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ScienceDirect EJSO 39 (2013) 1325e1331

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Periscapular amputation as treatment for brachial plexopathy secondary to recurrent breast carcinoma: A case series and review of the literature N.K. Behnke, S.N. Crosby, C.M. Stutz, G.E. Holt* Department of Orthopaedics and Rehabilitation, Vanderbilt University Medical Center, Medical Center East, South Tower, Suite 4200, 1215 21st Ave South, Nashville, TN 37232, USA Accepted 7 October 2013 Available online 23 October 2013

Abstract Aims: Recurrent breast carcinoma with brachial plexus involvement is often misinterpreted as a radiation- or chemotherapy-induced brachial plexopathy. We review a case series of 4 patients at our institution within a 1-year period, and describe their diagnostic workup and treatment with a palliative periscapular amputation. Our aim is to describe this entity, indications and benefits of this procedure, when required for progressive disease, with the goal of raising a collective index of suspicion to aid in earlier diagnosis. Methods: Four patients with recurrent axillary breast cancer and symptoms consistent with a brachial plexopathy were prospectively collected over a 1-year period. A Pubmed search was conducted; pertinent articles were reviewed and reported. Results: Patients presented with intractable pain and flaccid paralysis of the ipsilateral limb. All had been previously treated with surgical resection, axillary lymph node dissection, chemotherapy, and radiation therapy. Average time from breast surgery to presentation was 78.75 months (range 11e216 months.) Workup included MRI and biopsy to confirm recurrence. Periscapular amputation was performed for each patient, all of who experienced subjective pain relief postoperatively. Three of the 4 patients are still living; one patient died of disease. Conclusion: Breast cancer survivors presenting with a brachial plexopathy should raise suspicion for recurrent disease. Close evaluation with MRI is the best first step in diagnosis. Although periscapular amputation is an aggressive surgical treatment, it is an acceptable option when disease has progressed to neurovascular involvement and a functionless limb. Ó 2013 Elsevier Ltd. All rights reserved. Keywords: Brachial plexopathy; Breast cancer; Periscapular amputation; Forequarter amputation

Introduction Periscapular amputation, including forequarter amputation (FQA) has been historically used as a curative measure for bone and soft tissue tumors.1,2 With the advent of more advanced surgical techniques, chemotherapy and radiation therapy, major amputations have been less commonly employed as a cure for recurrent and metastatic carcinomas, and infrequently used for palliative purposes.3e5 The use of periscapular amputation for palliation has been questioned, as it is a physically disfiguring and emotionally scarring procedure.6 Despite its limitations, in selected patients, periscapular amputation provides effective pain * Corresponding author. Tel.: þ1 615 343 8612; fax: þ1 615 343 1028. E-mail addresses: [email protected], nkbehnke@ uabmc.edu (G.E. Holt). 0748-7983/$ - see front matter Ó 2013 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ejso.2013.10.005

relief and satisfactory functional and symptomatic results.5,7e9 Axillary recurrence of breast cancer is an uncommon event, but can be significantly debilitating, especially if there is infiltrative involvement of the brachial plexus. Typically, unrelenting pain is the presenting symptom of metastatic plexus involvement,10 though a cardinal triad of pain, motor loss and an obliterated axillary vein, as described by Wittig et al., often accurately indicates brachial plexus infiltration and unresectability.3 Unfortunately, this diagnosis is often delayed due to vague neurovascular complaints, the use of chronic narcotics in these patients, or symptoms being misinterpreted as radiationinduced plexopathy or chemotherapy-induced neuropathy.11e16 The overlap of symptoms arising from disease itself, vascular compromise within the axilla, and those arising from radiation- or chemotherapy-induced vascular

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events further complicates the clinical picture.17e20 The delay in diagnosis leads to progression of the disease and worsening pain. We describe a case series of 4 patients who presented to our institution over a 12-month period with brachial plexopathy secondary to metastatic disease. Each patient experienced 1e2 years of pain before accurate diagnosis. Each of them had a painful, flaccid, dysfunctional limb with neck, chest and upper back pain secondary to the weight of the functionless limb. All 4 patients were referred to Orthopaedic Oncology for evaluation, and subsequently went on to amputation for palliation of symptoms with excellent relief of pain. There are scatted reports in the literature about this disease process and its progression to this level of severity. The goal of this paper is description of this entity to aid in earlier diagnosis, and to describe the benefits of periscapular amputation, when required for progressive disease. Patients and methods We performed a retrospective review of medical records on a prospectively collected group of patients presenting to our institution within a one-year period. Four patients with recurrent axillary breast cancer presenting with a brachial plexopathy were identified. Demographics and treatment history of the four patients are detailed in Table 1. At initial presentation, all patients had stage III tumors, indicating lymph node spread but no evidence of distant disease. Three patients had undergone mastectomy as their index surgical procedure; a lumpectomy was performed for one patient. All had axillary lymph node dissection at the

time of surgery. Adjuvant chemotherapy and radiation therapy, with 50 Gy as standard dose, were given to all 4 patients. Notably, all patients received a taxane-based chemotherapeutic agent during their course of treatment. The three patients with estrogen- and progesteronereceptor positive tumors were treated with targeted hormonal therapies; Her2/neu positive tumors were treated with trastuzumab (HerceptinÒ, Genentech, San Francisco, CA.) Once diagnosed with axillary recurrence within the brachial plexus, all patients underwent a salvage chemotherapy regimen; one patient had additional salvage radiation therapy. All had rapid evidence of disease progression prior to referral to Orthopaedic Oncology. Results The criteria for diagnosis of brachial plexopathy were ipsilateral extremity pain and neurologic deficits. In each of the 4 patients, the symptoms had advanced to intractable pain and a flaccid limb before the diagnosis of axillary recurrence of breast cancer was made. Other causes of arm pain and neurologic deficits, such as metastases to the cervical spine, brain and humerus, were ruled out. The mean interval between primary surgery and presentation with brachial plexus symptoms was 78.75 months (Range: 11e216 months.) All patients had been free of pain and neurologic deficits at some point during this interval. The mean time from presentation with brachial plexus symptoms and definitive diagnosis of recurrent axillary breast cancer was 19 months (Range: 12e34 months.) Magnetic resonance imaging (MRI) of the axilla and brachial plexus was used as an initial step in workup

Table 1 Demographics, initial breast cancer stage, treatment regimen and relevant time intervals of 4 patients who underwent periscapular amputation for brachial plexopathy and axillary breast cancer recurrence.

Age at diagnosis Surgery Stagea ER/PRb Her2/neuc Chemotherapy Radiation therapy Interval to symptomsd Symptom duratione Salvage treatmentf Surgical management

Patient 1

Patient 2

Patient 3

Patient 4

59 Mastectomy with axillary dissection III / e Yes Yes 11 mos 12 mos Chemo Forequarter amputation

48 Mastectomy with axillary dissection III þ/þ þ Yes Yes 216 mos 17 mos Chemo Forequarter amputation

57 Lumpectomy with axillary dissection III /þ e Yes Yes 36 mos 13 mos Chemo, XRT Forequarter amputation

55 Mastectomy with axillary dissection III þ/þ þ Yes Yes 52 mos 34 mos Chemo Shoulder disarticulation

Stage at initial breast cancer diagnosis: 0 ¼ non-invasive breast carcinoma; I ¼ invasive breast carcinoma, tumor

Periscapular amputation as treatment for brachial plexopathy secondary to recurrent breast carcinoma: a case series and review of the literature.

Recurrent breast carcinoma with brachial plexus involvement is often misinterpreted as a radiation- or chemotherapy-induced brachial plexopathy. We re...
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