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Medicine by Alexandros G.Sfakianakis,Anapafseos 5 Agios Nikolao

Medicine by Alexandros G.Sfakianakis

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Τρίτη, 3 Ιανουαρίου 2017

Calvarial aneurysmal bone cyst associated with fibrous dysplasia







Highlights

Aneurysmal bone cysts are rare, benign lesions.
Calvarial aneurysmal bone cysts may present as a cosmetic defect and/or with neurologic symptoms.
A minority of fibrous dysplasia cases are due to McCune-Albright Syndrome.
Large aneurysmal bone cysts treated with cranioplasty may require complex skin excision.
Patients may benefit greatly from in situ cranioplasty with methyl methacrylate and molded titanium mesh.

Abstract

Aneurysmal bone cysts are benign, expansile bone lesions primary or secondary to other pathology. The majority are seen in long bones and the spine, rarely occurring within the cranial vault. Here we describe the case of a 17-year-old gentleman with McCune-Albright syndrome who developed a right parietal aneurysmal bone cyst in the setting of fibrous dysplasia. The patient was treated with lesion excision and in situ cranioplasty using methyl methacrylate and molded titanium mesh, ultimately rendering excellent cosmetic outcome. Our case report highlights the efficacy of in situ titanium cranioplasties in contouring to the native skull and enabling optimization of cosmesis, specifically through the use of titanium mesh plates with the bone flap as a template.

Keywords

  • Bone cyst
  • Calvarium
  • McCune-Albright syndrome
  • In situ cranioplasty
  • Cosmesis

1. Introduction

Aneurysmal bone cysts are benign lesions comprising less than 2% of bone tumors [1]. Fewer than 5% of these lesions occur in the calvarium, and very few have been described[2][3][4][5] and [6]. Herein we present the novel case of a patient with McCune-Albright syndrome and known fibrous dysplasia of the parietal bone that secondarily developed a large aneurysmal bone cyst. The lesion was treated with surgical excision and in situ cranioplasty with methyl methacrylate and molded titanium mesh. We review the pathogenesis of aneurysmal bone cysts as well as management and treatment strategies. Of note, we highlight the robust use of this unique in situ cranioplasty technique in achieving excellent skull cosmesis.

2. Case report

2.1. Patient description

A 17-year-old gentleman with McCune-Albright syndrome was found to have a right-sided skull abnormality. CT scan revealed a right parietal bone lesion without brain parenchyma compression (Fig. 1). The patient was asymptomatic and the lesion managed through observation. Three-and-a-half years later, he reported increasing difficulty with spatial awareness and concentration capacity over several months. Imaging revealed cystic expansion of the lesion to 7 cm × 6 cm × 6 cm with considerable compression of the underlying brain parenchyma and a resultant 6 mm midline shift (Fig. 2).
Fig. 1
Fig. 1. 
A right parietal bone lesion is seen on axial CT scan (left panel) and T2-weighted axial MRI (right panel), without underlying brain compression.
Full-size image (51 K)
Fig. 2. 
A large aneurysmal bone cyst is demonstrated on axial non-contrast CT scan (left panel) and T2-weighted axial MRI (middle panel) with compression of the parietal lobe and significant midline shift. CT bone window shows aneurysmal bone cyst is located within area of fibrous dysplasia (right panel).

2.2. Surgical procedure and postoperative outcome

Following a U-shaped right temporal-frontal-parietal-occipital skin incision and subsequent scalp flap retraction, an exophytic bone cyst was visualized with soft tissue erosion through cortical surface of the bone. A drill was used to excise the lesion, and the dura was visible and pulsating. Next, titanium mesh was molded to the presumed shape of the right parietal region and screwed into the bone, and the epidural space dissected. Abnormal dura was then opened in a circumferential manner and excised, and samples of abnormal bone and soft cystic tissue sent for pathologic examination. The dural defect was grafted with two pieces of SurgAssist and augmented with fibrin glue.
Molded titanium mesh was reattached to the surrounding craniotomy bone flap and methyl methacrylate was laid into the inner side of the mesh and flattened to approximate the width or the normal bone. Once hardened, in situ cranioplasty was performed. This technique has been described as enabling additional mesh cranioplasty strength after cranial defect repair [2]. The wound was irrigated and vancomycin powder placed over the cranioplasty. The galea was then closed with interrupted 3-0 Vicryl and running 2-0 Vicryl, and skin with running 4-0 Novofil suture. A Hemovac drain was placed in the subgaleal space, and a head dressing applied.
Histopathologic evaluation revealed aneurysmal bone cyst with traces of fibrous dysplasia. Post-operative imaging showed resolution of the lesion (Fig. 3). Cosmesis was deemed excellent by both the patient and family members.
Fig. 3
Fig. 3. 
The in situ cranioplasty is demonstrated on lateral x-ray (left panel) and axial non-contrast CT scan (right panel).

3. Discussion

Calvarial aneurysmal bone cysts may present as a cosmetic defect or with neurologic symptoms. Primary aneurysmal bone cysts arise from genetic rearrangements in the oncogene TRE17/USP6 [3], and may result from subsequent upregulation of matrix metalloproteinases via NF-κB [4] and [5]. The pathogenesis of secondary aneurysmal bone cysts is not well established, though an insult resulting in increased venous pressure has long been posited as a potential mechanism [6]. A minority of cases of fibrous dysplasia are due to McCune-Albright syndrome, caused by activating mutations in GNAS, polyostotic fibrous dysplasia co-occurs with café-au-lait spots and endocrinopathies including precocious puberty [7][8][9][10] and [11].
To ensure holistic treatment, a variety of medical modalities should be considered in the management of aneurysmal bone cysts. Although for cranial lesions the mainstay of treatment is surgical excision, pre-operative or stand-alone embolization has been used with success as well [12]. Radiotherapy has been used in the management of both primary and recurrent lesions, including a recurrent temporal lesion [13] and [14]. In aneurysmal bone cysts of the spine and pelvis deemed unresectable, bisphosphonates and denosumab have been used with some efficacy [15] and [16]. Finally, sclerotherapy has also been an effective management strategy [17].
It is important to note that large aneurysmal bone cysts treated with cranioplasty may require complex skin excision, as was the case with our patient who benefitted greatly from in situ cranioplasty with methyl methacrylate and molded titanium mesh. Major objectives of cranioplasty include wound healing, protection of cranial vault structures, as well as achieving cosmetic satisfaction. In situ cranioplasty for aneurysmal bone cysts avoids advanced preparation of custom prostheses often required in other cranioplasty techniques. Furthermore, it curtails the technical problems of bone graft cranioplasties such as harvest site disfigurement when autologous bone is taken from a donor site. As compared to the split calvarial approach, in situ cranioplasty avoids added morbidity to harvest autologous bone from donor skull. Lastly, it has been documented that in situ cranioplasty costs are reasonable and approximately 2.5 times less expensive than osteo-inductive materials such as hydroxyapatite [18]. As shown by our case report, use of this in situ cranioplasty technique can also result in superb cosmesis.

Sources of support

None relevant for disclosure.

Conflicts of interest/disclosures

The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.

References

    • [4]
    • L.M. Pringle, et al.
    • Atypical mechanism of NF-kappaB activation by TRE17/ubiquitin-specific protease 6 (USP6) oncogene and its requirement in tumorigenesis
    • Oncogene, 31 (30) (2012), pp. 3525–3535
    •  |   | 
    • [7]
    • C.E. Dumitrescu, M.T. Collins
    • McCune-Albright syndrome
    • Orphanet. J. Rare Dis., 3 (2008), p. 12
    • [8]
    • D. McCune
    • Osteitis fibrosa cystica; the case of a nine year old girl who also exhibits precocious puberty, multiple pigmentation of the skin and hyperthyroidism
    • Am. J. Dis. Child, 52 (743) (1936), p. 4
    • [9]
    • F. Albright, et al.
    • Syndrome characterized by osteitis fibrosa disseminata, areas of pigmentation and endocrine dysfunction, with precocious puberty in females: report of five cases
    • N. Engl. J. Med., 216 (17) (1937), pp. 727–746
    •  | 
    • [11]
    • A.M. Boyce, M.T. Collins
    • Fibrous Dysplasia/McCune-Albright Syndrome
    • R.A. Pagon (Ed.), et al., GeneReviews(R), University of Washington, Seattle: Seattle (WA) (1993)
    • [12]
    • B.Y. Sheikh
    • Cranial aneurysmal bone cyst “with special emphasis on endovascular management”
    • Acta Neurochir., 141 (6) (1999), pp. 601–610 (discussion 610-1)
Case Report
Corresponding author at: 505 Parnassus Ave, San Francisco, CA 94143-0112, USA.

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Tinnitus

1. Chadha NK, Gordon KA, James AL, Papsin BC. Tinnitus is prevalent in children with cochlear implants. International Journal of Pediatric Otorhinolaryngology. 2009;73:671-675. [abstract]

2. Akdogan O, Ozcan I, Ozbek C, Dere H. Tinnitus after cochlear implantation. Auris Nasus Larynx. 2009;36:210-212. [abstract]

3. Pan T, Tyler RS, Ji H, Coelho C, Gehringer AK, Gogel SA. Changes in the tinnitus handicap questionnaire after cochlear implantation. American Journal of Audiology. 2009;18:144-151. [abstract]

4. Andersson G, Freijd A, Baguley DM, Idrizbegovic E. Tinnitus distress, anxiety, depression, and hearing problems among cochlear implant patients with tinnitus. Journal of the American Academy of Audiology. 2009;20:315-319. [abstract]

5. Rothholtz VS, Tang Q, Wu EC, Fine EL, Djalilian H, Zeng F-G. Exploring the parametric space of tinnitus suppression in a patient with a cochlear implant. Laryngoscope. 2009;119.

6. Di NW, Cianfrone F, Scorpecci A, Cantore I, Giannantonio S, Paludetti G. Transtympanic electrical stimulation for immediate and long-term tinnitus suppression. International Tinnitus Journal. 2009;15:100-106.[abstract]

7. Litre CF, Theret E, Tran H et al. Surgical treatment by electrical stimulation of the auditory cortex for intractable tinnitus. Brain Stimulation. 2009;2:132-137. [abstract]

8. Evans RW, Ishiyama G. Migraine with transient unilateral hearing loss and tinnitus. Headache: The Journal of Head & Face Pain. 2009;49:756-759. [abstract]

9. Pirodda A, Brandolini C, Raimondi MC, Ferri GG, Borghi C. Tinnitus as a warning for preventing vasovagal syncope. Medical Hypotheses. 2009;73:370-371. [abstract]

10. Anderson JE, Teitel D, Wu YW. Venous hum causing tinnitus: case report and review of the literature. Clinical Pediatrics. 2009;48:87-89. [abstract]

11. Liess BD, Lollar KW, Christiansen SG, Vaslow D. Pulsatile tinnitus: a harbinger of a greater ill? Head & Neck. 2009;31:269-273. [abstract]

12. Singh DP, Forte AJ, Brewer MB, Nowygrod R. Bilateral carotid endarterectomy as treatment of vascular pulsatile tinnitus. Journal of Vascular Surgery. 2009;50:183-185. [abstract]

13. Delgado F, Munoz F, Bravo-Rodriguez F, Jurado-Ramos A, Oteros R. Treatment of dural arteriovenous fistulas presenting as pulsatile tinnitus. Otology and Neurotology. 2009;30:897-902. [abstract]

14. Cowley PO, Jones R, Tuch P, McAuliffe W. Pulsatile tinnitus from reversal of flow in an aberrant occipital artery: Resolved after carotid artery stenting. American Journal of Neuroradiology. 2009;30:995-997. [abstract]

15. Stimmer H, Borrmann A, Loer C, Arnold W, Rummeny EJ. Monaural tinnitus from a contralateral inferior colliculus hemorrhage. Audiology & Neurotology. 2009;14:35-38. [abstract]

16. Latifpour DH, Grenner J, Sjodahl C. The effect of a new treatment based on somatosensory stimulation in a group of patients with somatically related tinnitus. International Tinnitus Journal. 2009;15:94-99. [abstract]

17. Department of Health. Provision of services for adults with tinnitus: a good practice guide. 2009. [full text]

18. DH. Tinnitus Map of Medicine care pathway. 2010. [Full text]

19. BTA. Tinnitus: guidelines for primary care. 2010. [Full text]

20. Schneider P, Andermann M, Wengenroth M et al. Reduced volume of Heschl's gyrus in tinnitus. NeuroImage. 2009;45:927-939. [abstract]

21. Landgrebe M, Langguth B, Rosengarth K et al. Structural brain changes in tinnitus: grey matter decrease in auditory and non-auditory brain areas. NeuroImage. 2009;46:213-218. [abstract]

22. Melcher JR, Levine RA, Bergevin C, Norris B. The auditory midbrain of people with tinnitus: Abnormal sound-evoked activity revisited. Hearing Research. 2009;257:63-74. [abstract]

23. Lanting CP, de KE, van DP. Neural activity underlying tinnitus generation: Results from PET and fMRI. Hearing Research. 2009;255:1-13. [abstract]

24. Kaltenbach JA. Insights on the origins of tinnitus: an overview of recent research. Hearing Journal. 2009;62:26-31. [Full text]

25. Shulman A, Goldstein B, Strashun AM. Final common pathway for tinnitus: theoretical and clinical implications of neuroanatomical substrates. International Tinnitus Journal. 2009;15:5-50. [abstract]

26. Schutte NS, Noble W, Malouff JM, Bhullar N. Evaluation of a model of distress related to tinnitus. International Journal of Audiology. 2009;48:428-432. [abstract]

27. Hesser H, Pereswetoff-Morath CE, Andersson G. Consequences of controlling background sounds: the effect of experiential avoidance on tinnitus interference. Rehabilitation Psychology. 2009;54:381-390.[abstract]

28. Argstatter H, Krick C, Bolay HV. Music therapy for chronic tinnitus. Heidelberg treatment model. Psychotherapeut. 2009;54:17-26. [abstract]

29. Lugli M, Romani R, Ponzi S, Bacciu S, Parmigiani S. The windowed sound therapy: A new empirical approach for an effective personalized treatment of tinnitus. International Tinnitus Journal. 2009;15:51-61.[abstract]

30. Langguth B, Salvi R, Elgoyhen AB. Emerging pharmacotherapy of tinnitus. Expert Opinion on Emerging Drugs. 2009;14:687-702. [abstract]

31. Campbell KCM. Emerging pharmacologic treatments for hearing loss and tinnitus. ASHA Leader. 2009;14:14-18. [Full text]

32. Hesser H, Westin V, Hayes SC, Andersson G. Clients' in-session acceptance and cognitive defusion behaviors in acceptance-based treatment of tinnitus distress. Behaviour Research & Therapy. 2009;47:523-528. [abstract]

33. Hesser H, Andersson G. The role of anxiety sensitivity and behavioral avoidance in tinnitus disability. International Journal of Audiology. 2009;48:295-299. [abstract]

34. Shulman A, Goldstein B. Subjective idiopathic tinnitus and palliative care: a plan for diagnosis and treatment. Otolaryngologic Clinics of North America. 2009;42:15-38. [abstract]

35. Forti S, Costanzo S, Crocetti A, Pignataro L, Del BL, Ambrosetti U. Are results of tinnitus retraining therapy maintained over time? 18-month follow-up after completion of therapy. Audiology & Neuro-Otology. 2009;14:286-289. [abstract]

36. Bessman P, Heider T, Watten VP, Watten RG. The tinnitus intensive therapy habituation program: a 2-year follow-up pilot study on subjective tinnitus. Rehabilitation Psychology. 2009;54:133-138. [abstract]

37. Gudex C, Skellgaard PH, West T, Sorensen J. Effectiveness of a tinnitus management programme: A 2-year follow-up study. BMC Ear, Nose and Throat Disorders. 2009;9. [Full text]

38. Henry J, Zaugg T, Myers P, Kendall C, Turbin M. Principles and application of educational counseling used in progressive audiologic tinnitus management. Noise and Health. 2009;11:33-48. [abstract]

1. Hazell JW, Jastreboff PJ. Tinnitus. I: Auditory mechanisms: a model for tinnitus and hearing impairment. J Otolaryngol. 1990;19:1-5. [Abstract]

2. Jastreboff PJ, Jastreboff MM. Tinnitus Retraining Therapy (TRT) as a method for treatment of tinnitus and hyperacusis patients. J Am Acad Audiol. 2000 Mar;11(3):162-77. [Abstract]

3. Marcondes RA, Sanchez TG, Kii MA, Langguth et al. Repetitive transcranial magnetic stimulation improve tinnitus in normal hearing patients: a double-blind controlled, clinical and neuroimaging outcome study. Eur J Neurol. 2009. [Epub ahead of print] ) [Abstract]

4. Cannon SC Pathomechanisms in channelopathies of skeletal muscle and brain. Annu Rev Neurosci. 2006;29:387-415. [Abstract]

5. Davies E, Knox E, Donaldson I. The usefulness of nimodipine, an L-calcium channel antagonist, in the treatment of tinnitus. Br J Audiol. 1994;28:125-129. [Abstract]

6. Baguley DM, Jones S, Wilkins I, Axon PR, Moffat DA. The inhibitory effect of intravenous lidocaine infusion on tinnitus after translabyrinthine removal of vestibular schwannoma: a double-blind, placebo-controlled, crossover study. Otol Neurotol. 2005;26:169-176. [Abstract]

Eggermont JJ. Cortical tonotopic map reorganization and its implications for treatment of tinnitus. Acta Otolaryngol Suppl. 2006;9-12. [Abstract]

Hoke ES, Muhlnickel W, Ross B, Hoke M. Tinnitus and event-related activity of the auditory cortex. Audiol Neurootol. 1998;3:300-331. [Abstract]

Mirz F, Pedersen B, Ishizu K et al. Positron emission tomography of cortical centers of tinnitus. Hear Res. 1999;134:133-144. [Abstract]

Muhlnickel W, Elbert T, Taub E, Flor H. Reorganization of auditory cortex in tinnitus. Proc Natl Acad Sci U S A. 1998;95:10340-10343. [Abstract]

Norena AJ, Eggermont JJ. Enriched acoustic environment after noise trauma abolishes neural signs of tinnitus. Neuroreport. 2006;17:559-563. [Abstract]

Schlee W, Hartmann T, Langguth B, Weisz N. Abnormal resting-state cortical coupling in chronic tinnitus. BMC Neurosci. 2009;10:11. [Full text]

Schlee W, Mueller N, Hartmann T, Keil J, Lorenz I, Weisz N. Mapping cortical hubs in tinnitus. BMC Biol. 2009;7:80. [Full text]

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HIPPOCRATE'S OATH

"I swear by Apollo, the healer, Asclepius, Hygieia, and Panacea, and I take to witness all the gods, all the goddesses, to keep according to my ability and my judgment, the following Oath and agreement:

To consider dear to me, as my parents, him who taught me this art; to live in common with him and, if necessary, to share my goods with him; To look upon his children as my own brothers, to teach them this art.

I will prescribe regimens for the good of my patients according to my ability and my judgment and never do harm to anyone.

I will not give a lethal drug to anyone if I am asked, nor will I advise such a plan; and similarly I will not give a woman a pessaryto cause an abortion.

But I will preserve the purity of my life and my arts.

I will not cut for stone, even for patients in whom the disease is manifest; I will leave this operation to be performed by practitioners, specialists in this art.

In every house where I come I will enter only for the good of my patients, keeping myself far from all intentional ill-doing and all seduction and especially from the pleasures of love with women or with men, be they free or slaves.

All that may come to my knowledge in the exercise of my profession or in daily commerce with men, which ought not to be spread abroad, I will keep secret and will never reveal.

If I keep this oath faithfully, may I enjoy my life and practice my art, respected by all men and in all times; but if I swerve from it or violate it, may the reverse be my lot."

MAIMONIDE'S PRAYER

"Almighty God, Thou has created the human body with infinite wisdom. Ten thousand times ten thousand organs hast Thou combined in it that act unceasingly and harmoniously to preserve the whole in all its beauty the body which is the envelope of the immortal soul. They are ever acting in perfect order, agreement and accord. Yet, when the frailty of matter or the unbridling of passions deranges this order or interrupts this accord, then forces clash and the body crumbles into the primal dust from which it came. Thou sendest to man diseases as beneficent messengers to foretell approaching danger and to urge him to avert it.

"Thou has blest Thine earth, Thy rivers and Thy mountains with healing substances; they enable Thy creatures to alleviate their sufferings and to heal their illnesses. Thou hast endowed man with the wisdom to relieve the suffering of his brother, to recognize his disorders, to extract the healing substances, to discover their powers and to prepare and to apply them to suit every ill. In Thine Eternal Providence Thou hast chosen me to watch over the life and health of Thy creatures. I am now about to apply myself to the duties of my profession. Support me, Almighty God, in these great labors that they may benefit mankind, for without Thy help not even the least thing will succeed.

"Inspire me with love for my art and for Thy creatures. Do not allow thirst for profit, ambition for renown and admiration, to interfere with my profession, for these are the enemies of truth and of love for mankind and they can lead astray in the great task of attending to the welfare of Thy creatures. Preserve the strength of my body and of my soul that they ever be ready to cheerfully help and support rich and poor, good and bad, enemy as well as friend. In the sufferer let me see only the human being. Illumine my mind that it recognize what presents itself and that it may comprehend what is absent or hidden. Let it not fail to see what is visible, but do not permit it to arrogate to itself the power to see what cannot be seen, for delicate and indefinite are the bounds of the great art of caring for the lives and health of Thy creatures. Let me never be absent- minded. May no strange thoughts divert my attention at the bedside of the sick, or disturb my mind in its silent labors, for great and sacred are the thoughtful deliberations required to preserve the lives and health of Thy creatures.

"Grant that my patients have confidence in me and my art and follow my directions and my counsel. Remove from their midst all charlatans and the whole host of officious relatives and know-all nurses, cruel people who arrogantly frustrate the wisest purposes of our art and often lead Thy creatures to their death.

"Should those who are wiser than I wish to improve and instruct me, let my soul gratefully follow their guidance; for vast is the extent of our art. Should conceited fools, however, censure me, then let love for my profession steel me against them, so that I remain steadfast without regard for age, for reputation, or for honor, because surrender would bring to Thy creatures sickness and death.

"Imbue my soul with gentleness and calmness when older colleagues, proud of their age, wish to displace me or to scorn me or disdainfully to teach me. May even this be of advantage to me, for they know many things of which I am ignorant, but let not their arrogance give me pain. For they are old and old age is not master of the passions. I also hope to attain old age upon this earth, before Thee, Almighty God!

"Let me be contented in everything except in the great science of my profession. Never allow the thought to arise in me that I have attained to sufficient knowledge, but vouchsafe to me the strength, the leisure and the ambition ever to extend my knowledge. For art is great, but the mind of man is ever expanding.

"Almighty God! Thou hast chosen me in Thy mercy to watch over the life and death of Thy creatures. I now apply myself to my profession. Support me in this great task so that it may benefit mankind, for without Thy help not even the least thing will succeed."

Information for Health Professionals

Information for Patients

Modern challenged parts of the oath:

  1. To teach medicine to the sons of my teacher. In the past, medical schools gave preferential consideration to the children of physicians.
  2. To practice and prescribe to the best of my ability for the good of my patients, and to try to avoid harming them. This beneficial intention is the purpose of the physician. However, this item is still invoked in the modern discussions of euthanasia.
  3. I will not give a lethal drug to anyone if I am asked, nor will I advise such a plan. Physician organizations in most countries have strongly denounced physician participation in legal executions. However, in a small number of cases, most notably the U.S. states of Oregon,[10] Washington,[11]Montana,[12] and in the Kingdom of the Netherlands,[13] a doctor can prescribe euthanasia with the patient's consent.
  4. Similarly, I will not give a woman a pessary to cause an abortion. Since the legalization of abortion in many countries, the inclusion of the anti-abortion sentence of the Hippocratic oath has been a source of contention.
  5. To avoid violating the morals of my community. Many licensing agencies will revoke a physician's license for offending the morals of the community ("moral turpitude").
  6. I will not cut for stone, even for patients in whom the disease is manifest; I will leave this operation to be performed by practitioners, specialists in this art. The "stones" referred to are kidney stones or bladder stones, removal of which was judged too menial for physicians, and therefore was left for barbers (the forerunners of modern surgeons). Surgery was not recognized as a specialty at that time. This sentence is now interpreted as acknowledging that it is impossible for any single physician to maintain expertise in all areas. It also highlights the different historical origins of the surgeon and the physician.
  7. To keep the good of the patient as the highest priority. There may be other conflicting 'good purposes,' such as community welfare, conserving economic resources, supporting the criminal justice system, or simply making money for the physician or his employer that provide recurring challenges to physicians
http://www.worldallergy.org/educational_programs/world_allergy_forum/barcelona2008/rabe/

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