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A rare combination of tumor-induced osteomalacia caused by sinonasal glomangiopericytoma and coexisting parathyroid adenoma: case report and literature review.
Brociek-Piłczyńska, A, Brodowska-Kania, D, Szczygielski, K, Lorent, M, Zieliński, G, Kowalewski, P, Jurkiewicz, D
BMC endocrine disorders. 2022;(1):31
Abstract
BACKGROUND Tumor-induced osteomalacia (TIO) is a rare, acquired disease of renal phosphate wasting and disturbed vitamin D homeostasis as a result of the action of a phosphaturic protein - FGF-23, produced by a neoplasm. Although the clinical and biochemical profile of the syndrome is characteristic, it remains underreported and unrecognized by clinicians. Hyperparathyroidism is rarely associated with oncogenic osteomalacia, but it should be considered because of potentially life-threatening hypophosphatemia caused by both conditions. CASE PRESENTATION We report a case of a 42-year-old woman admitted to the Department of Otolaryngology of the Military Institute of Medicine in Warsaw for the endoscopic resection of hormonally active glomangiopericytoma extending into the anterior skull base. She presented with a 5-year history of musculoskeletal pain and progressive weakness of the extremities which finally led her to become bedridden. After the excision of the tumor her symptoms and laboratory results gradually improved except increasing PTH serum levels. Further examination revealed a parathyroid proliferative tumor, which was surgically removed. The patient walked without aids at follow-up 16 months after the surgery. CONCLUSIONS This case is unusual because of tumor-induced osteomalacia and parathyroid adenoma occurring concomitantly. Further investigations of FGF-23 and PTH interplay should be conducted to elucidate the pathogenesis of hyperparathyroidism and tumorigenesis in some cases of TIO. By presenting this case, we wanted to remind clinicians of a rare and misdiagnosed paraneoplastic syndrome and highlight the importance of monitoring PTH concentrations during the follow-up of patients with TIO.
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2.
Spinal phosphaturic mesenchymal tumors: Case report and literature review.
Wang, X, Gao, J, Han, S, Li, Y
Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia. 2019;:234-239
Abstract
Tumor-induced osteomalacia (TIO) is regarded as a rare paraneoplastic syndrome generally caused by phosphaturic mesenchymal tumors (PMTs). As far as we know, only 18 spinal PMTs have been described in the world's English literature. The purpose of this study is to increase familiarity with its clinical features, diagnosis, and treatment of spinal PMTs. A 34-year-old woman presented with bone pain for more than 3 years and was found to have decreased serum phosphorus, elevated serum alkaline phosphatase. A full body 68Ga-DOTA-TATE PET/CT revealed increased uptake at the left posterior L5 neural arch. Then the tumor was totally resection and the histopathology revealed a PMT. Postoperatively, the patient's symptoms were relieved and experienced no recurrence during the 3-year follow-up. Generally, the preoperative diagnosis and location of spinal PMT still remains challenging. We also retrospectively analyzed 18 cases of patients with spinal PMTs published in English. Clinical symptoms, laboratory findings, treatment and outcome were retrospectively analyzed. Spinal PMTs are extraordinary and it is challengeable in locating tumors. Besides surgical resection, the treatment also included radiation, monoclonal antibodies or medical therapy. However, the optimal treatment remains controversial. Therefore, we should exert all our energies on the exploration of etiology and adjuvant therapy for this disease.
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[Recurrent ataxia and respiratory failure with probable paraneoplastic syndrome responsive to plasma exchange therapy].
Kondo, T, Igari, R, Sato, H, Iseki, C, Ishizawa, K, Suzuki, K
Rinsho shinkeigaku = Clinical neurology. 2019;(6):339-344
Abstract
An 80-year-old male with prostatic adenocarcinoma who was treated with orchiectomy presented dysarthria and difficulty in walking. His symptoms subacutely progressed. Seven days later, he was non-ambulatory and was admitted to our hospital. He had poor vision and cerebellar ataxia of the lower extremities; however, his muscle strength, tendon reflexes, and sensory functions were preserved. Paraneoplastic retinopathy was diagnosed based on electroretinographic and visual field defect. Further, brain and spinal MRI, cerebral spinal fluid, and nerve conduction assessments were normal. These symptoms were followed by consciousness disturbance and respiratory failure; consequently, he required non-invasive positive pressure ventilation (NPPV) and tube feeding. Steroid pulse therapy and plasma exchange (PE) were performed. In response to the therapy, all these symptoms were relieved, and NPPV and tube feeding were withdrawn. However, the same symptoms occurred additional three times throughout the course of approximately 1 year. Each time, PE was the most effective treatment. Although paraneoplastic neurological syndrome associated with prostatic cancer is rare, immunotherapy could be a therapeutic choice to relive symptoms.
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4.
Supportive therapy in gastroenteropancreatic neuroendocrine tumors: Often forgotten but important.
Jin, XF, Spampatti, MP, Spitzweg, C, Auernhammer, CJ
Reviews in endocrine & metabolic disorders. 2018;(2):145-158
Abstract
Neuroendocrine tumors (NETs) are a group of rare and heterogeneous malignancies that can develop in various organs. A significant number of gastroenteropancreatic neuroendocrine tumours (GEP-NETs) is functionally active and presents with symptoms related to the secretion of biologically active substances, leading to the development of distinct clinical syndromes. There are various therapeutic approaches for GEP-NETs, including curative surgery, palliative surgery, local-ablative and loco-regional therapies as well as systemic therapeutic options including peptide receptor radionuclide therapy, cytotoxic therapy, and molecularly targeted therapies. Specific supportive therapy of patients with NETs includes management or prevention of hormone-related clinical syndromes and paraneoplastic states. Supportive therapy plays a key role in NET treatment. Supportive therapy includes debulking surgery and interventional radiologic techniques to reduce tumour bulk or load, as well as systemic medical treatment options to manage or prevent hypersecretion syndromes and treatment-related side effects. Supportive therapies are a type of of comprehensive treatment addressing the patient as a whole person throughout the process of NET treatment. Therefore, supportive therapy also encompasses psychosocial support, expert nursing, nutritional support and management of cancer related pain.
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5.
Nailfold Capillaroscopy Within and Beyond the Scope of Connective Tissue Diseases.
Lambova, SN, Muller-Ladner, U
Current rheumatology reviews. 2018;(1):12-21
Abstract
Nailfold capillaroscopy is a noninvasive instrumental method for morphological analysis of the nutritive capillaries in the nailfold area. In rheumatology, it is a method of choice among instrumental modalities for differential diagnosis between primary and secondary Raynaud's phenomenon (RP) in systemic rheumatic diseases. RP is a common diagnostic problem in rheumatology. Defining the proper diagnosis is a prerequisite for administration of the appropriate treatment. Thus, nailfold capillaroscopic examination is of crucial importance for the every-day practice of the rheumatologists and is currently gaining increasing attention. The most specific capillaroscopic changes are observed in Systemic Sclerosis (SSc). Due to the high prevalence of the capillaroscopic changes in this clinical entity and their early appearance, they could be used for early and very early diagnosis of the disease. More recently, "scleroderma" type capillaroscopic changes have been defined as diagnostic criterion in the new EULAR/ACR classification criteria for SSc together with the presence of scleroderma-related autoantibodies, RP, telangiectasia and other clinical signs. Capillaroscopic changes in other connective tissue diseases and in different rheumatic-like conditions like those in diabetes mellitus (e.g., diabetic stiff-hand syndrome) and paraneoplastic syndromes associated with microvascular pathology should be interpreted properly in order to obtain precise diagnosis in the shortest possible differential diagnostic process.
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6.
Disseminated intravascular coagulation as clinical manifestation of colorectal cancer: a case report and review of the literature.
van Bunderen, CC, de Weger, VA, Griffioen-Keijzer, A
The Netherlands journal of medicine. 2014;(4):186-9
Abstract
We describe the case of a 65-year-old woman, known with ulcerative colitis, who presented with progressive headaches, haematomas and rectal bleeding which turned out to be the initial manifestation of disseminated intravascular coagulation (DIC) associated with colorectal cancer. The presentation posed as a general medicine case but turned out to be a rare oncological complication. The patient revealed possible carcinocythaemia and bone marrow infiltration with signet ring-like cells, as indicators of advanced adenocarcinoma. Treatment of the underlying disease resolved the DIC and contributed to prolonged survival. Subsequently, we reviewed the English literature since 1990 on similar cases and demonstrated that this association is extremely rare and is associated with a poor prognosis. Prompt recognition and treatment of the underlying disease is confirmed to be of utmost importance to prolong (progression-free) survival.
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7.
Humoral hypercalcemia associated with gastric carcinoma secreting parathyroid hormone: a case report and review of the literature.
Nakajima, K, Tamai, M, Okaniwa, S, Nakamura, Y, Kobayashi, M, Niwa, T, Horigome, N, Ito, N, Suzuki, S, Nishio, S, et al
Endocrine journal. 2013;(5):557-62
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Abstract
Hypercalcemia with concomitant elevation of serum parathyroid hormone (PTH) and PTH-related protein (PTHrP) levels was found in a patient with advanced gastric carcinoma and multiple liver metastases. The most common features are hypercalcemia associated with hypersecretion of PTHrP and physiological suppression of PTH secretion in the syndrome of humoral hypercalcemia of malignancy (HHM). Although we initially made a diagnosis of primary hyperparathyroidism concomitant with HHM due to gastric cancer, diagnostic imaging studies, such as echography, CT, sestamibi scintigraphy, and autopsy findings, did not reveal evidence of any parathyroid tumors or ectopic parathyroid glands in the mediastinum. Both primary and metastatic tumor cells showed positive staining with PTH-specific antibody as well as PTHrP-specific antibody on immunohistochemical examination. PTH concentration in the cytosolic fraction of the metastatic tumor was elevated compared to that from a control patient with no calcium metabolic disorders in vitro. These findings indicated that PTH secreted ectopically by gastric cancer cells, not by parathyroid glands, caused hypercalcemia in this patient. To our knowledge, this is the first case report of PTH-secreting gastric carcinoma cells. We report the case and a review of the previous reported PTH-secreting non-parathyroid tumors along with the mechanisms of secretion.
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8.
Recovery of paraneoplastic hypercalcemia by sunitinib treatment for renal cell carcinoma: a case report and review of the literature.
Karaca, H, Lale, A, Dikilitas, M, Ozkan, M, Er, O
Medical oncology (Northwood, London, England). 2010;(3):1023-6
Abstract
Sunitinib is a novel oral multitargeted tyrosine kinase inhibitor. It has higher response rates and progression-free survival in patients with metastatic renal cell carcinoma (RCC) when compared with standard chemotherapy and interferon-alpha. We report a case of paraneoplastic hypercalcemia, resistant to conventional treatment but recovers by sunitinib treatment as the first case in the literature, in a 33-years-old man with metastatic RCC. At the sixth month of follow-up period, in this case, serum calcium level was still in normal ranges. Besides sunitinib is effective in symptom control, it is also helpful in management of paraneoplastic hypercalcemia, a life-threatening entity.
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Efficacy of high-dose intravenous immunoglobulin therapy in Japanese patients with steroid-resistant polymyositis and dermatomyositis.
Saito, E, Koike, T, Hashimoto, H, Miyasaka, N, Ikeda, Y, Hara, M, Yamada, H, Yoshida, T, Harigai, M, Ichikawa, Y
Modern rheumatology. 2008;(1):34-44
Abstract
Intravenous immunoglobulin (IVIG) therapy was administered to 15 patients who were refractory to traditional steroid therapy [eight with polymyosis (PM), seven with dermamyosis (DM)] to evaluate its efficacy. Serum creatine kinase (CK) significantly decreased from week 1, and manual muscle test scores (MMT) and activities of daily living (ADL) significantly increased from week 2. Efficacy rates were 93.3% (14/15 patients) as assessed using the MMT score, 80.0% (12/15 patients) using the ADL score, and 100% (15/15 patients) using the serum CK level. When changes in the serum CK level over two four-week periods, one before IVIG therapy (from week -4 to week 0) and one after IVIG therapy (from week 0 to week 4), were transformed to natural logarithms, the four-week change after IVIG therapy was significantly greater than that before IVIG therapy. The estimated duration of the serum CK level remaining normal in 50% of the patients after IVIG therapy was 334.5 days. Adverse reactions were observed in seven of 16 patients (43.8%) during the study period, but none of the adverse reactions were considered to be serious or required emergency treatment. In conclusion, the present study indicates that IVIG therapy is effective for steroid-resistant PM/DM.
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10.
The kidney, cancer, and the eye: current concepts.
Kurli, M, Finger, PT
Survey of ophthalmology. 2005;(6):507-18
Abstract
Herein, we review the associations between the kidney, renal cancers, and the eye. Renal cancers have been reported to metastasize to the eye and the orbit. As these tumors can be confused with other amelanotic or vascular tumors, a high index of suspicion is required for early detection and management of the primary tumor. We discuss the physiology of metastases, clinical features and management of metastatic disease. A variety of ocular anomalies have been associated with renal disease. Wilms tumor, a renal tumor of childhood, can present with aniridia, which may be the first clue leading to the diagnosis of the primary tumor. Paraneoplastic syndromes are common manifestations of renal cancers and can present as retinopathies and neuro-ophthalmic disorders. Multiple cancer syndromes involve both the eye and the kidney. For example, the diagnosis of von Hippel retinal tumors can lead to a systemic evaluation and discovery of associated visceral tumors. The prognosis, screening, and counseling of such patients is discussed. Newer systemic treatments available for renal tumors, such as interferon alfa, may lead to ocular side effects including retinopathy. These patients require periodic ophthalmic examinations. This review demonstrates the essential role of the ophthalmologist, for early diagnosis and treatment that can help reduce the morbidity and mortality associated with kidney tumors and renal-associated disease.