulk, it does not provide adequate skeletal support to the chest wall. Another disadvantage is the poor color match, though it is not a major problem at this site because it is usually hidden by clothes. Despite these drawbacks and the reported variations in vascular anatomy and technical difficulties in raising the ALT free flap, [sup][33] Candia has shown that the ALT free flap is a practical approach that is particularly useful in reconstructing wide central complex chest wall defects. [sup][34] Our single case report shows that the ALT free flap is both safe and effective for reconstructing large complex defects, as it adequately obliterated

the dead space and provided reliable coverage of synthetic material with acceptable esthetic and functional outcomes. We, therefore, suggest that this versatile flap should be considered as a practical and reliable option for massive chest wall reconstruction, even in the hands of low-volume operators. [sup][31]

The historical challenges of diagnosis, complete resection, and reconstruction have posed difficulty for surgeons attempting to minimize perioperative morbidity, but advances in all of these areas have greatly improved long-term survival and decreased tumor recurrence rates. [sup][4] The role of surgical resection of metastatic diseases to the chest wall or of local recurrent breast cancer is controversial because the former reflects disseminated disease and the latter has a poor prognosis. Resection may be considered when:

*The chest wall is the only site of disease; *loco-regional disease is controlled; and *complete resection with a negative margin is possible.

Resection of the chest wall, followed by free-flap coverage was done in the present case. The latissimus dorsi was not recommended as a flap source by the plastic surgeon because of the large defect and the small available muscle region in this thin female patient. In addition, the patient has preserved good functional outcome after the operation. Patients with known carcinoma of the cervix who develop pruritic or nonpruritic maculopapular skin eruptions, plaques, or nodules should undergo prompt skin biopsy to rule out metastatic disease and cutaneous metastatic lymphangitis. [sup][35] We propose that skin inspection should be included in the follow-up of cervical cancer, and prompt biopsy recommended after patients are discharged to the care of their primary physician. [sup][35] Early diagnosis and prompt institution of therapy may prolong survival. A metastatic SCC invading the chest wall from the cervix is rarely reported, and free-flap coverage is a good choice for reconstruction of the chest wall after resection. Aggressive surgical intervention resulted in good local control and survival.

Disclosure

The authors declare this study has no conflict of interest

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Title Annotation:Case Report
Author:Chen, Ying-Yi; Wang, Chih-Hsin; Tsai, Wen-Chuan; Yu, Mu-Hsien; Lee, Shih-Chun; Chang, Hung; Huang, T
Publication:Journal of Medical Sciences
Article Type:Report
Geographic Code:1USA
Date:Nov 1, 2014
Words:3146
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