Letters to the editor / Joint Bone Spine 73 (2006) 570–578
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Spinal metastasis from a primary parotid carcinoma: a case report Keywords: Spinal secondary; Parotid metastasis
1. Introduction Spinal metastasis originating from various organs is documented in literature but there are few reports of malignant salivary gland tumours causing secondary deposits in the spine. We report a rare case of spinal metastasis from a carcinoma ex-pleomorphic adenoma. Salivary gland neoplasms and the clinical presentation of parotid tumours in particular are described. The treatment options and indications for surgical intervention are reviewed. 2. Case presentation A 65-year-old man presented with low back pain radiating down the left leg for a duration of 6 months. He related the pain to a twisting injury, which he had while playing golf. The pain got worse over a six-month period, but he was able to get along with most of his daily routine activity with the help of analgesics. He also noticed that he had lost about 5 kg in three months. Seven years previous to this presentation he had undergone a superficial parotidectomy for pleomorphic adenoma in the left parotid gland (diagnosed on histopathology). Three years later he had a total parotidectomy for a recurrence, and the following year he had another recurrence. Fine needle aspiration cytology (FNAC) confirmed carcinoma ex-pleomorphic adenoma, which was then treated by radical excision and radiotherapy. Clinical examination revealed spinal tenderness from L3– L5, with paraspinal muscle spasm, but no obvious deformity in the spine. On a detailed neurological examination he had sensory hypoesthesia in the left L4/5 dermatome and no motor deficits. Deep tendon reflexes were normal except for depressed ankle jerk on the left side. Digital rectal examination was normal. Roentgenograms of lumbosacral spine showed osteolysis in the left half of body of L4 with absence of the pedicle of the same side. Bone scan showed an area of increase uptake around the L4 vertebrae, surrounding soft tissues, and in the left hip. MRI scan of the lumbar spine showed an invasive mass involving the left half of the L4 vertebral body, lamina, pedicle and transverse process. There was invasion of the surrounding para-vertebral muscles (Figs. 1a,b,c). A CT-guided biopsy of the lesion confirmed metastasis from malignant pleomorphic adenoma of the parotid (Fig. S1; see the complementary material associated to this article). Further radiographs showed a lytic lesion in the left acetabulum consistent with the increased uptake seen on the bone scan. In view of the evidence of more than one metastatic lesion and the absence
Fig. 1a. Coronal MRI scan of the vertebral lesion showing involvement of the 3rd and 4th lumbar vertebrae and extending into the para-spinal region.
Fig. 1b. Sagittal MRI scan of the lesion in the lumbar vertebrae.
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Letters to the editor / Joint Bone Spine 73 (2006) 570–578
Fig. 1c. Axial MRI scan at the level of the 4th lumbar vertebrae showing destruction of the body of the vertebrae and extension into the surrounding para-spinal region.
of compression on the neural elements, it was decided to treat the patient with radiotherapy. Following radiotherapy, there was a marked decrease in pain and there was no further deterioration of the neurology. Four months later he developed extensive metastatic lesions in the both lungs, which were treated with radiotherapy. He remains under regular review and it is now 18 months after the initial diagnosis of the spinal metastasis. At the last review he had persistence of the hypoasthesia at the left L4/5 dermatome, but no further deterioration of the neurological deficits. 3. Discussion 50–85% of cancer patients ultimately develop skeletal metastasis, most commonly to the spine [1,2]. Skeletal metastasis is seen in 60% of cancer patients at autopsy [3], but this may be an underestimate since detailed dissection of each vertebra is seldom practical, unless this is being specifically sought for. The main sources of vertebral metastasis are tumours of the breast, prostate, lungs, kidney and thyroid. The presence of a valveless venous system [4,5] and the high content of active marrow [6] in the vertebrae are the recognized factors for the high incidence of secondaries in the spine. Salivary gland tumours are relatively rare, forming only about 2% of head and neck neoplasms. Over 80% of these occur in the parotid gland, 80% of which are benign, the commonest being benign mixed tumours (pleomorphic adenoma) [7]. Malignant parotid gland tumours tend to be locally invasive and may show involvement of regional lymph nodes. However, if all malignant parotid tumours are put together, distant metastasis has been reported in 21% [8]. Carcinoma ex-
pleomorphic adenoma is a rare aggressive tumour with a reported 5-year survival rate of only 50% [9]. It develops in a pleomorphic adenoma and takes the form of either adenocarcinoma, undifferentiated carcinoma or, more rarely, a biphasic “carcinosarcoma”. Distant metastasis from carcinoma expleomorphic adenoma is an uncommon presentation but has been reported in the lung and bone [9,10]. This occurs due to haematogenous spread and hence may involve the spine. Skeletal and intradural metastasis has been reported from adenoid cystic carcinoma of the submandibular gland [11,12]. Recommended therapy for benign mixed tumour is its total surgical excision with a rim of normal salivary gland. Recurrence rates and metastases after such a procedure are almost zero but are very high if the tumour is simply enucleated, because of the high chance of incomplete excision [13]. Malignant transformation of benign mixed tumour is an uncommon but well-recognized event, occurring as a complication in up to 5% cases. The malignant component always has an epithelial nature, may assume the appearance of one of the wellrecognized types of salivary gland carcinoma, or, as was the case here, manifest as an undifferentiated small cell carcinoma. It is important to have a high index of suspicion for secondaries in the spine in a patient presenting with low back pain with known history of a primary malignancy. Although life expectancy after spinal metastasis is compromised, prompt treatment is required for pain relief and improvement of the quality of life of these patients. The treatment may be surgery or radiotherapy. Operative decompression is indicated if there is evidence of compression of the neural elements by the tumour. Surgery may be curative in solitary metastasis once the primary has been treated. Operative stabilisation is indicated in pathological fractures, impending fractures, spinal instability or progressive deformity. Radiotherapy is useful for radiosensitive lesions, multiple metastasis, inoperable tumours and as a palliative measure for pain control. It has low morbidity and its efficacy for pain relief is about 80–90% [14,15]. Vertebral body collapse may be prevented and the growth of the tumour can be inhibited by radiation [16,17]. In this case radiotherapy has alleviated the symptoms and has prevented the progression of the neurological deficit at the 18-month follow-up. Supplementary material Supplementary material (Fig. S1) associated to this article can be found at (http://www.sciencedirect.com/, at doi: (10.1016/j.jbspin.2005.12.010). References [1] Asdourian PL. Metastatic disease of the spine. In: Bridwell KH, DeWald RL, editors. The textbook of spinal surgery. Philadelphia: JB Lippincott; 1991. p. 1187–242. [2] Galasko CSB. The Anatomy and Pathway of skeletal metastasis. In: Weiss R, Gilbert W, editors. Bone metastasis. Boston: G.K. Hall; 1981. p. 49–63.
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Drew M, Dickson RB. Osseous complications of malignancy. In: Lokich JJ, editor. Clinical cancer medicine: treatment tactics. Boston: G.K. Hall; 1980. p. 97–124. Batson OV. The role of the vertebral veins and their role in the spread of metastasis. Ann Surg 1940;112:138–49. Batson OV. The function of the vertebral veins in metastatic process. Ann Intern Med 1942;16:38–45. Berrettoni BA, Carter JR. Mechanism of cancer metastasis to bone. J Bone Joint Surg Am 1986;68:308–12. Townsend Jr. CM. In: Sabiston textbook of surgery. The biological basis of modern surgical practice, 16th edition. Saunders; 2001. p. 547–9. Kaplan MJ, John ME. Malignant neoplasms. In: Cummings C, editor. Otolaryngology, Head and Neck Surgery, 2nd edition. Mosby-Year Book Inc; 1993. p. 1043–77. Kerr A, Stell PM. Malignant Salivary Gland Tumours. In: Gleeson M, editor. Scott-Browns otolaryngology, Vol. 5. Reed Educational and Professional Publishing; 1997. p. 1–15. Terhaard CHJ, Lubsen H, van der Tweel I, Hilgers FJM, Eijkenboom WMH, Marres HAM, et al. Salivary gland carcinoma: Independent prognostic factors for locoregional control, distant metastasis and overall survival: results of the Dutch Head and Neck oncology cooperative group. Head Neck 2004;26:681–93. Birkeland S. Spinal metastasis of submandibular gland adenoid cystic carcinoma—a case report. Surg Neurol 2003;60:265–6. van der Wal JE, Becking AG, Snow GB, van der Waal I. Distant metastases of adenoid cystic carcinoma of the salivary glands and the value of diagnostic examinations during follow-up. Head Neck 2002;24:779–83. Thomas WH, Coppola ED. Distant metastasis from mixed tumours of the salivary glands. Am J Surg 1965;109:724–30. Tong D, Gillick L, Hendrickson FR. The palliation of symptomatic metastasis: final results of the study by the radiation therapy oncology group. Cancer 1982;50:893–9. Vargha ZA, Glicksmann AS, Boland J. Single dose radiation therapy in the palliation of metastatic disease. Radiology 1969;93:1181–4. Aabo K, Walbom-Jorgensen S. Central nervous system complications by malignant lymphomas: Radiation schedule and treatment results. Int J Radiat Oncol Biol Phys 1986;12:197–202. Matsubayashi T, Koga H, Nishiyama Y, et al. The reparative process of metastatic bone lesions after radiotherapy. Int J Clin Oncol 1981;11:253.
Abraham Manoj-Thomas* Harshad Dabke Katherine Hammer Richard Attanoos Sashin Ahuja The Department of Orthopaedics, Radiology and Pathology, University Hospital of Wales, CF14 4XW Cardiff, UK E-mail address:
[email protected] (A. Manoj-Thomas). Received 17 May 2005; accepted 15 December 2005 Available online 05 April 2006 *Corresponding author. Department of Trauma and Orthopaedics, University Hospital of Wales Cardiff, 46 Clos Cwm Creunant, Pontprennau, CF23 8LA Cardiff, UK.
1297-319X/$ - see front matter © Elsevier SAS. All rights reserved. doi:10.1016/j.jbspin.2005.12.010
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Microbiology and prognosis of septic arthritis in Brazzaville Keywords: Septic arthritis; Diagnosis; Microbiology; Sub-Saharan Africa
Little information is available on bacteriologically documented septic arthritis in sub-Saharan Africa. Because resources are scarce, bacteriological documentation is rarely obtained in patients with septic arthritis. Thus, although the prevalence of septic arthritis is high in sub-Saharan Africa, most of the published data come from studies on the diagnosis of arthritides in general and from epidemiological surveys of rheumatic diseases in hospital patients [1,2]. We describe the microbiological findings and outcomes in 47 patients with pyogenic septic arthritis documented by microbiological studies of joint fluid samples. These patients represented 24.3% of all patients with septic arthritis admitted to the rheumatology department of the Brazzaville Teaching Hospital, Brazzaville, Congo, from 1989 to 1996; during this period, 1730 patients were admitted to the department. There were 28 men and 19 women, with a mean age (± S.D.) of 41.7 ± 11.3 years. One joint was involved in 46 patients and two joints in one patient. The knee was involved in 37 patients, the hip in five, the ankle in two, the elbow in two, the shoulder in one, and the wrist in one. Mean time from symptom onset to presentation was 12 ± 7 days (range, 2– 23 days). Risk factors included CDC stage IV AIDS in nine patients, type 1 diabetes mellitus in five patients, and homozygous sickle cell disease in five patients. S. aureus was recovered from joint fluid in 28 patients, Streptococcus in 8 patients, and Gram-negative organisms in 11 patients (Salmonella, N = 4; Enterobacter, N = 3; Proteus, N = 2; Klebsiella, N = 1; and Escherichia coli, N = 1). All strains were susceptible to methicillin and gentamicin. Susceptibility to quinolones was not tested. Mean hospital stay length was 34.5 days and mean antibiotic therapy duration was 54 days (range, 26–92 days). Empirical antibiotic therapy consisted of oxacillin and gentamicin. Patients whose microbiological studies showed Gramnegative organisms were switched to amoxicillin. Repeated joint aspiration and immobilization were used routinely. None of the patients underwent surgical treatment. A 74-year-old patient died with evidence of septicemia 3 days after admission. Eighteen patients were lost to follow-up. Of the 28 patients who were evaluated after 3 months, 19 had residual joint damage, which consisted in cartilage loss in all 19 cases and osteolysis in cases. We found no differences between patients with and without AIDS, in terms of the causative organism or the outcome [3]. The organisms in the 9 patients with AIDS were S. aureus (N = 4), Streptococcus (N = 2), Enterobacter (N = 1), Proteus (N = 1), and Klebsiella (N = 1). In the 5 patients with sickle cell disease, the organisms were Salmonella (N = 2), Staphylococcus (N = 2), and Enterobacter (N = 1). None of our patients had joint prosthesis infection, probably because joint replacement surgery is not available in Brazzaville.
