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Care of a Patient with Fibrous Dysplasia of the Clavicle and Ribs

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Care of a Patient with Fibrous Dysplasia of the Clavicle and Ribs Case Report iMedPub Journals Health Science Journal 2015 http://journals.imedpub.com ISSN 1791-809X Vol. 10 No. 1:12 Care of a Patient with Fibrous Dysplasia of the Agnieszka Kruk1, Janina Książek1, Clavicle and Ribs: Case Report Wojciech Żurek2, Sylwia Terech1, Piotr Jarzynkowski1 and Abstract Renata Piotrkowska1 Introduction: Fibrous dysplasia of the ribs and clavicle is a polyostotic form of 1 Department of Surgical Nursing, Medical skeletal system disease. The most commonly reported symptom is pain. Surgical University of Gdansk, Poland treatment, which is the primary therapeutic approach, may contribute to the 2 Faculty and Clinic of Chest Surgery, emergence of complications in surgical site that are difficult to treat. The aim of Medical University of Gdansk, PolandIran the study was to analyze interdisciplinary care of a patient chronically treated for fibrous dysplasia of the clavicle and ribs. Methods: The interview, observation, and analysis of medical records method was Correspondence: Janina Książek used in the study. The case describes a 51-year-old man with a diagnosis of fibrous dysplasia of the clavicle and ribs, hospitalized in the Department of Thoracic Surgery of Medical University of Gdansk (MUG) in the years 1995-2013. Results: Progressive disease process caused a lot of health problems in the patient [email protected] and deterioration in the quality of life in every aspects of his life . Conclusions and relevance for clinical practice: Care and treatment in hospital Zakład Pielęgniarstwa Chirurgicznego, and at home are focused on the pain and chronic wound healing process, which Katedra Pielęgniarstwa GUMed, Dębinki 7, require the involvement of the entire interdisciplinary team. 15, Poland. Keywords: Fibrous dysplasia of bone; Smoking; Interdisciplinary care; Chronic Tel: +48 58 3491247 wound healing; Education Citation: Kruk A, Książek J, Żurek W, et al., Care of a Patient with Fibrous Dysplasia of the Received: November 30, 2015; Accepted: December 12, 2015; Published: December Clavicle and Ribs: Case Report. Health Sci J. 30, 2015 2015, 10:1. Introduction Epidemiological data say that it constitutes 2.5% of tumor-like changes and about 7% of benign tumors [3,4]. Fibrous dysplasia - FD (lat. dysplasia fibrosa – DF) is a rare The World Health Organization defines three forms of the chronic bone disease, characterized by the presence of one or disease: 1) monostotic, which accounts for about 70% of all more areas where healthy bone is replaced by fibrous tissue [1]. cases and affects one bone; 2) polyostotic, when the changes are The cause of this disease is not fully understood. It is currently observed in many bones; and 3) the form in which the disease considered that the reason for its development is a mutation involves craniofacial bones only. McCune-Albright syndrome in the GNAS1 gene, which leads to the production of abnormal is a polyostotic form where, in addition to multifocal changes, G protein responsible for the regulation of bone synthesis. This disorders of the endocrine glands occur (precocious puberty, promotes the development of metabolic bone disorders. Normal hyperthyroidism, hyperpituitarism, hyperadrenocorticism, trabecular bone structure is rebuilt in fibrous tissue, in which, overactive adrenal medulla) and the presence of café au lait as the disease progresses, metaplastic bone structure occurs. spots on the skin, scoliosis [1,4]. The image of the disease may This can cause impaired growth, brittleness, and deformation of be preceded by the appearance of disease symptoms, such as bones affected by disease process. The effects of mutation apply pain, deformations, and bone fractures. However, the diagnosis also to endocrine balance and disrupt the body pigmentation [2]. is based on the result of histopathological and imaging tests. The clinical picture of a generalized form of FD (lat. osteitis fibrosa An additional diagnostic procedure is the determination of the alkaline phosphatase level in serum. generalisata) was first described in 1981 by Recklinghausen. It was only in 1938 when Lichtenstein distinguished fibrous dysplasia of The treatment of choise is surgery if possible, which reduces pain bone as a separate disease entity (lat. dysplasia fibrosa ossium) and improves mobility, and a secondary healing of fractures [5]. [1,2]. The incidence of the disease is not precisely known. The surgery involves the removal of pathological focus with a © Copyright iMedPub | This article is available in: www.hsj.gr/archive 1 ARCHIVOSHealth Science DE MEDICINA Journal 2015 ISSNISSN 1698-94651791-809X Vol. 10 No. 1:12 margin of healthy bone and subsequent tissue transplantation. In plexus at the left side was performed. The image clearly showed some cases a stabilizing action is required with the use of metal compression of the upper lobe of the left lung by pathological implants and bone cement. Conservative treatment consists tissue with low signal at time T1 and T2 dependent. Probably the of regularly taking bisphosphonates, which influence bone changes corresponded to the postoperative scar and partly to remodeling, reduce pain, and slow the pathogenic processes. the main process - FD. Nerve trunks with blurred outlines were Availability of bisphosphonates in Poland is limited because they seen, probably in the course of accompanying fibrosis due to have not been registered for the treatment of FD [6,7]. compressive and postoperative changes. What does this Paper contribute to the wider In 1999, on the basis of radiological documentation from the years global clinical community? 1994 to 1999, an opinion was given, based on which the constant progression of lesions of the dysplasia fibrosa ossium type was The described case shows the need for the introduction of stated. The nature of progression was uneven, i.e. on the right consultation meetings involving specialist doctors, psychologists, side it is clearly slower than on the left side, where currently the and health workers into clinical practice in the care of FD patients. morphological status of the bone corresponds almost exactly to Methods the degree of severity of the changes just before surgery. In 2001, the patient was again qualified for surgery, in this time The interview, observation, analysis of medical records, and case for partial resection of the left clavicle. In anatomopathological study method was used in the study. The place of research is the Department of Thoracic Surgery, University Clinical Center, examination of surgical specimen irregular trabeculae of bone Medical University of Gdansk. The case describes a 51-year-old and intertrabecular spaces filled with the connective tissue man diagnosed with fibrous dysplasia of the clavicle and ribs, in larger spaces in the purulent inflammation of the clavicle hospitalized in the years 1995-2013. The report is additionally fragment were seen. There were no characteristics of fibrous supplemented with figures of the wound healing process in years dysplasia of bone. 2012-2014. In 2004, resection of a fragment of the sternum, part of clavicle, and The aim of this case report is to analyze interdisciplinary care of first and second rib on the left was performed, and subsequently a patient chronically treated for fibrous dysplasia of the clavicle the patient was qualified for chest radiation in a single fraction and ribs . 6Gy. Following treatment the patient was discharged home. In 2006, the patient came to the Department of Thoracic Surgery Case Report because of severe pain. It was caused by compression of the A 51-year-old male, heavy smoker (30 pack years, without hypertrophic clavicle and first rib on the brachial plexus on the willingness to quit the habit) was treated for bone fibrous dysplasia right side. During qualification for the surgery, the patient was since 1994. The first symptoms appeared at the age of 30. The found to be a carrier of hepatitis C. Right clavicle resection and patient came to the physician because of severe pain in the chest. partial resection of the first rib were done. The postoperative In 1995, based on computed tomography scan (CT) of the chest, course was uneventful. The patient was discharged home with features of thickening of the proximal parts of clavicles and ribs I and II on the left side in the parasternal part with characteristics of fibrous-like intraosseous hypertrophy and sclerosis. In addition to intraosseous changes, thickening of the adjacent soft tissues was observed. In the CT image bone remodeling characteristic for fibrous dysplasia of the clavicle and ribs was observed. Initially, the patient was qualified for the partial resection of the left clavicle. After three months thoracotomy was done with resection of parasternal fragments of the second and the third rib on the left. On the anatomopathological examination, the image of FD was confirmed. Results of laboratory tests showed normal serum levels of inorganic phosphorus, total calcium, and alkaline phosphatase. In 1996 the case was consulted by representatives of the Polish Osteoporosis Foundation (Polska Fundacja Osteoporozy, PFO). The PFO advised that due to the lack of established methods for the treatment of FD, pharmacological treatment is recommended. It was stated that the use of calcitonin, a known stimulant of somatostatin should have a positive effect on bone morphological image. According to the recommendations, treatment with Miacalcic Nasal Spray, a 100 I.U. in a scheme of two times a day Figure 1 3D CT reconstruction of bony elements of the chest for a month, then a month break, was used for the next three after resection of upper ribs and upper part of the years. Despite therapy, the man continued to suffer pain. sternum. Source: Own material provided by the patient. In 1998, an Magnetic Resonance Imaging (MRI) of the brachial 2 This article is available in:www.hsj.gr/archive ARCHIVOSHealth Science DE MEDICINA Journal 2015 ISSNISSN 1698-94651791-809X Vol. 10 No. 1:12 instructions for urgent control in the Pain Clinic to reduce the the hospital with purulent fistula at the upper part of the sternum, doses of opioid drugs.
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