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Contents Vol. 4, No. 10 (October 2013)

Cover Image 554 Polyarticular tophaceous gouty arthritis: A case report Figure 1: Swelling of dorsum of right hand and Sheikh Javeed Ahmad, Sumyra Khurshid wrist joint with cellulitic overlying skin. Computed tomography scan of wrist and carpal bones 559 Pulmonary artery dilatation in a young man showing extensive multiple erosions of carpal presenting with a left mandibular fracture: bones and adjacent radial styloid. Do not forget cocaine Kohmal Ashok Solanki, Zhan Yun Lim, Andrea Pisesky, John Hogan Case Series 563 Mid-aortic dysplastic syndrome as a rare cause of hypertension in young Cover Figure: 527 Chondroid lipoma, a rare lipomatous tumor: A Kaushik Saha, Dipa Saha, Parinita Ranjit, case series Sujoy Sarkar, Rabi Ranjan Sow Mondal, Mohamed A Shawarby, Maissa N El-Maraghy, Thiyagrajan G Ragaa A Salem, Nafissa M El-Badawy, Tarek M El-Sharkawy, Tarek M Hashem, 567 An unusual case of a misplaced left internal Ahmad S Kamel jugular vein catheter Sunil Rangarajan, Sunad Rangarajan, Lindsey Smith Hinton Case Report 571 Retroperitoneal neurilemmoma with cystic 532 An unusual hip dislocation during tennis playing degeneration mimicking hydatid cyst Ismail Turkmen, Fatih Turkmensoy, Korhan Manash Ranjan Sahoo, Anil Kumar T Ozkan, Salih Soylemez, Feyza Unlu Ozkan, 575 Frank hematuria as sole manifestation of Yalcin Turhan acute myeloid leukemia: A case report 536 A variant common hepatic artery originating ML Patel, Rekha Sachan, Apul Goel from the normal celiac trunk and passing 578 Spontaneous regression of lumbar disc behind the portal vein successfully treated with herniation: Conservative treatment in a case pancreaticoduodenectomy for middle bile duct with motor deficit cancer: A case Report Saliha Eroğlu Demir, Nihal Özaras, Ebru Yoshihito Ohta, Michiki Narushima, Adoru Aytekin Okaue, Hisashi Nakata, Hisahiro Matsubara

541 Isolated tuberculosis of the wrist: A rare case of Case in Images extrapulmonary tuberculosis Mohamed Altayeb Mussa, Edmund Fitzgerald O’Connor, Stuart Waterston, Michael Taylor, 582 Treatment with closed manipulation and Fortune Iwuagwu functional bracing of a humeral refracture with implant failure: A case report 546 Thoracic epidural anesthesia for modified Ali Ersen, Atakan Guvendiren, Ozgur Yazici radical mastectomy in carcinoma of breast patient with chronic obstructive pulmonary disease: A case report Clinical Images Balaji Narayan Asegaonkar, Sujata Rahul Zine, Unmesh Vidyadhar Takalkar, Umesh Kulkarni, Shilpa Balaji Asegaonkar, Pushpa Kodlikeri 586 Otogenic pneumocephalus secondary to All Articles: recurrent cholesteatoma and associated 551 ‘Wallpaper paste sign’ of mucinous breast temporal bone cerebrospinal fluid leak carcinoma Johannes Christiaan Oosthuizen, Fintan Muhammad Asad Parvaiz, Brian Isgar, Nedra Wallis, John Fenton Aluwihare

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CASE series OPEN ACCESS Chondroid lipoma, a rare lipomatous tumor: A case series

Mohamed A Shawarby, Maissa N El-Maraghy, Ragaa A Salem, Nafissa M El-Badawy, Tarek M El-Sharkawy, Tarek M Hashem, Ahmad S Kamel

Abstract for CD68. The Ki67 proliferative index was less than 5%. The common differentials like Introduction: Chondroid lipoma is a rare, myxoid liposarcoma and extraskeletal myxoid benign lipomatous tumor that may be were ruled out based mainly mistaken histologically for a liposarcoma or on pure histologic criteria. Both the patients chondrosarcoma. Herein, two cases of chondroid underwent simple curative excision and tumors lipoma are reported in two Egyptian females were free for at least one and three years after aged 38 and 51 years and literature about this , respectively. Conclusion: A high level rare tumor briefly reviewed with discussion of of suspicion by the pathologist and familiarity diagnostic criteria that may help distinguishing with its features are of practical importance to it from other tumors with overlapping avoid misdiagnosis and overtreatment because histologic features. Case Series: The tumor was chondroid lipoma is easily misdiagnosed as a subcutaneous in both the cases. One was located of either adipose tissue or . in the right knee area and the other in an infra- mammary location on the right side of the chest. Keywords: Chondroid, Myxoid, Liposarcoma, Grossly, both neoplasms were encapsulated Chondrosarcoma, Lipoma and exhibited a nodular, myxoid cut surface. Histologically, they consisted of an abundant ********* myxoid and chondroid stroma interspersed by small round cells with eosinophilic or vacuolated Shawarby MA, El-Maraghy MN, Salem RA, El-Badawy cytoplasm, signet ring lipoblast-like cells, NM, El-Sharkawy TM, Hashem TM, Kamel AS. adipocytes and foci of mature adipose tissue. Chondroid lipoma, a rare lipomatous tumor: A case Immunohistochemically, diffuse reactivity of the series. International Journal of Case Reports and Images neoplastic cells for vimentin and variable reactivity 2013;4(10):527–531. for S-100 protein were present in both the cases. One case also showed focal immunoreactivity *********

doi:10.5348/ijcri-2013-10-372-CS-1 Mohamed A Shawarby1, Maissa N El-Maraghy2, Ragaa A Salem2, Nafissa M El-Badawy2, Tarek M El-Sharkawy3, Tarek M Hashem3, Ahmad S Kamel3 Affiliations: 1College of , University of Dammam, Dammam, Saudi Arabia; 2College of Medicine, Ain-Shams University, Cairo, EGYPT; 3College of Medicine, University Introduction of Dammam, Dammam, Saudi Arabia. Corresponding Author: Mohamed Shawarby, PhD, P.O. Box Chondroid lipoma is a rare, benign lipomatous tumor 2208, Al-Khobar 31952, Saudi Arabia; Tel: +966 3 8966666, that may be mistaken histologically for a sarcoma of either Ext 3111; Mob: 0966508180807; Fax: +966 3 8966807; adipose tissue or cartilage [1]. A high level of suspicion Email: [email protected] by the pathologist and familiarity with its features are of practical importance to avoid an overtreatment as the Received: 24 April 2013 tumor does not recur or metastasize, and simple excision Accepted: 21 June 2013 is curative because it is easily misdiagnosed as a malignant Published: 01 October 2013 tumor [2]. Herein, we report two cases of chondroid lipoma followed by a brief literature review about this

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):527–531. Shawarby et al. 528 www.ijcasereportsandimages.com rare tumor with discussion of diagnostic criteria that may myxoid liposarcoma and the presence of a chondroid help distinguish it from other tumors with overlapping matrix and mature adipose tissue. After three years of histologic features. surgery, the patient was tumor free. Case 2: A 51-year-old Egyptian female presented with a painless subcutaneous inframammary mass on the right CASE SERIES side of the chest. Past medical history was unremarkable. The mass was well defined and non-tender, and the Case 1: A 38-year-old Egyptian female presented overlying skin was normal. It was excised and submitted with a painless subcutaneous swelling in the right knee for pathological study. area. Her past medical history was unremarkable. On examination, the swelling was well defined and non- tender. The overlying skin was normal. Excision was done and the specimen submitted for pathological examination. Gross: An encapsulated, well circumscribed tumor received fragmented as two grayish-white pieces measuring 12x8x4 cm and 8x5x4 cm. The cut surface was nodular with myxoid areas (Figure 1). Microscopic: Sections revealed an encapsulated, lobulated tumor of varying cellularity, composed of strands, clusters and sheets of small round cells with eosinophilic or vacuolated cytoplasm in an abundant chondroid and myxoid stroma interspersed by scattered adipocytes and foci of mature adipose tissue (Figure 2). Glycogen could be demonstrated in the cytoplasm of rare cells. There was no significant nuclear pleomorphism and only very occasional mitoses were seen. Immunohistochemically, there was strong diffuse cytoplasmic reactivity for vimentin. Most cells also Figure 2: Case 1. (A) Note capsule and abundant myxochondroid showed strong cytoplasmic and nuclear reactivity for stroma (H&E stain, x100), (B) Note cells with eosinophilic or S-100 protein. A few scattered cells showed positive vacuolated cytoplasm (H&E stain, x200), (C) Note adipocytes cytoplasmic staining for CD68. Positive nuclear staining and intracytoplasmic glycogen (PAS stain, x200), (D) Note for Ki67 was noted in only less than 5% of cells (Figure 3). mature adipose tissue underneath tumor capsule (PAS stain, There was no reactivity for CK, EMA, SMA or HMB45. x100). The initial impression was myxoid liposarcoma. However, the final diagnosis after intradepartmental consultation was chondroid lipoma, mainly based on the lack of the typical ‘chicken wire’ vasculature seen in

Figure 3: Case 1. Immunoreactivity for: (A) Vimentin. Note diffuse cytoplasmic positivity of neoplastic cells (IHC, x100), (B) S-100 protein. Note cytoplasmic and nuclear reactivity of neoplastic cells (IHC, x200), (C) CD68. Note cytoplasmic reactivity of scattered neoplastic cells (IHC, x200), (D) Ki67. Figure 1: Case 1. Gross appearance. Note nodular, myxoid cut Note low proliferative index with nuclear reactivity of only rare surface. neoplastic cells (IHC, x100).

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):527–531. Shawarby et al. 529 www.ijcasereportsandimages.com

Gross: An encapsulated, well circumscribed grayish- tan mass with a myxoid, nodular cut-surface, measuring 8x6x5 cm (Figure 4). Microscopic: Sections revealed a distinctly lobulated tumor with a thin capsule, composed of an abundant myxoid to chondroid stroma interspersed by small round cells with eosinophilic cytoplasm, mainly located at the periphery of the lobules along with scattered signet ring lipoblast-like cells and more mature adipocytes. Adipocytes were also focally seen around blood vessels and occasionally formed islands within the tumor lobules (Figure 5). There was no significant nuclear pleomorphism and mitoses could not be demonstrated. Immunohistochemically, there was strong diffuse cytoplasmic reactivity for vimentin. Occasionally, cells also showed nuclear reactivity for S-100 protein and Ki67. There was no reactivity for CD68, CK, EMA, SMA or HMB45. Figure 5: Case 2. (A) Note lobulation and myxoid stroma (H&E stain, x50), (B) Note cells with eosinophilic cytoplasm in periphery of lobule as well as signet ring lipoblast-like cells and rare mature adipocytes (H&E stain, x100), (C) Note mature adipose tissue around blood vessel (H&E stain, x50), (D) Note island of mature adipose tissue in lower left corner (H&E stain, x50).

The vast majority of chondroid lipomas occur in females and are seen between ages of 14 and 70 years (median of 36 years) [1]. Both of our patients were female and the mean age was 44.5 years. They are usually located in the subcutaneous tissue, superficial muscular fascia, or skeletal muscles, most frequently in the proximal extremity and limb girdle, then in decreasing frequency in the leg, trunk, head and neck region, foot, and hand [4, 5]. The tumor mass in both of our cases was subcutaneous Figure 4: Case 2. Gross appearance. Note nodular, myxoid cut with one located in the knee area and the other in the surface. trunk in a infra-mammary location. Chondroid lipomas are well-circumscribed, may be encapsulated, lobulated tumors. They range in size from 1.5–11 cm, but are usually around 4 cm. The cut The initial impression was extraskeletal myxoid surface is smooth and yellow [6]. Both of our cases were chondrosarcoma. However, the final diagnosis after well circumscribed, encapsulated and lobulated but one intra-departmental consultation was chondroid lipoma, of them was distinctly larger than the sizes reported in mainly based on the presence of adipocytes and mature literature (20x13x8 cm in aggregate). adipose tissue within the tumor lobules. After one year of Microscopically, the tumor is composed of a varying surgery, the patient was tumor free. admixture of eosinophilic, multivacuolated cells arranged in strands, nests and sheets, together with mature adipocytes. These are present in a myxoid and chondroid DISCUSSION stroma. Mature adipose tissue may be interspersed within the tumor. Fibrous bands often divide the tumor Chondroid lipoma is a rare, benign lipomatous tumor into lobules. Identical histologic features were noted in that may be confused histologically with a liposarcoma both of our cases. Multivacuolated cells contain fat and or chondrosarcoma. Meis and Enzinger were the first often glycogen (demonstrated by periodic acid–Schiff to recognize chondroid lipoma as a distinct entity when and Oil-red-O stains) and may resemble , they reported a series of 20 cases in 1993 [1]. An identical lipoblasts or hibernoma cells. The nuclei have irregular tumor had, however, been reported in 1986 by Chan et contours and lack pleomorphism and mitotic activity [1]. al. as an example of ‘an extraskeletal with One of our cases showed intracytoplasmic glycogen within lipoblast-like cells’ [3].

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):527–531. Shawarby et al. 530 www.ijcasereportsandimages.com the neoplastic cells and nuclei did not show significant CONCLUSION pleomorphism or mitotic activity in either case. Immunohistochemically, the neoplastic cells stain A high level of suspicion by the pathologist and for vimentin, S-100 and CD68 and some tumors may familiarity with its features are of practical importance to be keratin positive. Ki67 immunoreactivity is typically avoid an overtreatment as the tumor does not recur or very low and detected only in the more primitive cell metastasize, and simple excision is curative [2] because population [4, 7]. Both of our cases showed strong chondroid lipoma is easily misdiagnosed as a sarcoma of immunoreactivity for vimentin and variable reactivity for either adipose tissue or cartilage. S-100. However, only one case showed CD68 positivity. The Ki67 proliferation index was very low in both the cases confirming the very low mitotic activity observed in ACKNOWLEDGEMENTS H&E stained sections. Ultrastructurally, the cells of chondroid lipoma exhibit The authors acknowledge the services of Mr a spectrum of differentiation, ranging from primitive Shakir Ahmed and Mrs Maria Rosario Lazaro from cells sharing features of prelipoblasts and chondroblasts, the histopathology laboratory of the University of to lipoblasts and preadipocytes, to mature adipocytes. Dammam, Saudi Arabia for conducting the histology and The myxohyaline matrix has ultrastructural features of immuunohistochemistry work. cartilage. Numerous mitochondria and lysosomes are absent, indicating that chondroid lipoma is neither a ********* fibromatous lesion nor a lipogranuloma [8]. Cytogenetic analysis of chondroid lipoma revealed Author Contributions a balanced translocation t (11, 16) (q13; p12-13) distinct Mohamed A Shawarby – Substantial contributions to from the known translocation involving 16p11 in myxoid conception and design, Acquisition of data, Analysis and round-cell liposarcoma. The 11q13 breakpoint was and interpretation of data, Drafting the article, Revising previously noted in hibernomas, raising the possibility of it critically for important intellectual content, Final a common genetic deregulation [9, 10]. approval of the version to be published The cellularity of chondroid lipomas and their Maissa N El-Maraghy – Acquisition of data, Revising myxochondroid matrix have caused them to be it critically for important intellectual content, Final histologically confused with myxoid liposarcoma or approval of the version to be published extraskeletal . The histologic Ragaa A Salem – Acquisition of data, Revising it critically differential diagnosis also includes mixed tumors, soft for important intellectual content, Final approval of the tissue chondroma, cartilagenous metaplasia in a lipoma version to be published [11] and parachordoma [12]. Nafissa M El-Badawy – Acquisition of data, Revising Myxoid liposarcoma may contain areas of mature fat it critically for important intellectual content, Final and vacuolated and eosinophilic cells in a myxoid stroma approval of the version to be published similar to chondroid lipoma. However, chondroid lipoma Tarek M El-Sharkawy – Acquisition of data, Revising lacks the distinct delicate branching vasculature present it critically for important intellectual content, Final in myxoid liposarcoma and is usually less cellular. approval of the version to be published Myxoid liposarcoma also lacks the chondroid matrix seen Tarek M Hashem – Acquisition of data, Revising it in chondroid lipoma [11]. critically for important intellectual content, Final Extraskeletal myxoid chondrosarcoma is composed of approval of the version to be published cords of eosinophilic cells that are smaller than those in Ahmad S Kamel – Acquisition of data, Revising it critically chondroid lipoma. The tumor is more lobulated and there for important intellectual content, Final approval of the is absence of adipocytes and mature adipose tissue [11]. version to be published Mixed tumors of the dermis or deep soft tissue are composed of nests and cords of eosinophilic cells in a Guarantor myxochondroid stroma, but they are not as vacuolated The corresponding author is the guarantor of submission. as the cells in chondroid lipoma, and do not demonstrate adipocytic differentiation. Epithelial differentiation Conflict of Interest manifested by glands and myoepithelial cells are also Authors declare no conflict of interest. usually present in mixed tumors. Soft tissue chondroma usually has true hyaline cartilage, does not contain adipose Copyright tissue and arises in the soft tissues of the hand and feet. In © Mohamed A Shawarby et al. 2013; This article is cartilaginous, metaplasia in a lipoma (chondrolipoma), distributed under the terms of Creative Commons true hyaline cartilage is present [11]. attribution 3.0 License which permits unrestricted use, Parachordoma does not contain mature fat and is distribution and reproduction in any means provided EMA and Keratin positive [12]. the original authors and original publisher are properly

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):527–531. Shawarby et al. 531 www.ijcasereportsandimages.com credited. (Please see www.ijcasereportsandimages.com/ brief report of two cases with ultrastructural analysis. copyright-policy.php for more information.) Am J Surg Pathol 1995;19(11):1272–6. 7. Yang YJ, Damron TA, Ambrose JL. Diagnosis of chondroid lipoma by fine-needle aspiration biopsy. Arch Pathol Lab Med 2001;125(9):1224–6. REFERENCES 8. Kindblom LG, Meis-Kindblom JM. Chondroid lipoma: An ultrastructural and immunohistochemical analysis with further observations regarding its 1. Meis JM, Enzinger FM. Chondroid lipoma. A differentiation. Hum Pathol 1995;26(7):706–15. unique tumor simulating liposarcoma and myxoid 9. Ballaux F, Debiec-Rychter M, De Wever I, Sciot chondrosarcoma. Am J Surg Pathol 1993;17(11):1103– R. Chondroid lipoma is characterized by t(11;16) 2. (q13;p12-13). Virchows Arch 2004;444(2):208–10. 2. Thway K, Flora RS, Fisher C. Chondroid lipoma: an 10. Thomson TA, Horsman D, Bainbridge TC. Cytogenetic update and review. Ann Diagn Pathol 2012;16(3):230– and cytologic features of chondroid lipoma of soft 4. tissue. Mod Pathol 1999;12(1):88–91. 3. Chan JK, Lee KC, Saw D. Extraskeletal chondroma with 11. Nielsen GP. Chondroid lipoma. Bone and Soft Tissue lipoblast-like cells. Hum Pathol 1986;17(12):1285–7. . USCAP Virtual slide box. http://www. 4. Vasili C, Kligman M, Kirsh G. Incidental finding of uscap.org/site~/iap2006/slides18-6v.htm chondroid lipoma at total hip arhtroplasty. J South 12. Folpe AL, Agoff SN, Willis J, Weiss SW. Parachordoma Orthop Assoc 2000;9(3):219–1. is immunohistochemically and cytogenetically distinct 5. Lakshmiah SR, Scott KW, Whear NM, Monoghan A. from axial and extraskeletal myxoid Chondroid lipoma: a rare but diagnostically important chondrosarcoma. Am J Surg Pathol 1999;23(9):1059– lesion. Int J Oral Maxillofac Surg 2000;29(6):445–6. 67. 6. Nielsen GP, O’Connell JX, Dickersin GR, Rosenberg AE. Chondroid lipoma, a tumor of white fat cells. A

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CASE REPORT OPEN ACCESS An unusual hip dislocation during tennis playing

Ismail Turkmen, Fatih Turkmensoy, Korhan Ozkan, Salih Soylemez, Feyza Unlu Ozkan, Yalcin Turhan

Abstract *********

Introduction: Isolated traumatic posterior hip Turkmen I, Turkmensoy F, Ozkan K, Soylemez S, Ozkan dislocation is seen as an extremely rare incident FU, Turhan Y. An unusual hip dislocation during tennis during sport activities. In this case report we playing. International Journal of Case Reports and aimed to present an isolated traumatic posterior Images 2013;4(10):532–535. hip dislocation of an amateur tennis player. Case Report: A 26-year-old female patient who was ********* injured during playing tennis was brought to our orthopedic emergency unit and diagnosed doi:10.5348/ijcri-2013-10-373-CR-2 with pure posterior hip dislocation. Her hip was immediately reduced under sedation which then a rehabilitation program was begun. Conclusion: Amateur people generally play tennis on cement floor which may result in hip dislocation, if Introduction slipped. To prevent dislocations and these kinds of extreme injuries non-slippery shoes and shock Acute traumatic hip dislocation is usually caused absorbable relatively soft surfaces to play must by traffic accidents as dashboard injuries. Although be preferred by the players. it happens with high energy trauma, also low energy traumas, which usually happens during sport activities, Keywords: Pure traumatic hip dislocation, might very rarely, cause traumatic hip dislocation. In Tennis player this report we aimed to present an isolated traumatic hip dislocation of an amateur tennis player and this is the first report of this kind of injury during tennis playing.

Ismail Turkmen1, Fatih Turkmensoy1, Korhan Ozkan2, Salih Soylemez1, Feyza Unlu Ozkan3, Yalcin Turhan4 Affiliations: 1MD, Istanbul Medeniyet University Goztepe CASE REPORT Training and Research Hospital, Department of Orthopaedics and Traumatology, Istanbul, TURKEY; 2Ass. Prof, Istanbul A 26-year-old amateur female tennis player was Medeniyet University, Department of Orthopaedics and admitted to our orthopedic emergency unit with hip pain Traumatology, Istanbul, TURKEY; 3MD, Istanbul Fatih Sultan and limitation of hip motion in her left extremity. Clinical Mehmet Training and Research Hospital, Department of examination revealed that her left leg was shortened, Pyscial and Rehabilitation. Istanbul, TURKEY; flexed, adducted and at an internally rotated position. 4MD, Duzce State Hospital, Department of Orthopaedics The patient was playing tennis on a cement floor and and Traumatology, Duzce, TURKEY. while she was running to catch a ball she slipped on a Corresponding Author: Ismail Turkmen, Istanbul Medeniyet leaf. Her legs had struddled and left leg was flexed and University Goztepe Training and Research Hospital, internally rotated. She fell down on the floor and felt an Department of Orthopaedics and Traumatology 34732 Istanbul, Turkey; Ph: 00905304622107; Fax: 0090216-566- extreme pain over her hip. 40-00; Email: [email protected] Radiographs of the pelvis showed posterior dislocation of the hip without fracture of acetabulum or femoral head (Figures 1 and 2). Type 1 dislocation was diagnosed Received: 11 April 2013 according to Thompson and Epstein classification. The Accepted: 10 May 2013 dislocation was reduced immediately under conscious Published: 01 October 2013 sedation (midazolam 5 mg intravenous) after diagnosis.

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Figure 1: X-ray before reduction.

Figure 3: Postreduction magnetic resonance imaging scan showing piriformis edema in left hip.

Traumatic hip dislocation can be rarely seen in contact sports like soccer and rugby and hip dislocation without fracture of the femoral head and acetabulum is also notably rare. In this case report, there was no contact force and pure dislocation had been occurred without fracture. In literature, traumatic hip dislocations in sports activity had been reported. But to our knowledge, there is no case reported that have a pure hip dislocation when playing non-contact sport like tennis. In English literature there are 21 traumatic hip dislocations with and Figure 2: Postreduction coronal computed tomography scan without fracture in sports activity [2–8]. Among all these showing no fracture. —6 rugby, 5 soccer, 2 futsal and one each water skiing, equestrian, gymnastic, jogging, skiing, biking, sledge, Allis’s maneuver was used for reduction. There was no basketball player had traumatic hip dislocation— only sciatic nerve deficit before and after reduction. Skeletal five of them were pure dislocations. traction was not applied. X-ray and magnetic resonance Generally, mechanism of dislocation is that; when imaging (MRI) scan revealed concentric reduction of the hip and knee is flexed and internally rotated, a powerful hip joint (Figure 3). Indomethacin 75 mg was begun for strike comes to the knee and it fractures posterior lip of heterotopic ossification prophylaxis. Early mobilization the acetabulum. If the force is great enough, hip dislocates allowed without weight bearing. Hip flexion over 90 [9]. degrees and internally rotation over 10 degrees were In management of traumatic hip dislocation early prohibited. She was followed-up for six weeks with gentle reduction is essential. Risk of avascular necrosis at abductor and quadriceps strengthening exercises and femoral head increases as time goes on, especially over 6 weight bearing was allowed then. The patient was well six hours [10]. If patient is not proper for general anesthesia months after dislocation with normal activities. for immediate reduction, sedation by midazolam or diazepam can be tried simply. Reduction maneuver should be done gently with an assistant who stabilize DISCUSSION the pelvis otherwise femoral cartilage could be damaged and iatrogenic fractures can be seen. To avoid avascular Tennis is the one of the most popular sport in the necrosis, postreduction early mobilization and controlled world. Lower extremity ligament injuries, contusions, weight bearing should be done in pure dislocations [11]. sprains, abrasions and upper extremity overuse injuries Passive range of motion exercises and partially weight are frequently seen during playing tennis [1]. bearing can be done in following week after reduction.

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The third week patient can be allowed to walk with REFERENCES crutches and full weight bearing. After eight weeks of the dislocation light sport activities can be done and return to 1. Bylak J, Hutchinson MR. Common sports injuries in the competition can take at least six months [12]. young tennis players. Sports Med 1998 Aug;26(2):119– 32. 2. Giannoudis PV, Zelle BA, Kamath RP. Posterior Fracture-Dislocation of the Hip in Sports. European CONCLUSION Journal of Trauma 2003 Dec;29(6):399–2. 3. Schuh A, Doleschal S, Schmickal T. Anterior Hip Tennis is a well-known and widely played sport around Dislocation in a Football Player: A Case Report. Case the world. Usually minor injuries like shoulder rotator Report Med 2009;2009:363461. cuff tears, slap lesions, elbow problems or ligament 4. Yates C, Bandy WD, Blasier RD. Traumatic dislocation injuries due to ankle sprain can be seen during playing of the hip in a high school football player. Phys Ther tennis. Amateur people generally play tennis on cement 2008 Jun;88(6):780–8. floor which may result in hip dislocation, if slipped. To 5. Venkatachalam S, Heidari N, Greer T. Traumatic prevent dislocations and these kinds of extreme injuries fracture-dislocation of the hip following rugby tackle: non-slippery shoes and shock absorbable relatively soft a case report. Sports Med Arthrosc Rehabil Ther Technol 2009 Dec 15;1:28. surfaces to play must be preferred by the players. 6. Giza E, Mithöfer K, Matthews H, Vrahas M. Hip fracture-dislocation in football: a report of two cases ********* and review of the literature. Br J Sports Med 2004 Aug;38(4):E17. Author Contributions 7. Yasin FN, Singh VA. Can posterior hip fracture- Ismail Turkmen – Substantial contributions to dislocation occur in indoor football (futsal)? A conception and design, Acquisition of data, Analysis report of two cases. BMJ Case Rep 2009;2009. pii: and interpretation of data, Drafting the article, Revising bcr12.2008.1317. 8. Nahas RM, Netto E, Chikude T, Ikemoto R. Traumatic it critically for important intellectual content, Final hip fracture-dislocation in soccer: a case report. Rev approval of the version to be published Bras Med Esporte 2007 Jul;13(4). Fatih Turkmensoy – Acquisition of data, Drafting the 9. Epstein HC. Traumatic dislocations of the hip. Clin article, Revising it critically for important intellectual Orthop Relat Res 1973;(92):116–42. content, Final approval of the version to be published 10. Hougaard K, Thomsen PB. Traumatic posterior Korhan Ozkan – Acquisition of data, Drafting the article, dislocation of the hip--prognostic factors influencing Revising it critically for important intellectual content, the incidence of avascular necrosis of the femoral Final approval of the version to be published head. Arch Orthop Trauma Surg 1986;106(1):32–5. Salih Soylemez – Acquisition of data, Drafting the article, 11. Amihood S. Posterior dislocation of the hip. Clinical observations and review of literature. S Afr Med J Revising it critically for important intellectual content, 1974 May 18;48(24):1029–32. Final approval of the version to be published 12. Collins J, Trulock S, Chao DJ. Field management and Feyza Unlu Ozkan – Acquisition of data, Drafting the rehabilitation of an acute posterior hip dislocation in article, Revising it critically for important intellectual a professional football player. Professional Football content, Final approval of the version to be published Athletic Trainer 2001;19:1–3. Yalcin Turhan – Acquisition of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Guarantor The corresponding author is the guarantor of submission.

Conflict of Interest Authors declare no conflict of interest.

Copyright © Ismail Turkmen et al. 2013; This article is distributed under the terms of Creative Commons attribution 3.0 License which permits unrestricted use, distribution and reproduction in any means provided the original authors and original publisher are properly credited. (Please see www.ijcasereportsandimages.com/copyright-policy.php for more information.)

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CASE REPORT OPEN ACCESS A variant common hepatic artery originating from the normal celiac trunk and passing behind the portal vein successfully treated with pancreaticoduodenectomy for middle bile duct cancer: A case Report

Yoshihito Ohta, Michiki Narushima, Adoru Okaue, Hisashi Nakata, Hisahiro Matsubara

Abstract arteriography (CTA) (3D-CT angiography image in arterial phase). The variant course of the common Introduction: Pancreaticoduodenectomy (PD) hepatic artery in this patient was preoperatively consists of multiple complex surgical procedures, identified by fused images from 3D-CTA and including skeletonization of the hepatic artery for venography. Conclusion: Fusing images from 3D- lymph node dissection. Variations in the hepatic CTA and venography should be recommended as artery can be observed in 25–45% of cases, so part of the routine preoperative examination for it is important for surgeons to have knowledge depicting anatomical variations, such as in the of the hepatic blood supply to avoid injury that variant course of the common hepatic artery in may result in biliary fistula or hepatic ischemia the patients planning to undergo PD. after surgery. Case Report: We encountered a 67-year-old male patient with a variant common Keywords: Variant common hepatic artery, hepatic artery which originated from the normal Pancreaticoduodenectomy, Bile duct cancer celiac trunk and passed behind the portal vein. This patient successfully underwent a subtotal- ********* stomach-preserving PD for middle bile duct cancer. The frequency of this variant common Ohta Y, Narushima M, Okaue A, Nakata H, Matsubara hepatic artery is approximately 0.1%; according H. A variant common hepatic artery originating from to two large-scale retrospective studies on the the normal celiac trunk and passing behind the portal variations of the hepatic blood supply. Unlike vein successfully treated with pancreaticoduodenectomy the different branches or number variations, for middle bile duct cancer: A case Report. International it is difficult to identify the course variations Journal of Case Reports and Images 2013;4(10):536–540. with conventional visceral angiography or three-dimensional computed tomography *********

doi:10.5348/ijcri-2013-10-374-CR-3 Yoshihito Ohta1, Michiki Narushima1, Adoru Okaue1, Hisashi Nakata2, Hisahiro Matsubara3 Affiliations: 1Department of Surgery Shimizu Kousei Hos- pital, Shimizu-ku, Shizuoka, Japan; 2Department , Shimizu Kousei Hospital, Shimizu-ku, Shizuoka, Japan; 3Department of Frontier Surgery, Graduate School of Introduction Medicine, Chiba University, Chuo-ku, Chiba, Japan. Corresponding Author: Yoshihito Ohta, Department of Sur- Pancreaticoduodenectomy (PD) requires complex gery, Shimizu Kousei Hospital, 578-1 Ihara-cho, Shimizu-ku, surgical procedures associated with high morbidity Shizuoka city, Shizuoka 424-0114, Japan; Tel: 81-54-366- and even the risk of mortality [1–9]. Variant arterial 3333, Fax: 81-54-366-3333; Email: [email protected] anatomy in patients undergoing PD increases the risk of injury of the hepatic blood supply which may result in biliary fistula or hepatic ischemia after surgery [1, Received: 01 January 2013 7–9]. Meanwhile anatomical variations of the hepatic Accepted: 08 June 2013 artery are not uncommon, which can be observed in Published: 01 October 2013

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25–45% of the cases [5–9]. Therefore, it is extremely important to identify these anomalous vessels prior to surgery when planning therapeutic strategies for PD. Visceral angiography or three-dimensional computed tomography (CT) arteriography (the arterial phase of 3D-CT angiography) is usually performed as a routine diagnostic procedure. However, it is difficult to identify the course variations with these examinations. We encountered a patient with a variant common hepatic artery (CHA) originating from the normal celiac trunk and passing behind the portal vein suffering from middle bile duct cancer. According to two large-scale Figure 1: (A) Abdominal ultrasonography showing a low echoic retrospective studies concerning the variations of the mass (arrows) in the middle of the common bile duct, which hepatic blood supply, this course variation of the CHA is caused the dilatation of the proximal biliary tract, (B) Contrast- very rare, approximately 0.1% of the cases [10, 11]. We enhanced computed tomography of the abdomen showing identified this rare variation by fusing images from 3D- a tumor (arrows) in the middle of the common bile duct, CT arteriography and venography (the venous phase of approximately 2 cm in diameter, which resulted in dilatation of 3D-CT angiography) before performing PD and therefore the proximal biliary tract. were able to safely deal with this variant CHA at surgery.

CASE REPORT

A 67-year-old male patient was referred to our hospital to investigate a case of jaundice on October 2010. He denied anorexia, abdominal pain, alcohol abuse and cigarette smoking. Physical examination revealed a mildly cachectic and icteric male with no acute distress. An abdominal examination revealed no palpable abdominal masses, organomegaly or evidence of ascites. He denied abdominal tenderness. On admission his total bilirubin level 7.52 mg/dL, alkaline phosphatase 1032 IU/L (normal range 105–320 IU/L), carcinoembryonic antigen 11.8 ng/dL (range, 0.0–5.0 ng/dL), carbohydrate antigen 19–9 was 765.1 Um/L (normal range 0.0-37.0 U/ mL). Ultrasonography and contrast-enhanced computed tomography (CT) of the abdomen revealed a mass, approximately 2 cm in diameter, in the middle of the common bile duct (CBD), which caused the dilatation of the proximal biliary tract (Figure 1A–B). Endoscopic retrograde naso-biliary drainage was successfully performed to improve the jaundice. Cholangiography and magnetic resonance cholangiopancreatography Figure 2: Cholangiography showing a 2-cm filling defect revealed a 2.5-cm filling defect in the middle of the CBD (arrows) in the middle of the common bile duct. (Figure 2). Based on these findings, the patient was diagnosed with middle bile duct cancer and a subtotal- stomach-preserving PD was planned. we easily identified and safely dealt with the variant CHA A 3D-CT angiography was performed prior to the PD and completed the complicated procedure including the procedure in order to identify the anatomy of the hepatic skeletonization of this artery for lymph node dissection blood supply. The 3D-CT arteriography showed a normal without any vascular injury. trifurcation of the celiac trunk, CHA, left gastric artery Pathological findings showed moderately and splenic artery (Figure 3A). However, fused images differentiated tubular adenocarcinoma, T2, N1, M0, from the 3D-CT arteriography and venography showed Stage IIB in terms of the UICC TNM classification. The an unusual trajectory of the CHA, passing behind the patient had an uneventful postoperative course except for a portal vein, immediately running towards the ventral grade B pancreatic fistula which was treated by percutaneous side between portal vein and CBD, and ascending to the abdominal puncture. The patient was discharged on hepatic hilum in front of the portal vein after giving off postoperative day-24 and remained disease-free for 16 the gastroduodenal artery (Figure 3B). During surgery, months.

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the hepatic artery in liver transplantation, Abdullah et al. [18] identified one case with the same variant CHA. Based on the retrospective evaluation of the findings in 5002 patients who underwent spiral CT and digital subtraction angiography, Song et al. [19] found six CHAs which had a suprapancreatic retroportal course. To the best of our knowledge, this is the first case reported of a patient with this CHA variation who successfully underwent PD. It may be difficult to identify the course variations with conventional visceral angiography or 3D-CT Figure 3: (A) Three-dimensional computed tomography arteriography, because these examinations cannot showing arteriography (the arterial phase of three-dimensional delineate the dimensional relationship between CHA and computed tomography angiography) the normal anatomy of the portal vein separately by themselves. In our case, fused hepatic blood supply. (B) Fused image of the three-dimensional images of 3D-CT arteriography and venography clearly computed tomography arteriography and venography (the showed the abnormal course of the CHA which passed venous phase of three-dimensional computed tomography behind the portal vein, although CT arteriography by angiography) demonstrated the variant common hepatic artery itself showed a normal configuration of the CHA. arising from normal celiac trunk and passing behind the portal vein (arrows). CONCLUSION

DISCUSSION We herein presented a patient with a variant common hepatic artery originating from the celiac Pancreaticoduodenectomy requires complex surgical artery and passing behind the portal vein which we procedures associated with high morbidity and even identified by fusing images from three-dimensional the risk of mortality [1–9]. Variant arterial anatomy in computed tomography arteriography and venography patients undergoing PD increases the risk of injury of the prior to performing pancreaticoduodenectomy for hepatic blood supply which may result in the inadvertent middle bile duct cancer. Fused images from three- bleeding, biliary fistula or hepatic ischemia after surgery dimensional computed tomography arteriography [1,7–9]. Traverso et al. [1] reported that accidental and venography should be recommended as one of ligation of the replaced CHA can lead to ischemia of the routine preoperative examinations for depiction of bilioenteric anastomosis and a consequent leak. Biehl anatomical variations including the variant course of the et al. [7] and Kim et al. [8] demonstrated poor clinical common hepatic artery in patients planning to undergo outcomes after PD in patients with variant vascular pancreaticoduodenectomy. anatomy and recommended angiography in all patients preparing to undergo PD. After analyzing studies which ********* compared imaging modalities, Shukla et al. [9] concluded that routine preoperative CT angiography for PD helps Author Contributions to identify the hepatic vascular anatomy and thereby Yoshihito Ohta – Substantial contributions to prepares the surgeon to better deal with the vascular conception and design, Acquisition of data, Analysis anomalies intraoperatively [10–12]. and interpretation of data, Drafting the article, Revising As mentioned above, the anatomical variations of the it critically for important intellectual content , Final hepatic blood supply are not rare, which can be observed in approval of the version to be published 25–45% of the cases [13–17]. Variant left or right hepatic Michiki Narushima– Acquisition of data, Analysis and arteries are common anatomical variations of the hepatic interpretation of data, Drafting the article, Revising blood supply. Common hepatic artery divided from the it critically for important intellectual content, Final superior mesenteric artery can be observed in 1.5–4.5% approval of the version to be published of the cases [13–17]. An international classification Adoru Okaue – Acquisition of data, Analysis and of the variations in the vascular anatomy of the liver interpretation of data, Drafting the article, Revising was proposed by Michels [13] in 1966 and modified by it critically for important intellectual content, Final Hiatt et al. [14] in 1994. Additionally, anatomic, surgical approval of the version to be published and radiological studies have described other rare Hisashi Nakata – Acquisition of data, Analysis and hepatic arterial variations [16–19]. We encountered a interpretation of data, Drafting the article, Revising male patient with a variant CHA originating from the it critically for important intellectual content, Final normal celiac trunk and passing behind the portal vein approval of the version to be published who underwent PD for middle bile duct cancer. This Hisahiro Matsubara – Substantial contributions variant course of the CHA has seldomly been previously to conception and design, Analysis and identified or reported. After studying 932 dissections of interpretation of data, Drafting the article, Revising

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):536–540. Ohta et al. 539 www.ijcasereportsandimages.com it critically for important intellectual content, 11. Egorov VI, Yashina NI, Fedorov AV, Karmazanovsky Final approval of the version to be published GG, Vishnevsky VA, Shevchenko TV. Celiaco- mesenterial arterial aberrations in patients Guarantor undergoing extended pancreatic resections: correlation of CT angiography with findings at The corresponding author is the guarantor of submission. surgery. JOP 2010;11(4):348–57. 12. Winston CB, Lee NA, Jarnagin WR, et al. CT Conflict of Interest Angiography for delineation of celiac and superior Authors declare no conflict of interest. mesenteric artery variants in patients undergoing hepatobiliary and pancreatic surgery. AJR Am J Copyright Roentgenol 2007;189(1):W13–9. © Yoshihito Ohta et al. 2012; This article is distributed 13. Michels NA. Newer anatomy of the liver and its under the terms of Creative Commons attribution 3.0 variant blood supply and collateral circulation. Am J License which permits unrestricted use, distribution and Surg 1966;112(3):337–47. 14. Hiatt JR, Gabbay J, Busuttil RW. Surgical anatomy reproduction in any means provided the original authors of the hepatic arteries in 1000 cases. Ann Surg and original publisher are properly credited. (Please see 1994;220(1):50–2. www.ijcasereportsandimages.com/copyright-policy.php 15. Chen H, Yano R, Emura S, Shoumura S. Anatomic for more information.) variation of the celiac trunk with special reference to hepatic artery patterns. Ann Anat 2009;191(4):399– 407. REFERENCES 16. Covey AM, Brody LA, Maluccio MA, Getrajdman GI, Brown KT. Variant hepatic arterial anatomy revisited: 1. Traverso LW, Freeny PC. Pancreaticoduodenectomy. digital subtraction angiography performed in 600 The importance of preserving hepatic blood flow to patients. 2002;224(2):542–7. prevent biliary fistula. Am Surg 1989;55(7):421–6. 17. De Cecco CN, Ferrari R, Rengo M, Paolantonio P, 2. Woods MS, Traverso LW. Sparing a replaced common Vecchietti F, Laghi A. Anatomic variations of the hepatic artery during pancreaticoduodenectomy. Am hepatic arteries in 250 patients studied with 64-row J Surg 1994 Jan;167:27-33 CT angiography. Eur Radiol 2009;19(11):2765–70. 3. Volpe CM, Peterson S, Hoover EL, Doerr RJ. 18. Abdullah SS, Mabrut JY, Garbit V, et al. Anatomical Justification for visceral angiography prior to variations of the hepatic artery: study of 932 pancreaticoduodenectomy. Am Surg 1998;64(8):758– cases in liver transplantation. Surg Radiol Anat 61. 2006;28(5):468–73. 4. Miyamoto N, Kodama Y, Endo H, et al. Embolization 19. Song SY, Chung JW, Yin YH, et al. Celiac axis and of the replaced common hepatic artery before surgery common hepatic artery variations in 5002 patients: for pancreatic head cancer: report of a case. Surg systematic analysis with spiral CT and DSA. Radiology Today 2004;34(7):619–22. 2010;255(1):278–8. 5. Yamamoto S, Kubota K, Rokkaku K, Nemoto T, Sakuma A. Disposal of replaced common hepatic artery coursing within the pancreas during pancreatoduodenectomy: report of a case. Surg Today 2005;35(11):984–7. 6. Wang MJ, Cheng Z, Wang R, Li Y, Zhou ZG. Unusual course of the common hepatic artery originating from the celiac trunk. Surg Radiol Anat 2010 Nov;32(9):883–5. 7. Biehl TR, Traverso LW, Hauptmann E, Ryan JA Jr. Preoperative visceral angiography alters intraoperative strategy during the Whipple procedure. Am J Surg 1993 May;165(5):607–12. 8. Kim AW, McCarthy WJ 3rd, Maxhimer JB, et al. Vascular complications associated with pancreaticoduodenectomy adversely affect clinical outcome. Surgery 2002;132(4):738–44. 9. Shukla PJ, Barreto SG, Mohandas KM, Shrikhande SV. Defining the role of surgery for complications after pancreatoduodenectomy. ANZ J Surg 2009;79(1- 2):33–7. 10. Sakai H, Okuda K, Yasunaga M, Kinoshita H, Aoyagi S. Reliability of hepatic artery configuration in 3D CT angiography compared with conventional angiography--special reference to living-related liver transplant donors. Transpl Int 2005;18(5):499–505.

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CASE REPORT OPEN ACCESS Isolated tuberculosis of the wrist: A rare case of extrapulmonary tuberculosis

Mohamed Altayeb Mussa, Edmund Fitzgerald O’Connor, Stuart Waterston, Michael Taylor, Fortune Iwuagwu

Abstract Conclusion: Persistent swelling of bones or joints can be a presenting sign of tuberculosis. A normal Introduction: Atypical infections with chest radiograph or the absence of systemic mycobacteria are unusual in the developed symptoms does not exclude the possibility of bone countries and tuberculous involvement of the tuberculosis. When confronted with unusual wrist or carpal bones is a rare presentation. Case inflammatory findings, always send tissue for Report: This is a new rare case of tuberculosis histology and alcohol and acid-fast bacilli (AAFB) of the wrist joint in a non-immunocompromised culture. 24-year-old male without concomitant pulmonary tuberculosis. He then underwent two months Keywords: Tuberculosis, Wrist, Oligoarthritis, course of intra-articular steroid injections Carpal bones and methotrexate therapy for a presumed diagnosis of inflammatory oligoarthritis of the ********* wrist as radiographs were reported as normal. The diagnosis was initially obscured by lack Mussa MA, O’Connor EF, Waterston S, Taylor M, of systemic symptoms and was established by Iwuagwu F. Isolated tuberculosis of the wrist: A rare case direct visualization of acid-fast bacilli on joint of extrapulmonary tuberculosis. International Journal of fluid and biopsy of the abscess. Musculoskeletal Case Reports and Images 2013;4(10):541–545. involvement in tuberculosis may be easily missed because of its non-specific clinical signs. The ********* disease may mimic inflammatory arthritis and high index of suspicion is required when dealing doi:10.5348/ijcri-2013-10-375-CR-4 with long standing inflammatory swellings.

Mohamed Altayeb Mussa1, Edmund Fitzgerald O’Connor2, Stuart Waterston2, Michael Taylor3, Fortune Iwuagwu4 Affiliations: 1Orthopaedics Specialist Registrar, Hull Royal In- Introduction firmary, 4 School Lane Mews, Beverley, East Riding of York- shire, HU17 9LS, United Kingdom; 2Plastic Surgery Special- There has been a significant worldwide increase in the ist Registrar, Mid Essex Hospital, Court Road, Broomfield, prevalence of tuberculosis (TB) and of its extrapulmonary 3 Chelmsford, Essex, CM1 7ET, United Kingdom; Consultant manifestations [1, 2]. Osteoarticular TB accounts for 1–2% Orthopaedics and Traumatology, Mid Essex Hospital, Court of all the TB cases in the western world [3]. The spine being Road, Broomfield, Chelmsford, Essex, CM1 7ET, United Kingdom; 4Consultant Hand and , Mid Essex the most common site of osseous involvement, accounting Hospital, Court Road, Broomfield, Chelmsford, Essex, CM1 for about 50% of cases [4]. Tuberculous involvement of 7ET, United Kingdom. the wrist joint is a rare presentation. The diagnosis may Corresponding Author: Mohamed Altayeb Mussa, Orthopae- be initially difficult owing to the rarity of the disease, its dics Specialist Registrar, Hull Royal Infirmary, 4 School Lane non-specific clinical signs and lack of awareness resulting Mews, Beverley, East Riding of Yorkshire, HU17 9LS, United in delay in starting treatment, progression of disease and Kingdom; Tel: 00447590334551; Email: mohamedaltayeb@ more advanced disability [5]. gmail.com

Received: 11 February 2013 CASE REPORT Accepted: 10 May 2013 Published: 01 October 2013 A previously healthy 24-year-old British male was admitted to the accident and emergency department

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):541–545. Mussa et al. 542 www.ijcasereportsandimages.com with a facial laceration after being involved in a fight. An incidental finding of a swollen right wrist was made and was subsequently referred to our hand trauma and plastic surgery unit with a diagnosis of an abscess. The patient gave a one-year history of a spontaneously appearing painful lump on the dorsum of the right dominant hand, which had gradually increased in size with no history of trauma. There was no associated history of fever, loss of weight, night sweats or fatigue. Prior to presentation, he had been initially treated with non-steroidal anti-inflammatories for a painful wrist joint. He then underwent two months course of intra- articular steroid injections and methotrexate therapy for a presumed diagnosis of inflammatory oligoarthritis of the wrist as radiographs were reported as normal. The condition did not improve and wrist movements became increasingly painful and restricted. Clinical examination revealed an average built man with no peripheral lymphadenopathy. There was a painless swelling located on the dorso-radial aspect of the right wrist, measuring approximately 4×4 cm with cellulitic inflamed overlying skin (Figure 1). The patient was very reluctant to actively move his affected wrist and passive range of movement was limited by pain. The rest of the clinical examination including respiratory system examination was unremarkable. Blood tests showed hemoglobin 15.3 g/dL, total leukocyte count 8,900/mm3, neutrophils 5.2x103/L, lymphocytes 1.0x109/ µL, monocytes 0.9x103/L, eosinophil 0/µL, erythrocyte sedimentation rate 32 mm/h and C-reactive protein 5.0 mg/L. He was HIV seronegative and chest radiograph Figure 1: Swelling of dorsum of right hand and wrist joint with was normal with no signs of pulmonary tuberculosis cellulitic overlying skin. (Figure 2). Computed tomography (CT) scan (Figure 3) and radiographs of the right hand and wrist AP and lateral views (Figures 4 and 5) revealed multiple carpal and distal radius erosions. Magnetic resonance imaging (MRI) scan with contrast confirmed the presence of multiple osteolytic lesions and showed extensive soft tissue thickening, edema of the extensor tendons and fluid noted affecting mainly the dorsum of the wrist (Figure 6). Drainage of the wrist yielded pus and inflammatory tissue. Histological assessment showed caseating granuloma, whilst staining confirmed the presence of acid-fast bacilli. The diagnosis of extrapulmonary tuberculosis affecting the distal radius and carpal bones with tuberculous tenosynovitis was made. Anti- tuberculous treatment was commenced with four drugs (rifampicin, ethambutol, isoniazid and pyrazinamide) for two months, followed by two drugs (rifampicin, isoniazid) for 12 months. The infection settled with antitubercular drugs and no further intervention was needed.

DISCUSSION

Tuberculous infection of the musculoskeletal system is rare even in areas of high TB prevalence [6]. Hand Figure 2: Chest X-ray showing no lesions.

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Figure 3: Computed tomography scan of wrist and carpal bones showing extensive multiple erosions of carpal bones and adjacent radial styloid.

Figure 5: X-ray of right hand and wrist lateral projection.

Figure 6: Magnetic resonance imaging scan of right hand and wrist with contrast showing extensive soft tissue thickening, Figure 4: X-ray of right hand and wrist joint (AP) projection edema and fluid around the extensor tendons and also to the showing erosions of carpal bones. common flexor sheath in the wrist. Multiple erosions are seen in the carpal bones and the MCP bases.

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):541–545. Mussa et al. 544 www.ijcasereportsandimages.com and wrist involvement are seen in 10% of patients with responding to conservative measures. A normal chest musculoskeletal disease [7]. The symptoms initially radiograph or the absence of systemic symptoms does are non-specific in the form of joint pain, swelling, not exclude the possibility of bone tuberculosis. When effusion, stiffness, carpal tunnel syndrome, limitation confronted with unusual inflammatory findings, always of movement, and discharging sinuses. Constitutional send tissue for histology and alcohol and acid-fast bacilli symptoms include low-grade fever, night sweats, weight culture. Surgery is an adjunct to drugs to control the loss, and anorexia. Blood tests may show mildly raised or disease or to improve the function. Because results are even normal inflammatory markers. much better during earlier stages of the disease, prompt Sclerosis and osteolytic lesions, the main radiographic diagnosis and adequate treatment are essential. features of bony tuberculosis, are non-specific and are present in other conditions such as inflammatory ********* arthritis, pyogenic osteomyelitis, and some malignancies [8]. Other radiographic features include osteopenia, Author Contributions soft-tissue swellings, periosteal reaction, narrowing of Mohamed Altayeb Mussa – Substantial contributions the joint space and bone cysts [8–10]. The CT and MRI to conception and design, Acquisition of data, Analysis scans, although non-specific, may help in the differential and interpretation of data, Drafting the article, Revising diagnosis and in evaluating the extent of the lesion. The it critically for important intellectual content, Final diagnosis of TB may be confirmed on the recognition of approval of the version to be published Mycobacterium tuberculosis on either histology study or Edmund Fitzgerald O’Connor – Acquisition of data, culture, or, ideally, both [11–13]. But the gold standard Analysis and interpretation of data, Drafting the article, for the diagnosis of osseous tuberculosis would be culture Revising it critically for important intellectual content, of Mycobacterium tuberculosis from bone tissue. Final approval of the version to be published There is no consensus on the accurate duration of Stuart Waterston – Acquisition of data, Revising it treatment and this remains a matter of great debate. critically for important intellectual content, Final However, a prolonged course of antituberculous drugs approval of the version to be published is the basis of treatment [5, 7, 14]. Watts and Lifeso Michael Taylor – Acquisition of data, Revising it critically recommended that treatment should be continued for a for important intellectual content, Final approval of the minimum of 12 months for osteoarticular involvement version to be published [15]. Kotwal and Khan [16] treated cases of hand Fortune Iwuagwu – Acquisition of data, Revising it tuberculosis with antitubercular chemotherapy using four critically for important intellectual content, Final drugs (INH, rifampin, pyrazinamide, and ethambutol) approval of the version to be published for four months; followed by three drugs (INH, rifampin, and pyrazinamide) for three months; and finally two Guarantor drugs (INH and rifampin) for 11 months [17]. However, The corresponding author is the guarantor of submission. the optimal duration of treatment has been an issue of considerable debate. Conflict of Interest The absence of active pulmonary tuberculosis does not Authors declare no conflict of interest. eliminate the possibility of an osteoarticular tuberculosis [18]. Systemic disease, diabetes mellitus, and local factors Copyright such as trauma or intra-articular steroids may predispose © Mohamed Altayeb Mussa et al. 2013; This article to activation of a distant focus [9]. The clinical picture and is distributed under the terms of Creative Commons radiological features in the early stages of the disease may attribution 3.0 License which permits unrestricted use, simulate arthritis and a definitive diagnosis is established distribution and reproduction in any means provided by open biopsy and tissue culture [10]. Surgery is an the original authors and original publisher are properly adjunct to the antituberculosis medications as surgical credited. (Please see www.ijcasereportsandimages.com/ procedures may be necessary to control the disease or to copyright-policy.php for more information.) improve the function.

REFERENCES CONCLUSION 1. Barritt AW, Clark L, Teoh V, Cohen AM, Gibb PA. In summary, persistent swelling of bones or joints can Assessing the adequacy of procedure-specific consent be a presenting sign of tuberculosis. The diagnosis can forms in orthopaedic surgery against current methods be easily missed because of the non-specific clinical signs of operative consent. Annals of the Royal College of resulting in progression of disease and more advanced Surgeons of England 2010 Apr;92(3):246-9. 2. Evanchick CC, Davis DE, Harrington TM. Tuberculosis disability. The disease may mimic inflammatory arthritis of peripheral joints: An often missed diagnosis. The and therefore, high index of suspicion is required when Journal of 1986 Feb;13(1):187-9. dealing with long standing inflammatory swellings not

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3. Shah BA, Splain S. Multifocal osteoarticular 11. Karanas YL, Yim KK. Mycobacterium tuberculosis tuberculosis. Orthopedics 2005 Mar;28(3):329-32. infection of the hand: A case report and review 4. Moon MS. Tuberculosis of the spine. Controversies of the literature. Annals of plastic surgery 1998 and a new challenge. Spine 1997 Aug 1;22(15):1791-7. Jan;40(1):65-7. 5. Subasi M, Bukte Y, Kapukaya A, Gurkan F. 12. Watts HG, Lifeso RM. Tuberculosis of bones and Tuberculosis of the metacarpals and phalanges of the joints. J Bone Joint Surg Am 1996 Feb;78(2):288-98. hand. Annals of plastic surgery 2004 Nov;53(5):469- 13. Dhillon MS, Gupta RK, Bahadur R, Nagi ON. 72. Tuberculosis of the sternoclavicular joints. Acta 6. Leung PC. Tuberculosis of the hand. The Hand 1978 orthopaedica Scandinavica 2001 Oct;72(5):514-7. Oct;10(3):285-91. 14. Skoll PJ, Hudson DA. Tuberculosis of the 7. Martini M, Benkeddache Y, Medjani Y, Gottesman H. upper extremity. Annals of plastic surgery 1999 Tuberculosis of the upper limb joints. International Oct;43(4):374-8. orthopaedics 1986;10(1):17-23. 15. St Clair Strange FG. Current concepts review. 8. Agarwal S, Caplivski D, Bottone EJ. Disseminated Tuberculosis of bones and joints (78-A:288-298, Feb. tuberculosis presenting with finger swelling in a 1996) by Watts and Lifeso. J Bone Joint Surg Am 1998 patient with tuberculous osteomyelitis: A case report. Apr;80(4):604. Annals of clinical microbiology and antimicrobials 16. Kotwal PP, Khan SA. Tuberculosis of the hand: 2005;4:18. Clinical presentation and functional outcome in 32 9. Berney S, Goldstein M, Bishko F. Clinical and patients. J Bone Joint Surg Br 2009 Aug;91(8):1054- diagnostic features of tuberculous arthritis. The 7. American journal of medicine 1972 Jul;53(1):36-42. 17. Al-Qattan MM, Al-Namla A, Al-Thunayan A, Al- 10. Wallace R, Cohen AS. Tuberculous arthritis: A report Omawi M. Tuberculosis of the hand. J Hand Surg Am of two cases with review of biopsy and synovial fluid 2011 Aug;36(8):1413-21. findings. The American journal of medicine 1976 18. Mann KJ. Lung lesions in skeletal tuberculosis; review Aug;61(2):277-82. of 500 cases. Lancet 1946 Nov 23;2(6430):744-9.

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CASE REPORT OPEN ACCESS Thoracic epidural anesthesia for modified radical mastectomy in carcinoma of breast patient with chronic obstructive pulmonary disease: A case report

Balaji Narayan Asegaonkar, Sujata Rahul Zine, Unmesh Vidyadhar Takalkar, Umesh Kulkarni, Shilpa Balaji Asegaonkar, Pushpa Kodlikeri

Abstract anesthesia in a diagnosed case of carcinoma of breast with hypertension, type 2 diabetes mellitus Introduction: Modified radical mastectomy, the and severely compromised pulmonary function. standard surgical procedure in the management Case Report: A 66-year-old female, a known case of carcinoma of breast is routinely performed of chronic obstructive pulmonary disease since under general anesthesia. But the patients of seven years with comorbidites (ASA grade III), chronic obstructive pulmonary disease with presented with carcinoma of breast was scheduled other comorbidites are at increased risk of for modified radical resection. Continuous perioperative morbidity and mortality especially thoracic epidural anesthesia was administered because of pulmonary complications. We report at T4-5 level. Local anesthetic titrated as per the successful perioperative management of modified demands of surgery and postoperative analgesia. radical mastectomy only with thoracic epidural Chromic obstructive pulmonary disease has been considered as independent risk factor for postoperative morbidity and mortality Balaji Narayan Asegaonkar1, Sujata Rahul Zine2, because of cardiopulmonary complications. But Unmesh Vidyadhar Takalkar3, Umesh Kulkarni4, Shilpa Balaji Asegaonkar5, Pushpa Kodlikeri6 Thoracic epidural anesthesia, one of the regional Affiliations: 1MD, D.N.B. , Consultant anesthesia techniques, with use of low dose of Anesthesiologist Kodlikeri Memorials CIIGMA Institute local anesthetic helps preserving respiratory of Medical Sciences Aurangabad, Maharashtra, India; function. The procedure was well tolerated 2M.B.B.S.D.A.F.C.P.S. Anesthesiology, Consultant without cardiopulmonary complications which Anesthesiologist Kodlikeri Memorials CIIGMA Institute of lead to prompt recovery with additional effect of 3 Medical Sciences Aurangabad, Maharashtra, India; MS, prolonged postoperative analgesia. Conclusion: Surgery M.E.D.S, F.U.I.C.C. (Switzerland), MSSAT (USA), Thoracic epidural anesthesia provided not Cancer, Consultant Surgeon Kodlikeri Memorials CIIGMA only hemodynamic, cardiopulmonary stability Institute of Medical Sciences Aurangabad, Maharashtra, India; 4M.D. Biochemistry, Asistant Professor, Department but also adequate anesthesia, analgesia and of Biochemistry Government Medical College Aurangabad satisfaction to patient in postoperative phase. It Maharashtra, India; 5M.S. Surgery, Consultant Surgeon proved to be an excellent anesthesia technique Kodlikeri memorials CIIGMA Institute of Medical Sciences for modified radical mastectomy in patient with Aurangabad, Maharashtra, India; 6M.B.B.S, MIFEE, MIAHR, chronic obstructive pulmonary disease. Consultant Gynecologist Kodlikeri memorials CIIGMA Institute of Medical Sciences Aurangabad, Maharashtra, Keywords: Carcinoma of breast, Modified radical India. mastectomy, Thoracic epidural anesthesia, Corresponding Author: Dr. Balaji Narayan Asegaonkar, Chronic obstructive pulmonary disease (COPD) MD, D.N.B. Anesthesiology, C-13 Swarsangam Housing Society New Shrey Nagar, Aurangabad, Maharashtra, India - 431005; Ph: 919325078733; Fax: 912402359279; E-mail: ********* [email protected] Asegaonkar BN, Zine SR, Takalkar UV, Kulkarni U, Asegaonkar SB, Kodlikeri P. Thoracic epidural anesthesia Received: 11 February 2013 for modified radical mastectomy in carcinoma of breast Accepted: 10 May 2013 patient with chronic obstructive pulmonary disease: A Published: 01 October 2013

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):546–550. Asegaonkar et al. 547 www.ijcasereportsandimages.com case report. International Journal of Case Reports and three years and type 2 diabetes mellitus since four years Images 2013;4(5):546–550. maintained on oral medication. Her HbA1C was 7.5% suggesting moderate glycemic control. Chest radiograph ********* showed changes of chronic bronchitis with hyperlucency (Figure 1). On auscultation, ejection systolic murmur was doi:10.5348/ijcri-2013-10-376-CR-5 present in aortic area. She was referred to cardiologist for detail cardiological examination and pulmonologist for evaluation of pulmonary function. Echocardiography evaluation in clinic revealed aortic and mitral sclerosis with mild mitral regurgitation and mild Introduction pulmonary hypertension (PASP 26 mmHg). Pulmonary evaluation revealed severe obstructive Worldwide prevalence of carcinoma of the breast is airway disease with following results of pulmonary increasing at an alarming rate and is a leading cause of function tests (PFT) as given in Table 1. This indicates cancer related mortality in women. In India, prevalence severe airway obstructive disease. There is increased of carcinoma of breast varies from 12–31 cases per residual volume that means increased air trapping. 100,000 women [1]. Modified radical mastectomy With such compromised pulmonary status, COPD, (MRM), the standard surgical procedure of choice hypertension, diabetes mellitus and pulmonary remains the mainstay of management in these patients. hypertension patient was accepted as grade III as per Usually, MRM is performed under general anesthesia. ASA (American Society of Anesthesiologists) and posted But the patients of chronic obstructive pulmonary disease for MRM under thoracic epidural anesthesia. General (COPD) with other comorbidites are at high risk of anesthesia was relatively contraindicated so as to avoid perioperative morbidity and mortality especially because postoperative ventilatory support and complications. of pulmonary complications under general anesthesia Meanwhile we tried to optimize patient’s respiratory [2]. Several studies reported usefulness of cervical and status for five days by round the clock nebulisation thoracic epidural anesthesia for MRM in patients of with steam, N-acetyl cysteine, deep breathing exercises, carcinoma of breast [3, 4]. But these techniques are not maintaining adequate hydration. routinely practiced. We report successful perioperative Patient was explained about the procedure and management with thoracic epidural anesthesia in a technique of the anesthesia and high risk informed diagnosed case of carcinoma of breast with severely consent was obtained. Intravenous access was taken compromised pulmonary function due to COPD with 18 G angiocath on opposite upper limb. Monitoring undergoing MRM. started with pulse oxymetry, noninvasive blood pressure and five-lead electrocardiograms. Anesthesia technique: In sitting position, with all CASE REPORT aseptic precautions entry point marked at T4-5 level. Local anesthetic agent 2% xylocaine was injected A 66-year-old female presented to our center with a with number 26 hypodermic needle. Epidural space chief complaint of a lump in breast gradually increasing was identified by using loss of resistance with saline in size over last four months. On clinical examination, a technique. Then 18 G B Braun epidural catheter was hard lump of size 4x4 cm, with irregular surface, not fixed inserted 4 cm into epidural space through Touhy needle to skin with enlarged axillary lymph nodes were noticed. in cephalad direction. Epidural catheter was fixed and Fine needle aspiration cytology revealed presence of 3 mL test dose of xylocaine with 2% adrenalin was given. malignant cells and patient was scheduled for MRM. Patient was monitored for vital signs for three minutes Patient was a known case of COPD since last seven to rule out intravenous placement. The initial titrated years. She had episodes of exaggeration and remission dose of 7 mL of 0.75% ropivacaine and 1 mL fentanyl (50 once or twice in a year. She required admission twice microgram) injected through catheter which resulted in in last two years for COPD. During preoperative evaluation, anesthesiologists found that because of compromised respiratory status patient is at high risk Table 1: Pulmonary function tests of patient for the procedure to be done under general anesthesia. Parameters Measured volume % Predicted On clinical examination, she was afebrile with pulse rate volume 110/minute, respiratory rate 24/minute with SpO2 92% in lying down position and 94–96% on pulse oxymetry FEV1 1.17 44.6 in sitting position. Bilateral coarse crept were present. FVC 2.60 74.9 Her hemoglobin was 16 g/dL suggesting chronic hypoxia. Her other laboratory investigations blood glucose levels, Residual volume 2.93 112 liver, renal and thyroid parameters were within normal limit. Patient was a known case of hypertension since FEV1:FVC 45

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):546–550. Asegaonkar et al. 548 www.ijcasereportsandimages.com bilateral anesthesia of thoracic wall in the area from C6 to T7 level in next 15 minutes. After achieving adequate sensory block and analgesia patient was handed over to surgeon for MRM. At the time of axillary node clearance, ketamine 25 mg, fentanyl 50 mg and midazolam 1 mg were administered. Injection ondensetron 4 mg was given intravenously to avoid nausea and vomiting. During surgical procedure surgeon appreciated apparently less blood loss and relatively dry surgical field (Figure 2). We deliberately avoided oxygen supplementation to maintain patient’s hypoxic drive. All airway control equipment kept ready for emergency. Surgery lasted for 80 minutes without evidence of any untoward events and throughout the procedure patient was hemodynamically stable (Figure 3). Then patient was shifted to surgical intensive care unit with continuous monitoring for vital parameters. Postoperative analgesia was managed with epidural infusion of 0.125% bupivacaine 6 mL/h till next Figure 2: Dry surgical field 24 hours. Patient continued to receive nebulization and chest physiotherapy in postoperative period comfortably as she was pain free.

DISCUSSION

Thoracic epidural anesthesia has been established as a cornerstone in the perioperative management for

Figure 3: Intraoperative vital parameters.

thoracic, abdominal and lower limb with maximum clinical benefits of speedy recovery, effective analgesia and improved outcome [5]. However, it is practiced less frequently. The COPD has been considered as an independent risk factor for postoperative morbidity and mortality because of cardiopulmonary complications. From anesthesiologist point of view, general anesthetic agents, opiods, muscle relaxants and mechanical ventilation interfere with respiratory function. This combined effect of general anesthesia in supine position leads to instant fall in lung volumes with atelectasis in dependent part of lungs [2]. These patients are difficult to wean from ventilator and may require postoperative prolonged ventilation. So in our case of COPD with hypertension, T2DM and pulmonary hypertension, we planned the procedure using a sole anesthetic technique Figure 1: Chest X-ray showing changes of chronic bronchitis. of TEA which provided safe and excellent analgesia

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):546–550. Asegaonkar et al. 549 www.ijcasereportsandimages.com with improved surgical conditions. The procedure was advantage of the technique is good quality postoperative well tolerated without cardiopulmonary complications analgesia which enhances patient’s compliance for chest which lead to prompt recovery with additional effect of physiotherapy and hence speedy recovery. prolonged postoperative analgesia. Thoracic epidural anesthesia, one of the regional ********* anesthesia techniques, with use of low dose of local anesthetic helps to preserve respiratory function. Author Contributions O’Connor et al. reported successful anesthetic Balaji Narayan Asegaonkar – Anesthetic management of management for bilateral mammoplasty with TEA in a the patient, Analysis and interpretation of data, drafting Klippwl–Feil syndrome with difficult airway [6]. In a study and critical revision of article and final approval of among Thai women researchers observed TEA combined manuscript. with brachial plexus block an alternative safe anesthetic Sujata Rahul Zine – Anesthetic management of the technique for MRM provided effective anesthesia and patient, drafting and critical revision of article and final postoperative analgesia than general anesthesia [7]. approval of manuscript. Ashok Jadon highlighted utility of cervical epidural Unmesh Vidyadhar Takalkar – Diagnosis and surgical analgesia in managing a complex case of carcinoma of Management of case, critical revision of article, final breast with chronic regional pain syndrome [8]. A recent approval of manuscript. meta-analysis about pulmonary effects of TEA showed Unmesh Kulkarni – surgical Management of case, critical decline in postoperative pulmonary complication like revision of article, final approval of manuscript. pneumonia due to earlier ambulation, reduced opiods Shilpa Balaji Asegaonkar – Acquisition of data, Analysis consumption and improved compliance of patient for and interpretation of data, drafting and critical revision chest physiotherapy [9]. Some retrospective studies of article and final approval of manuscript. reported improved survival with reduced prevalence of Pushpa Kodlikeri – Concerned with perioperative tumor recurrence after TEA or paravertebral block in management of the patient, drafting and critical revision cancer patients [10, 11]. of article and final approval of manuscript. Successful use of high TEA avoids tracheal intubation hence also minimizes postoperative pulmonary Guarantor complications [12].With TEA using high concentrations The corresponding author is the guarantor of submission. of local anesthetics (lidocaine 2%, bupivacaine 0.5%) paralysis of the intercostal and abdominal wall muscles Conflict of Interest are responsible for 10–20% decrease in inspiratory Authors declare no conflict of interest. and expiratory capacity without affecting the hypoxic pulmonary vasoconstriction [13]. Diaphragmatic function Copyright remains unimpeded as far as the neuraxial blockade © Balaji Narayan Asegaonkar et al. 2013; This article remains below the cervical emergence of phrenic nerves is distributed under the terms of Creative Commons (C3–C5). So, it is extremely important to watch level of attribution 3.0 License which permits unrestricted use, epidural block because if level reaches above C6, Horner’s distribution and reproduction in any means provided syndrome may develop. If level goes up to C4, patient’s the original authors and original publisher are properly voluntary efforts of respiration stop and might require credited. (Please see www.ijcasereportsandimages.com/ ventilatory support. Site of puncture decides the cephalad copyright-policy.php for more information.) extension of block. But the higher the placement site, the lesser is cephalad spread and more caudal spread [14], hence we selected site T4-5. REFERENCES Even though we performed present surgery using technique of TEA, we always have to assess risk and benefit 1. Agarwal G, Ramakant P. Breast Cancer Care in India: ratio. Common complication of this technique is dural The Current Scenario and the Challenges for the puncture, neurological injury and epidural hematoma. Future. Breast Care (Basel) 2008;3(1):21–7. With maximum precautions and experienced hand dural 2. Licker M, Schweizer A, Ellenberger C, Tschopp JM, Diaper J, Clergue F. Perioperative medical puncture is rare while the incidence of neurological injury management of patients with COPD. Int J Chron is 0.01–0.001% [15]. Obstruct Pulmon Dis 2007;2(4):493–515. 3. Singh AP, Tewari M, Singh DK, Shukla HS. Cervical epidural anesthesia: a safe alternative to general CONCLUSION anesthesia for patients undergoing cancer breast surgery. World J Surg 2006;30(11):2043–7. Compared to general anesthesia, regional anesthesia 4. Trikha A, Sadhasivam S, Saxena A, Arora MK, Deo such as thoracic epidural anesthesia offers better SV. 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CASE REPORT OPEN ACCESS ‘Wallpaper paste sign’ of mucinous breast carcinoma

Muhammad Asad Parvaiz, Brian Isgar, Nedra Aluwihare

Abstract diagnosed on patient’s first visit to the breast clinic on gross inspection of the needle core Introduction: Mucinous carcinoma of the breast biopsy specimen. is an uncommon disease, characterized by a large amount of mucin production. Histologically, it Keywords: Mucinous breast carcinoma, is classified into two subgroups— pure type and Wallpaper paste sign, Extracellular mucin, Core mixed type. It usually affects postmenopausal biopsy women and is commonly associated with good prognosis. Case Report: We present a case of a ********* 53-year-old female with a palpable breast lump which was clinically as well as radiologically Parvaiz MA, Isgar B, Aluwihare N. ‘Wallpaper paste sign’ suspicious of breast cancer. A free hand needle of mucinous breast carcinoma. International Journal of core biopsy was carried out. The biopsy material Case Reports and Images 2013;4(10):551–553. retrieved had a distinctive gross appearance, with soft, gelatinous consistency and a glistening clear ********* surface. On this typical macroscopic appearance, a diagnosis of ‘mucinous breast carcinoma’ doi:10.5348/ijcri-2013-10-377-CR-6 was made, which was subsequently confirmed on histological examination. The typical gross appearance of specimen on needle core biopsy is similar to the appearance of wallpaper paste; hence we describe it as ‘wallpaper paste sign’. Introduction Conclusion: The hallmark of mucinous carcinoma Mucinous carcinoma of the breast is not a common is extracellular mucin production that gives it disease and its incidence has been reported to range a typical glistening macroscopic appearance. from 1–6% of all primary breast cancers [1–4]. It is Keeping in mind the ‘wallpaper paste sign’ we characterized by a large amount of mucin production and describe, mucinous breast carcinoma can be in general, defined as having a mucinous component of 50% or more [5, 6]. Histologically, it is classified into two Muhammad Asad Parvaiz1, Brian Isgar2, Nedra Aluwihare3 subgroups— pure type and mixed type [7, 8]. Affiliations: 1Specialty Doctor, Department of Breast Sur- gery, The Royal Wolverhampton NHS Trust, Wolverhamp- The pure type is the classical type composed entirely ton, WV10 0QP, UK; 2Consultant Surgeon, Department of of mucinous carcinoma and can be further subdivided Breast Surgery, The Royal Wolverhampton NHS Trust, Wol- into cellular and hypo-cellular variants based on the verhampton, WV10 0QP, UK; 3Consultant Histopathologist, degree of cellularity. When the mucinous component is Department of Histopathology, The Royal Wolverhampton mixed with another tumor type, this is the condition of NHS Trust, Wolverhampton, WV10 0QP, UK. mixed mucinous cancer which is most commonly ductal Corresponding Author: Muhammad Asad Parvaiz, Specialty type (mucinous-ductal). Doctor, Department of Breast Surgery, The Royal Wolver- Immunohistochemistry is not generally required for the hampton NHS Trust, Wolverhampton, WV10 0QP, UK; Ph: confirmation of mucinous breast carcinoma. However, a +44-1902-695978; E-mail: [email protected] notable proportion of lesions especially the cellular variant of pure mucinous carcinoma demonstrate neuroendocrine Received: 22 January 2013 differentiation. These tumors are immunoreactive for Accepted: 04 mAY 2013 chromogranin and synaptophysin [5, 9]. Typically, Published: 01 October 2013 mucinous carcinoma is estrogen receptor positive.

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CASE REPORT DISCUSSION

A 53-year-old female was presented to the breast clinic Mucinous carcinoma of the breast usually occurs with a palpable breast lump. Clinically, the lump was in postmenopausal women. Median age of diagnosis suspicious of breast cancer. Mammogram and ultrasound is 71 years (range 25–85 years) [4, 6]. It shows more scan also confirmed the suspicious nature of the lump. favorable clinicopathological characteristics, such as A free hand needle core biopsy was carried out in the lower incidence of nodal metastasis, higher expression of one-stop triple assessment clinic on index presentation. estrogen and progesterone receptors and differentiated The core biopsy material retrieved had a distinctive grade [2, 6, 10, 11]. gross appearance, with soft, gelatinous consistency and The typical gross appearance of the mucinous breast a glistening clear almost see-through surface (Figure 1). carcinoma is a well-defined cystic mass, consisting of On this typical macroscopic appearance, a diagnosis abundant transparent to bloodstained mucin as well as of ‘mucinous breast carcinoma’ was made, which was whitish solid parts [12]. This gelatinous and glistening subsequently confirmed on microscopic histological gross appearance of mucinous breast carcinoma on needle examination. The hallmark of mucinous carcinoma is core biopsy is similar to the appearance of wallpaper extracellular mucin production, the extent of which varies paste used to stick the wallpaper to a wall. We describe from tumor to tumor. Typically, mucinous cancer cells it as ‘wallpaper paste sign’ of mucinous breast carcinoma in small clusters, sheets, or papillary configurations are on needle core biopsy because of this similarity in dispersed within pools of extracellular mucin (Figure 2). appearances. This sign has been successfully reproducible in our practice over a period of time. Whenever such needle core biopsy appearance is retrieved in the clinic, we write ‘positive wallpaper paste sign– mucinous breast carcinoma?’ on the histology request form. This has always been confirmed on the microscopic analysis and immunohistochemistry when required. Mucinous breast carcinoma is associated with a relatively favorable prognosis with a 5-year breast cancer specific survival rate of 94%. Although slowly decreasing with time, 10, 15 and 20 years survival rates are 89%, 85% and 81%, respectively compared to 82% (5 year), 72% (10 year), 66% (15 year) and 62% (20 year) for invasive ductal carcinoma [1, 6, 13–15].

CONCLUSION

Figure 1: Wallpaper paste sign of macroscopic appearance of Mucinous carcinoma is relatively rare type of breast mucinous breast carcinoma on needle core biopsy. cancer and it can be diagnosed on patient’s first visit to the breast clinic on gross inspection of the needle core biopsy specimen.

*********

Author Contributions Muhammad Asad Parvaiz – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published Brian Isgar – Substantial contributions to conception and design, Revising it critically for important intellectual content, Final approval of the version to be published Nedra Aluwihare – Substantial contributions to conception and design, Drafting the article, Revising it critically for important intellectual content, Final Figure 2: Histological appearance of mucinous breast carcinoma; approval of the version to be published lakes of lightly staining mucin with islands of neoplastic cells floating (H&E stain, x200). Guarantor

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The corresponding author is the guarantor of submission. 6. Di Saverio S, Gutierrez J, Avisar E. A retrospective review with long term follow up of 11,400 cases of Conflict of Interest pure mucinous breast carcinoma. Breast Cancer Res Authors declare no conflict of interest. Treat. 2008;111:541–547. 7. Silverberg SG, Kay S, Chitale AR, Levitt SH. Colloid carcinoma of the breast. Am J Clin Copyright Pathol.1971;55:355–363. © Muhammad Asad Parvaiz et al. 2012; This article 8. Dogan E, Aksoy S, Dizdar O, Arslan C, Dede DS, is distributed under the terms of Creative Commons Ozisik Y, Altundag K. Pure mucinous carcinoma attribution 3.0 License which permits unrestricted use, of the breast: a single center experience. J Buon. distribution and reproduction in any means provided 2011;16(3):565-7. the original authors and original publisher are properly 9. Capella C, Eusebi V, Mann B, Azzopardi JG. Endocrine credited. (Please see www.ijcasereportsandimages.com/ differentiation in mucoid carcinoma of the breast. copyright-policy.php for more information.) Histopath. 1980;4:613–30. 10. Komenaka IK, El-Tamer MB, Troxel A, Hamele-Bena D, Joseph KA, Horowitz E, Ditkoff BA, Schnabel FR. Pure mucinous carcinoma of the breast. Am J Surg. REFERENCES 2004;187:528–32. 11. Barkley CR, Ligibel JA, Wong JS, Lipsitz S, Smith 1. Louwman MW, Vriezen M, van Beek MW, Nolthenius- BL, Golshan M. Mucinous breast carcinoma: Puylaert MC, van der Sangen MJ, Roumen RM, a large contemporary series. Am J Surg. 2008 Kiemeney LA, Coebergh JW. Uncommon breast Oct;196(4):549-51. tumors in perspective: incidence, treatment 12. Chen WY, Chen CS, Chen HC, Hung YJ, Chu and survival in the Netherlands. Int J Cancer. JS. Mucinous cystadenocarcinoma of the breast 2007;121:127–135. coexisting with infiltrating ductal carcinoma. Pathol 2. Andre S, Cunha F, Bernardo M, Meneses e Sousa Int. 2004;54(10):781-6. J, Cortez F, Soares J. Mucinous carcinoma of the 13. Page DL. Special types of invasive breast breast: a pathologic study of 82 cases. J Surg Oncol. cancer, with clinical implications. Am J Surg 1995;58:162–167. Pathol.2003;27:832–835. 3. Li CI, Uribe DJ, Daling JR. Clinical characteristics 14. Park S, Koo J, Kim JH, Yang WI, Park BW, Lee KS. of different 62 histologic types of breast cancer. Br J Clinicopathological characteristics of mucinous Cancer. 2005;93:1046–1052. carcinoma of the breast in Korea: comparison with 4. Diab SG, Clark GM, Osborne CK, Libby A, Allred invasive ductal carcinoma-not otherwise specified. 92 DC, Elledge RM. Tumor characteristics and clinical J Korean Med Sci. 2010;25(3):361-8. outcome of tubular and mucinous breast carcinomas. 15. Hanagiri T, Ono K, Baba T, So T, Yamasaki M, J Clin Oncol. 1999;17:1442–1448. Nagata Y, Uramoto H, Takenoyama M, Yasumoto 5. Tan PH, Tse GM, Bay BH. Mucinous breast lesions: K. Clinicopathologic characteristics of mucinous diagnostic challenges. J Clin Pathol. 2008;61:11–19. carcinoma of the breast. Int Surg. 2010;95(2):126-9.

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CASE REPORT OPEN ACCESS Polyarticular tophaceous gouty arthritis: A case report

Sheikh Javeed Ahmad, Sumyra Khurshid

Abstract Introduction

Introduction: Gout is a disorder of purine Gout is a metabolic disorder of purine degradation metabolism, affecting men 40–50 years of age pathway usually affecting middle aged and elderly men resulting in recurrent bouts of arthritis and and postmenopausal females. Usually, it results in acute, subcutaneous tophi in patients with long standing monoarticular arthritis, intercritical period and chronic disease. We report a case of a 45-year-old tophaceous gout associated with hyperuricemia and male with symmetric, deforming polyarticular characterized by the presence of monosodium urate arthritis, affecting all the joints of hands, wrists (MSU) crystals in connective tissues and kidneys [1, 2]. and feet with diffuse subcutaneous nodules over However, some patients develop chronic polyarthritis his body. The radiographic findings of the patient mimicking rheumatoid arthritis [3–6]. Both disease were atypical. Following clinical evaluation and entities have been reported to occur in adult population additional investigations, the patient received a to the extent of approximately 1% [2, 7, 8] with symmetric diagnosis of chronic tophaceous gouty arthritis polyarthritis. Symmetrical presentation or positive mimicking rheumatoid arthritis. rheumatoid factor (RF) can be seen in both the diseases. A few cases of polyarticular tophaceous gout have been Keywords: Gout, Arthritis, Tophi, Hand, Foot reported in literature [9, 10]. We report a rare case of a 45-year-old male with polyarticular tophaceous gout ********* with atypical involvement of hand and feet with disabling effects of untreated hyperuricemia. Ahmad SJ, Khurshid S. Polyarticular tophaceous gouty arthritis: A case report. International Journal of Case Reports and Images 2013;4(10):554–558. CASE REPORT

********* A 45-year-old male (from Kashmir) was presented to our department of physical medicine and rehabilitation at doi:10.5348/ijcri-2013-10-378-CR-7 Sher-i-Kashmir Institute of Medical sciences (Srinagar) a multispecialty hospital with long standing history of pain, swelling and deformity of small and large joints of both hands and feet for approximately 12 years. The patient reported with the history of polyarthralgia which had Sheikh Javeed Ahmad1, Sumyra Khurshid2 improved over a week’s time. This was followed by frequent 1 Affiliations: Departments of Physical Medicine and intermittent episodes of arthritis of small and large joints Rehabilitation, Sher I Kashmir Institute of Medical Sciences, of hands, knees, wrists, ankles and feet without morning Srinagar, India; 2Departments of Pathology, Sher I Kashmir Institute of Medical Sciences, Srinagar, India. stiffness. The disease had been treated symptomatically Corresponding Author: Sheikh Javeed Ahmad, Departments with non-specific non-steroid anti-inflammatory drugs of Physical Medicine and Rehabilitation, Sher I Kashmir (NSAIDs) leading to the improvement over a period of Institute of Medical Sciences, Srinagar 190011, India; Email: time. Thereafter, patient developed recurrent episodes [email protected] of polyarthritis with painless nodules on hand and feet. He was put on allopurinol for 6 months and dietary restriction of protein on account of hyperuricemia Received: 26 March 2013 with serum uric acid levels of 12.43 mg/dL. Patient Accepted: 17 June 2013 intentionally interrupted the treatment following his Published: 01 October 2013 betterment of health.

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When the patient visited the outpatient department A diagnosis of polyarticular tophaceous gout was made of our clinic on first physical examination, he was and patient was treated with colchicine 0.5 mg/day and conscious, afebrile, and normotensive. Cardiovascular febuxostat 40 mg/day, which was increased to 1 mg/day and respiratory system parameters were normal. and 80 mg/day respectively till acute stage. Currently, Physical examination of Locomotor system revealed patient is showing good response with febuxostat 80 mg/ muscle atrophy of all four limbs and interossei day alone. muscles of both hands; multiple deformities of wrists, metacarpophalangeal joints (MCP), proximal interphalangeal joints (PIP) of hands (Figure 1) and metatarsophalangeal joints of feet (Figure 2). Skin examination showed subcutaneous nodule of different sizes, measuring 1–2 cm along metacarpophalangeal joints (MCP) and proximal interphalangeal joints (PIP) of hands (Figure 1) metatarsophalangeal joints feet (Figure 2). These nodules were not showing any signs of inflammation and were fixed to deeper tissues. Laboratory workup revealed hemoglobin 12.1 g/dL, Leukocyte counts of 4,300/µL, platelet counts of 150x103/ µL, ESR of 14/mm Ist hr, uric acid of 14 mg /dL, creatinine of 1.35 mg/dL, rheumatoid factor (RF) negative, blood glucose fasting of 110 mg/dL. Urinary uric acid levels were 1.19 g/24 hr and creatinine clearance of 40 mL/ min/1.73 m2, C-reactive protein 15 mg/L. X-ray of hands Figure 3: Bilateral soft tissue swelling, narrowing of space and showed narrowing of joint space, subarticular cysts at subarticular cysts. proximal interphalangeal joint of middle finger of right hand with asymmetric soft tissue swelling (Figure 3). X-ray of feet showed the evidence of narrowing of joint spaces bilaterally with subarticular cysts and overhanging edges. There is complete osteolysis of little toe of right foot (Figure 4). Histopathological examination of nodule removed from left MCP joint revealed presence of uric acid crystals (tophus) with no atypical cells (Figure 5).

Figure 1: Tophaceous nodule on metacarple and interphalangeal joint of middle figure of right hand. Figure 4: Subarticularsclerosis,cysts bilaterally and osteolysis of little toe of right foot.

DISCUSSION

Gout is an inflammatory arthropathy affecting 2.13% of population in United States of America in 2009 [11] with high preponderance for old age, male gender, postmenopausal females and black race [12]. Gout is a Figure 2: Tophaceousnodule index toe and little toe. metabolic disorder which is characterized by elevation

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seropositive. Thus polyarthritis with subcutaneous nodules with negative rheumatoid factor should be investigated for tophaceous gout [37]. Early diagnosis and initiation of early treatment of gouty arthritis will halt the progression of disease to tophaceous state. The current medical management of gouty arthritis has changed a lot. At present management of gout includes non-steroid anti- inflammatory drugs (NSAIDs), colchicines and steroids. The intraarticular steroid has been mainstay to alleviate acute gouty arthritis. It is pertinent to mention interleukin 1 beta inhibitors are the future for amelioration of gouty syndrome. Rilonacept, an interleukin 1 antagonist has proven to be of great advantage in chronic gout and has been helpful in refractory gout but randomized control trails need to be done for its effectiveness.

CONCLUSION Figure 5: Monosodium urate crystals of tophi on microscopy. Polyarticular tophaceous gouty arthritis is uncommon considering pharmacological treatment of hyperuricemia and such cases may be considered as differential diagnosis of uric acid levels above 6.8 mg/dL resulting from for rheumatoid arthritis so that early treatment will stop impaired renal uric acid excretion. High uric acid level the disability effects in such patients. can be attributed to uric acid elevating drugs, genetic predisposition and dietary factors [13]. Gout is a metabolic ********* disorder in which needle shaped crystal of monosodium urate from super saturated fluids are deposited in tissues Author Contributions resulting in gouty arthritis, tophi formation, uric acid Sheikh Javeed Ahmad – Substantial contributions to nephrolithiasis and renal impairment [14, 15]. The tophi conception and design, Acquisition of data, Analysis formation usually occurs over a mean period of 10 years and interpretation of data, Drafting the article, Revising [16]. Tophi are present commonly as subcutaneous, it critically for important intellectual content, Final sharply circumscribed nodular collection of monosodium approval of the version to be published urate crystals [17] at periarticular site in and around Sumyra Khurshid – Acquisition of data, Analysis and bursae and in soft tissue overlying tendon and cartilage interpretation of data, Drafting the article, Final approval [18–20]. The most common locations are skin overlying of the version to be published joints and helix of the ears. Rarely, these are found in eyes, nose, larynx, breast and heart valves, penis, spinal Guarantor cord, tongue, epiglottis [21–31]. Adel and Janitzia have The corresponding author is the guarantor of submission. reported location of intradermal tophi on legs, forearm, buttock, abdominal wall, palm and sole [32, 33]. Conflict of Interest The optimal serum urate level necessary for Authors declare no conflict of interest. elimination of tissue deposits of monosodium urate in patients of chronic gout is controversial. Some studies Copyright reveal the decrease in serum urate levels achieved by © Sheikh Javeed Ahmad et al. 2013; This article is urate lowering drugs and the fast reduction in tophaceous distributed under the terms of Creative Commons deposition [34]. Surgical treatment is seldom required for attribution 3.0 License which permits unrestricted use, gout and is usually reserved for cases of recurrent attacks distribution and reproduction in any means provided with deformities, severe pain and joint destruction the original authors and original publisher are properly [35]. The main indication of surgical intervention in credited. (Please see www.ijcasereportsandimages.com/ gout is sepsis or infection of ulcerated tophi followed by copyright-policy.php for more information.) mechanical problems, confirmation of diagnosis and pain control [36]. The radiologic changes in gouty arthritis are asymmetrical, erosive arthritis with preserved articular REFERENCES surface except in late cases. Bone erosions are usually caused by tophi deposits [10]. 1. Cassetta M, Gorevic PD. Crystal arthritis: gout Almost 30% of patients of rheumatoid arthritis have and pseudogout in the geriatric patient. subcutaneous nodes and all these patients are usually 2004;59(9):25–30.

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Clin Rheumatol 2005;24(1):91. associated with valvular tophi. Mayo Clin Proc 6. Reginato AJ. Gota e outras artropatias causadas 1984;59(7):509–12. por cristais. In: Kasper DL,Fauci AS, Longo DL, 26. Gawoski JM, Balogh K, Landis WJ. Aortic valvular Braunwald E, Hauser SL, Jameson JL. Harrinson. tophus: identification by X-ray diffraction of urate and Medicina Interna. v. 2, 16 ed., Rio de Janeiro: calcium phosphates. J Clin Pathol 1985;38(8):873–6. McGraw-Hill Interamericanado Brasil Ltda., 2006, 27. Ferry AP, Safir A, Melikian HE. Ocular abnormalities in pp. 2146–51. patients with gout. Ann Ophthalmol 1985;17(10):632– 7. Bachmeyer C, Charoud A, Mougeot-Martin M. 5. Rheumatoid nodulesindicating seronegative 28. Martínez-Cordero E, Barreira-Mercado E, Katona rheumatoid arthritis in a patient with gout. Clin G. Eye tophi deposition in gout. J Rheumatol Rheumatol 2003;22(2):154–5. 1986;13(2):471–3. 8. Khosla P, Gogia A, Agarwal PK, Pahuja A, Jain S, Saxena 29. Fenton P, Young S, Prutis K. 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CASE REPORT OPEN ACCESS Pulmonary artery dilatation in a young man presenting with a left mandibular fracture: Do not forget cocaine

Kohmal Ashok Solanki, Zhan Yun Lim, Andrea Pisesky, John Hogan

Abstract discuss pulmonary artery dilatation and the effects of cocaine on the pulmonary vasculature. Introduction: The use of crack cocaine in the United Kingdom has risen dramatically over Keywords: Cocaine, Pulmonary artery dilatation the last decade, leading to an increase in serious respiratory complications, which can often go ********* undiagnosed for years. Here we report a case of a 29-year-old male admitted to the emergency Solanki KA, Lim AY, Pisesky A, Hogan J. Pulmonary department with a left mandibular fracture and artery dilatation in a young man presenting with a left chest pain after a cocaine binge and subsequent mandibular fracture: Do not forget cocaine. International assault. Case Report: An abnormal chest Journal of Case Reports and Images 2013;4(10):559–562. radiograph taken as part of a trauma series led to further investigations elucidating the eventual ********* diagnosis of pulmonary artery dilatation likely secondary to cocaine. Conclusion: To doi:10.5348/ijcri-2013-10-379-CR-8 our knowledge, this is the first case reporting pulmonary artery dilatation secondary to cocaine in the absence of pulmonary hypertension. The patient was treated conservatively with outpatient right-heart catheterization and serial Introduction transthoracic echocardiography to monitor for After cannabis, cocaine is the second most used illicit emerging pulmonary hypertension. We briefly substance in Europe, with the United Kingdom having the highest prevalence of cocaine use among young adults [1]. It is known to cause numerous respiratory complications, Kohmal Ashok Solanki1, Zhan Yun Lim2, Andrea Pisesky3, usually from smoking its free-base or “crack” form [2]. John Hogan4 This case illustrates the importance of a full systemic work- 1 Affiliations: BSc MBBS, Cardiology Foundation Year 1 up of patients with a history of cocaine use, regardless of Doctor, Department of Cardiology, Whipps Cross Hospital, their presentation. To our knowledge, this is the first case London, United Kingdom; 2MBBS MRCP, Cardiology Specialty Trainee, Department of Cardiology, Whipps Cross reporting pulmonary artery dilatation secondary to cocaine Hospital, London, United Kingdom; 3MBBS, Cardiology in the absence of pulmonary hypertension [3]. Foundation Year 1 Doctor, Department of Cardiology, Whipps Cross Hospital, London, United Kingdom; 4BSC(Hons), MBChB, MD, FRCP, Consultant Cardiologist, Department CASE REPORT of Cardiology, Whipps Cross Hospital, London, United Kingdom. A 29-year-old male (by profession carpenter) Corresponding Author: Kohmal Ashok Solanki, Flat 21 presented with head injury and chest pain after found by Jenner House, Hunter Street, London, United Kingdom. the police on the street pavement following an assault. WC1N 1BL; Ph: +447749111538; E-mail: kohmalsolanki@ He reported facial and chest wall tenderness. He had yahoo.co.uk been consuming spirits, smoking cannabis and 2–3 grams of crack cocaine weekly for the last six years. He Received: 20 March 2013 had no past medical history and family history of note. Accepted: 15 May 2013 He denied any other symptoms on systematic review. On Published: 01 October 2013 examination, his Glasgow coma scale (GCS) was 15/15

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):559–562. Solanki et al. 560 www.ijcasereportsandimages.com and he was hemodynamically stable. Examination of his Pulmonary artery dilatation (PAD) is associated with cardiovascular, respiratory, abdominal and neurological congenital cardiovascular disease, systemic vasculitides, systems was unremarkable. Facial examination connective tissue disorders, infection, including trauma, demonstrated swelling and a step deformity of his left and in rare cases labeled as idiopathic. In our case, lower jaw as well as a subgaleal hematoma over his left patient denied any symptoms of chronic cardiovascular fronto-parietal region. An abnormal chest radiograph or respiratory disease nor known to have any systemic or taken as part of a trauma series necessitated medical hematological features of vasculitic or infectious disease. review. As part of the workup for the underlying aetiology, a The electrocardiogram of patient showed partial right connective tissue disease screen, TTE and a multi-slice bundle branch block that was not dynamic. His full blood CT scan of chest with contrast should be performed. A count, urea and electrolytes, liver function tests and further right-heart cardiac catheterization should also be clotting were normal. Autoantibody screen showed no considered. The differential diagnosis at this stage would evidence of connective tissue disease. The 12 hour troponin include cocaine-induced PAD, although pulmonary was also negative. Chest radiograph revealed an enlarged hypertension would need to be formally excluded by heart and widened mediastinum (Figure 1). Subsequent right-heart catheterization even with normal pulmonary two-dimensional (2D) transthoracic echocardiography artery pressures on TTE in this patient. (TTE) excluded an intra-cardiac shunt and demonstrated The patient was managed conservatively due to the a dilated pulmonary artery, normal right atrium asymptomatic long-term history and lack of evidence of (RA) and right ventricle (RV) (Figure 2A–C) and no pulmonary hypertension. The patient was discharged with significant pulmonary regurgitation (Figure 2D). There a view to formally assessing for pulmonary hypertension was no evidence of pulmonary hypertension. Computed with right-heart cardiac catheterization. The patient will tomography (CT) scan of the chest with contrast was be subsequently followed-up up with serial TTEs every organized to further visualize the mediastinum and the 6-12 months to assess if any evidence of pulmonary great arteries. This revealed a 4.5-cm dilated pulmonary hypertension develops. artery trunk (normal upper limit is 3.3 cm) (Figure 3), as well as a shallow right-sided pneumothorax and bilateral airspace opacification at the lung bases compatible with fluid or blood within the airspaces [3]. There was no evidence of an intra- or extra-cardiac shunt on multi-slice CT scan.

Figure 2: 2D echocardiography views: (A) apical four chamber view, (B) parasternal long axis view both showing a non-dilated right atrium and right ventricle, (C) parasternal short axis view demonstrating an enlarged pulmonary artery, (D) Doppler echo Figure 1: Chest radiograph demonstrating cardiomegaly, a showing no significant pulmonary regurgitation. widened mediastinum and increased pulmonary vasculature.

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preparations such as talc, silica and lactose, causing granulomatous obstruction and microembolization [5, 10]. However, pulmonary artery medial hypertrophy was also found in cocaine users without foreign particle microembolisation [11], giving further credence to the pharmacological hypothesis that cocaine itself has a direct effect on the pulmonary vasculature. Chronic cocaine use is a rare cause of PAD. To our knowledge, this is the first report to characterise this in the absence of pulmonary hypertension. Providing its course remains asymptomatic without significant changes in size of the pulmonary artery, the condition is generally considered benign and patients can undergo a prolonged period of follow-up with surgical repair if necessary. This is further evidenced by previous case reports of patients with idiopathic PAD who have had lengthy follow-up periods of up to 46 years [12]. However, in the case we Figure 3: Contrast-enhanced computed tomography of the chest have presented, continuing use of cocaine by the patient demonstrating a 4.5 cm dilated main pulmonary artery (blue may necessitate follow-up at more frequent intervals. arrow) extending into the right and left pulmonary arteries.

CONCLUSION

DISCUSSION Patients with a history of cocaine use require a full, thorough systematic work-up regardless of their Cocaine is a naturally occurring alkaloid, that when presentation. Pulmonary complications, including extracted from the leaf of the Erythroxylon coca plant pulmonary artery dilatation, can occur asymptomatically forms cocaine hydrochloride. However, due to its high in cocaine users. Pulmonary artery dilatation can occur in melting point, it is unable to be smoked in this form. the absence of pulmonary hypertension on transthoracic Cocaine hydrochloride can be transformed into its echocardiography. Asymptomatic pulmonary artery alkaloid form as either free-base (dissolved in water, dilatation can be managed conservatively with right then adding a base and a solvent) or crack (dissolved in heart cardiac catheterization and surveillance serial water with sodium bicarbonate) cocaine which is more transthoracic echocardiography. stable and thus suitable for smoking [4]. Crack cocaine reaches the cerebral circulation in less than half the time ********* taken by the intravenous route, and 30–50 times faster than by nasal insufflation [2], thus making it the route Author Contributions of choice amongst most cocaine users. Cocaine is often Kohmal Ashok Solanki – Substantial contributions to cut with adulterants including bulking agents (e.g., talc, conception and design, Acquisition of data, Drafting the lactose or mannitol), stimulants (e.g., caffeine) and local article, Critical revision of the article, Final approval of anaesthetics (e.g., lidocaine) [4], inevitably leading to the version to be published further systemic sequelae. Zhan Yun Lim – Substantial contributions to conception The pulmonary complications of either free-base and design, Acquisition of data, Drafting the article, or crack cocaine are numerous and have been well Critical revision of the article, Final approval of the documented in literature [2, 5, 6]. Cocaine mediates its version to be published local anaesthetic effects by inhibiting sodium channels in Andrea Pisesky – Substantial contributions to conception neurons, thus blocking nerve conduction. It also acts as and design, Acquisition of data, Drafting the article, Final a sympathomimetic by inhibiting central and peripheral approval of the version to be published neuronal catecholamine uptake by adrenergic receptor John Hogan – Conception and design, Drafting the stimulation [7]. Alpha -adrenoceptors in the pulmonary 1 article, Critical revision of the article, Final approval of vasculature have been implicated in cocaine-induced the version to be published pulmonary hypertension by pulmonary vasoconstriction [8]. However, intravenous cocaine hydrochloride was not Guarantor found to cause short-term increases in pulmonary artery The corresponding author is the guarantor of submission. pressure or stroke volume, suggesting that there may be other contributory pathophysiological mechanisms [9]. Conflict of Interest Significant abnormalities in the pulmonary vasculature Authors declare no conflict of interest. have also been associated with foreign additives in cocaine

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Copyright 5. Restrepo CS, Carrillo JA, Martinez S, Ojeda P, © Kohmal Ashok Solanki et al. 2013; This article is Rivera AL, Hatta A. Pulmonary complications distributed under the terms of Creative Commons from cocaine and cocaine-based substances: attribution 3.0 License which permits unrestricted use, Imaging manifestations. Radiographics 2007 Jul- Aug;27(4):941–56. distribution and reproduction in any means provided 6. Terra Filho M, Yen CC, Santos Ude P, Muñoz DR. the original authors and original publisher are properly Pulmonary alterations in cocaine users. Sao Paulo credited. (Please see www.ijcasereportsandimages.com/ Med J 2004 Jan 8;122(1):26–31. copyright-policy.php for more information.) 7. Billman GE. Cocaine: a review of its toxic actions on cardiac function. Crit Rev Toxicol 1995;25(2):113–32. 8. Salvi SS. Alpha1-adrenergic hypothesis for pulmonary REFERENCES hypertension. Chest 1999 Jun;115(6):1708–19. 9. Kleerup EC, Wong M, Marques-Magallanes 1. The EMCDDA annual report 2010: The state of the JA, Goldman MD, Tashkin DP. Acute effects of drugs problem in Europe. Euro surveillance: Bulletin intravenous cocaine on pulmonary artery pressure europeen sur les maladies transmissibles [European and cardiac index in habitual crack smokers. Chest communicable disease bulletin] 2010;15(46). 1997 Jan;111(1):30–5. 2. Haim DY, Lippmann ML, Goldberg SK, Walkenstein 10. Tomashefski JF Jr, Hirsch CS. The pulmonary MD. The pulmonary complications of crack cocaine. vascular lesions of intravenous drug abuse. Hum A comprehensive review. Chest 1995 Jan;107(1):233– Pathol 1980 Mar;11(2):133–45. 40. 11. Murray RJ, Smialek JE, Golle M, Albin RJ. Pulmonary 3. Edwards PD, Bull RK, Coulden R. CT measurement artery medial hypertrophy in cocaine users without of main pulmonary artery diameter. Br J Radiol 1998 foreign particle microembolization. Chest 1989 Oct;71(850):1018–20. Nov;96(5):1050–3. 4. Warner EA. Cocaine abuse. Ann Intern Med 1993 Aug 12. Ring NJ, Marshall AJ. Idiopathic dilatation of the 1;119(3):226–35. pulmonary artery. Br J Radiol 2002 Jun;75(894):532– 5.

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CASE REPORT OPEN ACCESS Mid-aortic dysplastic syndrome as a rare cause of hypertension in young

Kaushik Saha, Dipa Saha, Parinita Ranjit, Sujoy Sarkar, Rabi Ranjan Sow Mondal, Thiyagrajan G

Abstract of the abdominal aorta with an abrupt focal dilatation of the abdominal aorta approx 1.7 Introduction: Mid-aortic syndrome (MAS), cm below origin of superior mesenteric artery. coarctation of abdominal aorta is a rare disease Magnetic resonance angiography of the aorta and with only 200 reported cases. It is characterized by its branches showed a distinct fusiform dilatation constriction of distal thoracic and/or abdominal of abdominal aorta just below the origin of the aorta and its branches, therefore is also known superior mesenteric artery. Focal narrowing as abdominal aortic coarctation. The MAS is of abdominal aorta was noted just proximal to characterized radiologically by severe narrowing the dilatation. Conclusion: We diagnosed a case of abdominal aorta and its branches and most of mid-aortic coarctation with post stenotic of these patients usually die due to progressive dilatation associated with left renal artery severe hypertension before the age of 35–40 if stenosis leading to secondary hypertension in a left untreated. Case Report: A 13-year-old boy was teenage boy presenting with reversible posterior admitted with persistent headache and vomiting leukoencephalopathy and seizures. The boy was for one month and repeated generalized tonic referred for but the patient clonic seizures for two days. His past history refused any operative procedure. was unremarkable. The highlight of the clinical examination blood pressure was 240/150 mmHg Keywords: Mid-aortic dysplastic syndrome in both the upper limbs, and all peripheral pulses (MAS), Secondary hypertension, Posterior were palpable. Blood pressure was similar in leukoencephalopathy upper and lower limbs and a systolic bruit heard over the epigastrium. Ultrasonography showed ********* a localized narrowing of a suprarenal segment Saha K, Saha D, Ranjit P, Sarkar S, Mondal RRS, Thiyagrajan G. Mid aortic dysplastic syndrome as a rare 1 2 3 3 Kaushik Saha , Dipa Saha , Parinita Ranjit , Sujoy Sarkar , cause of hypertension in young. International Journal of 4 4 Rabi Ranjan Sow Mondal , Thiyagrajan G Case Reports and Images 2013;4(10):563–566. Affiliations: 1MBBS, DCH, MD (Medicine), Assistant Professor, Department of General Medicine, Calcutta National Medical College, Kolkata; 2MBBS, MD (Physiology), ********* Assistant Professor, Department of Physiology, College of Medicine & JNM Hospital, Kalyani, West Bengal; 3MBBS, doi:10.5348/ijcri-2013-10-380-CR-9 MD (PGT), 3rd year post graduate trainee, Department of General Medicine, Calcutta National Medical College, Kolkata; 4MBBS, MD (PGT), 2nd year post graduate trainee, Department of General Medicine, Calcutta National Medical College, Kolkata. Introduction Corresponding Author: Dr. Kaushik Saha, 1/133, M. M. Ghosh Road, Nager Bazar, Kolkata, West Bengal, India The coarctation of the abdominal aorta, also known as 700074; Ph: 009831937270, 009883080371; E-mail: middle aortic syndrome (MAS) or mid-aortic dysplastic [email protected] syndrome, is a rare vascular pathology caused by localized or extended narrowing of the abdominal or Received: 10 August 2012 distal descending thoracic aorta secondary either to a Accepted: 16 December 2012 congenital anomaly in the development of the abdominal Published: 01 October 2013 aorta or to one of several acquired conditions such as

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):563–566. Saha et al. 564 www.ijcasereportsandimages.com infection, obliterative panarteritis, neurofibromatosis, suggestive of infarcts were noted. But repeat CT scan retroperitoneal fibrosis, fibromuscular dysplasia, was normal and suggesting the diagnosis of reversible mucopolysaccharidosis and Takayasu’s arteritis [1]. Most posterior leukoencephalopathy rather than infarct. patients are young, with a mean age of 22 at diagnosis [2]. Contrast enhanced computed tomography (CECT) We report a case of a young boy admitted with headache abdomen corroborated the findings on USG and showed and seizures who was found to have this rare cause of relatively small left kidney with poor enhancement secondary hypertension. patterns, suggestive of left renal artery stenosis. A focal dilatation of the abdominal aorta was also noticed on the CT scan. Tests for ANF (HEp2 method), RA factor CASE REPORT and cANCA were all unrewarding. Magnetic resonance angiography of the aorta and its branches were A 13-year-old boy was admitted with persistent performed. This showed a normally located aortic arch headache and vomiting for one month and repeated and the ascending aorta was seen to arise from the left generalized tonic clonic seizures for two days. He had ventricle with no evidence of any abnormal dilatation no history of fever, chest pain, palpitations, claudication or flap. The arch of aorta and thoracic aorta were also or visual disturbance. Enquiry about joint pain, normal. A distinct fusiform dilatation of abdominal aorta photosensitivity, oral ulcers and recurrent infections were was seen just below the origin of the superior mesenteric not contributory. His past history was unremarkable and artery. This dilated segment had a diameter of 19 mm his parents had no reason to worry about his milestones. and the craniocaudal extension of the dilated segment The boy had good scholastic performance and had no was about 63 mm. Focal narrowing of abdominal aorta difficulty in outdoor games. There was no history of as well as left renal artery was noted just proximal to the contact with tuberculosis. Clinical assessment revealed an dilatation. The dilated segment extended below up to the alert, conscious and co-operative boy, with corroborative apparent and chronological ages. Clinical examination of the patient showed blood pressure was 240/150 mmHg in both the upper limbs, and all peripheral pulses were palpable. Blood pressure was similar in upper and lower limbs and no radio radial or radio femoral delay was appreciated. Edema was absent and neck veins were not engorged. Systemic examination was unrewarding except for a systolic bruit heard over the epigastrium, which extended along the midline up to 1 cm above the umbilicus. Ophthalmoscopy was normal as well. Counts, baseline biochemistry (sugar, urea, creatinine, LFT, lipid profile, Na, K, TSH and FT4) and ECG were all within normal limits. X-ray of chest suggested borderline left ventricular hypertrophy and echocardiography with Doppler demonstrated eccentric hypertrophy of the left ventricle (left ventricular internal diameter 4.35 cm, LVIDs 2.98 cm, LVEF 60%). Moreover, a localized Figure 1: Echo Doppler study showing narrowed aortic segment narrowing of a suprarenal segment of the abdominal with pre-stenotic dilatation. aorta with a systolic pressure gradient around 40 mmHg, systolic pressure gradient 100 mmHg in celiac axis and superior mesenteric arteries (Figure 1). Ultrasonography (USG) of abdomen with Color Doppler was done. Kidney sizes were 10.6 cm (right) and 8.5 cm (left) in the long axes. The right renal artery and interlobar arteries demonstrated normal spectral waveforms but the left renal artery could not be imaged properly. An abrupt focal dilatation of the abdominal aorta 1.7 cm (approx.) below the origin of superior mesenteric artery was observed. The dilated segment spanned 6.27 cm in the craniocaudal direction and measured 1.6 cm in diameter. A focal narrowing just before the dilatation was noted on USG (Figure 2). In view of the seizures prior to admission, contrast enhanced computed tomography (CT) of brain was performed and bilateral symmetrical non-enhancing Figure 2: Contrast enhanced computed tomography of abdomen showing small left kidney and focal dilatation of the abdominal hypodense lesions involving the parieto-occipital areas aorta.

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epistaxis, convulsions and other complication of severe hypertension are reported. Claudication and intestinal ischemia are present only in a minority of the patients, probably due to the gradual development of stenosis, which gives the body time to create effective collateral pathways [5]. From the embryological perspective, it has been suggested that a failure in fusion of the paired dorsal aortae during the fourth week of gestation may lead to MAS. Acquired conditions such as infection, obliterative panarteritis, neurofibromatosis, retroperitoneal fibrosis, fibromuscular dysplasia, mucopolysaccharidosis and Takayasu’s arteritis have been incriminated in MAS. In approximately 60% of cases, no etiology can be found. The renal arteries are involved in about 90% of the cases, and the coeliac axis and superior mesenteric artery in 35–50%, while the inferior mesenteric artery is almost never affected. A common histopathological finding in idiopathic MAS is fibroplasia of the intima and variable distortions of the internal elastic lamina with a lack of inflammatory changes that characteristically distinguish it from Takayasu’s arteritis [6]. The natural history of untreated symptomatic MAS is invariably death before the fourth decade due to complications secondary to severe hypertension such as cerebral hemorrhage or heart failure. Surgical correction remains the only definitive treatment when technically feasible. However, with newer and more effective antihypertensive drugs, surgery can be postponed until the patient reaches a more appropriate age. It is probably best to wait until full adult Figure 3: Magnetic resonance angiography of the aorta and its growth and adult vascular size are reached [7]. branches showing fusiform dilatation of abdominal aorta just below the origin of the superior mesenteric artery and focal narrowing of abdominal aorta just proximal to the dilatation. CONCLUSION

Middle aortic syndrome is a rare cause of uncontrolled division of aorta into the iliac vessels (Figure 3). The boy hypertension with poor outcome if left untreated. We was referred for vascular surgery but the patient refused diagnosed a case of mid aortic coarctation with post any operative procedure. He was put on metoprolol stenotic dilatation associated with left renal artery stenosis (50 mg) twice daily and amlodipine (10 mg) daily and has leading to secondary hypertension in a teenage boy been on irregular follow-up for the last 14 months, his last presenting with reversible posterior leukoencephalopathy blood pressure record was 160/90 mmHg. and seizures. Although we have diagnosed this case early but definitive surgical treatment was denied by patient.

DISCUSSION *********

Coarctation of the abdominal aorta or mid-aortic Author Contributions dysplastic syndrome (MAS) is an extremely rare Kaushik Saha – Conception and design, Acquisition of vascular defect in which congenital etiologies have data, Analysis and interpretation of data, Drafting the described. Typical symptoms are hypertension in article, Critical revision of the article, Final approval of young and lower limb and/or visceral ischemia, which the version to be published mostly occur later in life [3]. It is a rare condition that Dipa Saha – Acquisition of data, Analysis and accounts for 0.5–2% of all aortic narrowing, most of interpretation of data, Drafting the article, Critical which occur at the level of the proximal thoracic aorta revision of the article, Final approval of the version to be [4]. Hypertension is the cardinal clinical feature in MAS published and is present in more than 90% of the cases. Weak or Parinita Ranjit – Conception and design, Acquisition of absent femoral pulses may be appreciated and an audible data, Analysis and interpretation of data, Drafting the bruit can typically be heard over the aorta. Most often, article, Critical revision of the article, Final approval of the patient is asymptomatic, but headache, vomiting, the version to be published

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Sujoy Sarkar – Conception and design, Acquisition of REFERENCES data, Analysis and interpretation of data, Drafting the article, Critical revision of the article, Final approval of 1. Daghero F, Bueno N, Peirone A, Ochoa J, Torres the version to be published GF, Ganame J. Coarctation of the Abdominal Aorta: Rabi Ranjan Sow Mondal – Conception and design, An Uncommon Cause of Arterial Hypertension and Acquisition of data, Analysis and interpretation of data, Stroke. Circ Cardiovasc Imaging 2008;1(1):e4–6. 2. Graham LM, Zelenock GB, Erlandson EE, Coran Drafting the article, Final approval of the version to be AG, Lindenauer SM, Stanley JC. Abdominal aortic published coarctation and segmental hypoplasia. Surgery Thiyagrajan G – Conception and design, Acquisition of 1979;86(4):519–29. data, Analysis and interpretation of data, Drafting the 3. Hajsadeghi Sh, Chitsazan M, Rahbar H. Suprarenal article, Critical revision of the article, Final approval of Abdominal Aortic Coarctation Diagnosed During the version to be published Pregnancy. Iranian Cardiovascular Research Journal 2010;4(4):182–5. Guarantor 4. Connolly JE, Wilson SE, Lawrence PL, Fujitani The corresponding author is the guarantor of submission. RM. Middle aortic syndrome: Distal thoracic and abdominal coarctation, a disorder with multiple etiologies. J Am Coll Surg 2002;194(6):774–81. Conflict of Interest 5. Sethna CB, Kaplan BS, Cahill AM, Velazquez OC, Authors declare no conflict of interest. Meyers KE. Idiopathic mid-aortic syndrome in children. Pediatr Nephrol 2008;23(7):1135–42. Copyright 6. Poulias GE, Skoutas B, Doundoulakis N, et al. © Kaushik Saha et al. 2013; This article is distributed The mid-aortic dysplastic syndrome. Surgical under the terms of Creative Commons attribution 3.0 considerations with a 2 to 18 year follow-up and License which permits unrestricted use, distribution and selective histopathological study. Eur J Vasc Surg reproduction in any means provided the original authors 1990;4(1):75–82. 7. Sohn V, Herbert G, Arthurs Z, Starnes B, Andersen and original publisher are properly credited. (Please see C. Mid-aortic syndrome and renovascular www.ijcasereportsandimages.com/copyright-policy.php hypertension in a 14-year-old Iraqi girl: Pitfalls in for more information.) diagnosis and surgical management. Ann Vasc Surg 2007;21(5):648–51.

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CASE REPORT OPEN ACCESS An unusual case of a misplaced left internal jugular vein catheter

Sunil Rangarajan, Sunad Rangarajan, Lindsey Smith Hinton

Abstract in her left IJV under ultrasound guidance. The catheter was inserted without any perceived Introduction: Most often, cannulation of the right resistance. Blood withdrawn from the ports was internal jugular vein (IJV) is preferred over the however, bright red, and gas analysis revealed left IJV. However, in situations where the right arterial blood values. A bed side X-ray revealed IJV cannot be utilized for accessing the central that the catheter was in the heart. Conclusion: circulation, as in our case, the left IJV is used. This is the first reported case where the tip of the Cannulation of left IJV has additional risks due cannula was in the left atrium. Despite the use of to the anatomical variations. Despite the use of ultrasound guidance, the anatomical variations ultrasound guidance, anatomical variations and and the tortuous course of the left internal tortuous course of the left IJV make cannulation jugular vein make its cannulation more prone of the left IJV more prone to the failure of to failure or malposition of the cannula leading cannulation or malposition of the cannula leading sometimes to catastrophic complications. This to catastrophic complications. Case Report: A case reinforces studies which have shown that 63-year-old female with multiple comorbidities even with ultrasound guidance, left internal presented with progressive shortness of breath jugular vein cannulation is fraught with higher and mild respiratory distress due to bibasilar risk of complications. pneumonia. Despite aggressive management of her pneumonia, she continued to deteriorate and Keywords: Central vein cannulation, Internal became increasingly hypoxemic, hypotensive jugular vein (IJV) with abnormal renal functions necessitating medical intensive care treatment and continuous ********* hemodialysis. As she had a chemo-port in her right subclavian vein, it was determined to avoid Rangarajan S, Rangarajan S, Hinton LS. An unusual insertion of the dialysis catheter in her right case of a misplaced left internal jugular vein catheter. IJV. An attempt was made to insert the catheter International Journal of Case Reports and Images 2013;4(10):567–570.

Sunil Rangarajan1, Sunad Rangarajan2, Lindsey Smith ********* Hinton3 1 Affiliations: MBBS, Postdoctoral Scholar, University of doi:10.5348/ijcri-2013-10-381-CR-10 Alabama at Birmingham, Birmingham, AL; 2MD, Fellow, Pulmonary and Critical Care, University of Alabama at Birmingham, Birmingham, AL; 3MD, Resident, Internal Medicine, University of Alabama at Birmingham, Birmingham, AL. Corresponding Author: Sunil Rangarajan, Postdoctoral Introduction Scholar, University of Alabama at Birmingham, 1720, 2nd Avenue South, LHRB 452, Birmingham, Alabama – 35294; Internal jugular veins (IJVs) are often used for an Ph: 205-934-5783; Fax: 205-975-6288; Email : sunilr@uab. access into the central circulation. The right IJV provides edu a straight path to the heart. The anatomical variations that are often present in the left IJV pose a challenge in accessing the central circulation. There are very few Received: 27 October 2012 prospective studies to compare the safety of inserting Accepted: 18 February 2012 Published: 01 October 2013 the catheter into the right against the left IJV [1]. We

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):567–570. Rangarajan et al. 568 www.ijcasereportsandimages.com present a case of a misplaced catheter, which perforated the left IJV into the mediastinum, the bifurcation of the pulmonary artery, and the left atrium.

CASE REPORT

A 63-year-old female with history of diabetes mellitus, hypertension, atrial fibrillation, colon cancer managed by surgical resection and chemotherapy presented with progressive shortness of breath. She had recently undergone cholecystectomy and then splenectomy elsewhere for pancytopenia which had subsequently been attributed to myelodysplastic syndrome. Clinical examination revealed a middle aged woman in mild respiratory distress with significant systemic exam findings being scattered crackles and irregular heart rate. Chest roentgenogram suggested bibasilar pneumonia. As she became increasingly hypoxemic, hypotensive and short of breath, she was admitted to the medical intensive care unit for Figure 1: Scout image showing aberrant downward course of the the management of septic shock and respiratory catheter on the left side of the mediastinum with its tip in the failure from acute respiratory distress syndrome left atrium (yellow arrow). due to pneumonia. Despite aggressive treatment she continued to deteriorate and her renal function worsened, necessitating continuous hemodialysis. As she had a chemo-port in her right subclavian vein, it was determined to avoid insertion of the dialysis catheter in her right internal jugular vein (IJV). An attempt was made to insert the catheter by Seldinger technique in her left IJV under ultrasound guidance. The catheter was inserted without any perceived resistance. Blood that returned from the ports was however, bright red, and gas analysis revealed arterial blood values. The screening chest X-ray showed a possible malposition of the catheter. Further, a computed tomography (CT) scan scout image (Figure 1) showed an aberrant downward Figure 2: (A) Catheter (yellow arrow) seen puncturing the left course of the catheter on the left side of the mediastinum internal jugular vein (blue arrow) into the mediastinum, (B) with its tip in the cardiac area. Further imaging revealed Catheter (yellow arrow) seen inside the pulmonary artery at its the catheter to have punctured the left IJV (Figure 2A), bifurcation (green arrow). into the mediastinum lateral to the aortic arch missing it narrowly, the main pulmonary artery at its bifurcation (Figure 2B) and the left atrium of the heart (Figure 3). The patient was unstable for sternotomy to remove the In addition, the left IJV has several anatomical variations. catheter. Her overall condition worsened further and as Occasionally, people may have a normal variant of the left per family’s wishes, care was withdrawn and she expired. IJV that drains into the left superior vena cava with or without a bridging vein. The overlap of the IJV over the DISCUSSION carotid artery is more on the left when the head rotation is greater than 30 degrees [2, 3]. One should be cognizant of this difference while inserting a catheter into the left Most often, cannulation of the right IJV is preferred IJV. It is observed that in more than one-third of the over the left IJV. However, in situations where the right normal population, left IJV is half the size of the right IJV IJV cannot be utilized for accessing the central circulation making the procedure more difficult [4, 5]. as in our case, the left IJV is used. The anatomy of the left Cannulation of the IJV can be done either using the IJV makes a central venous line pass through two near- landmark approach (using point of needle insertion as 90-degree turns, the first at the junction of the left IJV and the apex of the triangle formed by the two heads of the the left subclavian vein, and the second at the junction of sternocleidomastoid muscle with the base as the clavicle) the left brachiocephalic vein and the superior vena cava. or the ultrasound guided approach. The risk of a serious

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cannulation is fraught with higher risk of complications than on the right side. In situations where the right internal jugular vein cannot be catheterized, extra caution has to be taken while attempting to obtain venous access through the left internal jugular vein.

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Author Contributions Sunil Rangarajan – Conception and design, Acquisition of data, Drafting of the article, critical revision of the article, Final approval of the version to be published Sunad Rangarajan – Conception and design, acquisition of data, Drafting of the article, Critical revision of the article, Final approval of the version to be published Lindsey Smith Hinton – Acquisition of data, Drafting of the article, Final approval of the version to be published

Guarantor The corresponding author is the guarantor of submission.

Conflict of Interest Figure 3: Computed tomography of the chest: Catheter (yellow Authors declare no conflict of interest. arrow) noted millimeters from puncturing the aorta (red arrow), traversing the pulmonary artery (green arrow), and entering the left atrium (white arrow). Copyright © Sunil Rangarajan et al. 2013; This article is distributed under the terms of Creative Commons attribution 3.0 License which permits unrestricted use, distribution and complication while performing the landmark approach is reproduction in any means provided the original authors much higher. In a prospective study, the most dangerous and original publisher are properly credited. (Please see complications which occurred in patients who were www.ijcasereportsandimages.com/copyright-policy.php catheterized using the landmark approach included for more information.) puncture of the carotid artery occurred in 10.6%, hematoma in 8.4%, hemothorax in 1.7%, pneumothorax in 2.4% and central venous catheter-associated blood REFERENCES stream infection in 16% [6]. The success rate of inserting the cannula was higher in the ultrasound guided approach 1. Sulek CA, Blas ML, Lobato EB. A randomized study of than in the landmark approach. Muralidhar reported a left versus right internal jugular vein cannulation in 19% incidence of failure or cannula malposition while adults. J Clin Anesth 2000 Mar;12(2):142–5. 2. Lorchirachoonkul T, Ti LK, Manohara S, et al. attempting on the left IJV compared to 3% with the right Anatomical variations of the internal jugular vein: IJV, using the landmark technique [7]. implications for successful cannulation and risk In the present day, the use of ultrasound guidance of carotid artery puncture. Singapore Med J 2012 has become ubiquitous while inserting a cannula into the May;53(5):325–8. IJV. Despite the use of ultrasound guidance, the above 3. Lieberman JA, Williams KA, Rosenberg AL. Optimal mentioned factors make cannulation of the left IJV more head rotation for internal jugular vein cannulation prone to failure of cannulation or malposition of the when relying on external landmarks. Anesth Analg cannula leading to catastrophic complications [8–10]. 2004 Oct;99(4):982–8. There are a handful of cases reported in the medical 4. McGee WT, Mallory DL. Cannulation of the internal and external jugular veins. Prob Crit Care literature where the catheterization of the left IJV has 1988;2:217–41. caused a cardiac tamponade [11, 12]. This is the first 5. Lobato EB, Sulek CA, Moody RL, Morey TE. reported case where the cannula has finally landed inside Cross-sectional area of the right and left internal the left atrium without cardiac tamponade. jugular veins. J Cardiothorac Vasc Anesth 1999 Apr;13(2):136–8. 6. Karakitsos D, Labropoulos N, De Groot E, et al. CONCLUSION Real-time ultrasound-guided catheterisation of the internal jugular vein: a prospective comparison with This case reinforces studies which have shown that the landmark technique in critical care patients. Crit Care 2006;10(6):R162. even with ultrasound guidance, left internal jugular vein

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7. Muralidhar K. Left internal versus right internal 10. Ghosh S, Dewan H, Bhattacharyya S. A rare jugular vein access to central venous circulation using malposition of the thoracic venous catheter introduced the Seldinger technique. J Cardiothorac Vasc Anesth via the left internal jugular vein. Indian J Crit Care 1995 Feb;9(1):115–6. Med 2008 Oct;12(4):201–3. 8. Lee EK. An unexpected left hydrothorax after left 11. van Haeften TW, van Pampus EC, Boot H, Strack internal jugular venous catheterisation for total van Schijndel RJ, Thijs LG. Cardiac tamponade from parental nutrition and antibiotics. Ann Acad Med misplaced central venous line in pericardiophrenic Singapore 2006 Oct;35(10):742–4. vein. Arch Intern Med 1988 Jul;148(7):1649–50. 9. Khajavi MR, Sedighi M. Malposition of central venous 12. Al-Azawi O, Shehab R, Ababneh MO. Cardiac catheter in the left internal jugular vein--a case report. temponade following left internal jugular venous Middle East J Anesthesiol 2006 Oct;18(6):1157–60. catheterization--a case report. Middle East J Anesthesiol 2006 Oct;18(6):1161–4.

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CASE REPORT OPEN ACCESS Retroperitoneal neurilemmoma with cystic degeneration mimicking hydatid cyst

Manash Ranjan Sahoo, Anil Kumar T

Abstract sectional imaging alone. Conclusion: A retroperitoneal schwannoma is a rare disorder Introduction: Retroperitoneal neurilemmoma and most cases are benign for which enucleation is a common tumor in an uncommon place. is curative. Secondary changes can misdiagnose If secondary changes occur in the tumor, it the cases both clinically and radiologically and becomes very difficult to diagnose by clinical and one should be aware of it. radiological examination. In this case report, we have shown how a neurilemmoma with cystic Keywords: Retroperitoneum, Hydatid cyst, degeneration was misdiagnosed as hydatid cyst Neurilemmoma, Cystic degeneration till it was confirmed by histopathology. Case Report: A 30-year-old female presented with ********* pain on right side of abdomen since one month. Clinical examination revealed firm mass of Sahoo MR, Anil Kumar T. Retroperitoneal 8x10 cm in right hypochondriac and lumbar neurilemmoma with cystic degeneration mimicking region. Radiological examination showed it as hydatid cyst. International Journal of Case Reports and complex cystic lesion with multiple membrane Images 2013;4(10):571–574. probably hydatid cyst. With preoperative diagnosis as hydatid cyst laparoscopy revealed ********* cystic lesion with degenerated cheesy tissue then by opening the abdomen by sub costal incision doi:10.5348/ijcri-2013-10-382-CR-11 deroofing of the cyst was done. Histopathological examination confirmed it as neurilemmoma. Schwannomas have true capsules composed of epineurium. Although target and fascicular signs are characteristic radiological features of Introduction schwannoma, these are not frequently seen in Schwannomas are nerve sheath tumors, usually retroperitoneal schwannomas. In the absence found in the head, neck and on the flexor surfaces of the of typical signs, diagnosing a retroperitoneal extremities. They are believed to be benign tumors with schwannoma is difficult when using cross- female predominance occurring between 2–5 decade [1, 2], with few cases of malignant transformation reported. Schwannomas are rarely found in the retroperitoneal Manash Ranjan Sahoo1, Anil Kumar T2 Affiliations: 1MS, Associate Professor, Department of cavity [3, 4], only about 0.3–3.2% [7]. Pathologically, it Surgery, S.C.B. Medical College, Cuttack, Odisha, India; is a well-encapsulated lesion [5] demonstrating specific 2Post Graduate, Department of Surgery, S.C.B. Medical Antoni A/B areas. Characteristic immunochemical College, Cuttack, Odisha, India. features are a positive S-100 [6] and a negative CD-34. In Corresponding Author: Dr. Manash Ranjan Sahoo, Mailing this report, we aimed to illustrate how cystic degenerated Address: Orissa Nursing Home, Medical road, Ranihat, neurilemmoma was misdiagnosed hydatid cyst based on Cuttack, Odisha, India - 753007; Ph: +919937025779; Fax: radiological imaging. 0671-2414034; Email: [email protected]

Received: 27 October 2012 CASE REPORT Accepted: 18 February 2012 Published: 01 October 2013 A 30-year-old female presented with pain on right side of abdomen since a month. Clinical examination revealed

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):571–574. Sahoo et al. 572 www.ijcasereportsandimages.com a firm mass of 8x10 cm in the right hypochondrium and lumbar region. Routine hemogram was normal and liver function test was normal. Ultrasound revealed well defined heterogeneous lesion with multiple membrane, solid cystic portion with internal echoes, of size 12.4x7 cm in sub hepatic space probably hydatid cyst. Computed tomography (CT) scan revealed a large well defined circumscribed oval complex cystic mass lesion in right hypochondrium and lumbar region abutting right lobe of liver and right kidney (Figure 1). On the operation table laparoscopy showed cystic mass (Figure 2). With preoperative diagnosis of hydatid cyst, aspiration revealed straw colour fluid at the beginning with cheesy material with hydatid cannula (Figure 3). Deroofing of the cyst was done (Figure 4). Later frank blood started coming which could not be controlled Figure 3: Suction of contents of the cyst using hydatid cannula. (Figure 5), so abdomen was opened by right sub costal incision. Whole of the cyst cannot be removed since its close proximity to inferior vena cava and duodenum, however, most of the cyst wall was removed. Hemostasis

Figure 4: Deroofing of the cyst.

Figure 1: Computed tomography scan showing retroperitoneal cyst with multiple septation may be hydatid cyst abutting liver.

Figure 5: Oozing started from the redundant cyst, hence Figure 2: Intracorporeal laparoscopic view of cyst. converted to open method.

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):571–574. Sahoo et al. 573 www.ijcasereportsandimages.com achieved by placing a ribbon pack which was removed followed-up for a period of eight months which revealed three days postoperatively. Postoperative period was no recurrence of disease which might have been due to uneventful. degeneration that had already set in. A week later histopathological examination came out as benign neurilemmoma. No recurrence of the cyst or no symptoms were noted after following-up patient for a CONCLUSION period of eight months. Retroperitoneal schwannoma is a rare disorder and most cases are benign for which enucleation is curative. DISCUSSION Secondary changes can misdiagnose the cases both clinically and radiologically and one should be aware of it. Schwannomas (neurilemmoma) are nerve sheath tumors that usually affect the head, neck and the flexor ********* surfaces of the extremities. It is rare to find a schwannoma in the retroperitoneal cavity [3, 4] accounts for 0.3–3.2% Author Contributions of schwannomas [7]. They usually affect adult patients Manash Ranjan Sahoo – Conception and design, aged 20–50 years and females are more frequently Acquisition of data, Analysis and interpretation of data, affected than males [1, 2]. Drafting the article, Final approval of the version to be Schwannomas have true capsules composed of published epineurium [5]. The tumor mass is characteristically Anil Kumar T – Conception and design, Acquisition of eccentric with respect to the affected nerve. Retroperitoneal data, Analysis and interpretation of data, Drafting the schwannomas show cystic degeneration in up to 60% of article, Critical revision of the article, Final approval of cases while calcification is seen in 23% of cases only. These the version to be published changes make diagnosis even more difficult. Although target and fascicular signs are characteristic radiological Guarantor features of schwannoma, these are not frequently seen The corresponding author is the guarantor of submission. in retroperitoneal schwannomas. In the absence of typical signs, diagnosing a retroperitoneal schwannoma Conflict of Interest is difficult when using cross-sectional imaging alone. Authors declare no conflict of interest. Computed tomography and ultrasonography should be used to guide tissue biopsies and identify the aggressive Copyright features of a malignant schwannoma, which account for © Manash Ranjan Sahoo et al. 2013; This article is about 1% of retroperitoneal schwannomas. Schwannomas distributed under the terms of Creative Commons are encapsulated tumors [5]. Microscopically, they attribution 3.0 License which permits unrestricted use, demonstrate Antoni A areas (densely cellular, arranged distribution and reproduction in any means provided in short bundles or interlacing fascicles) and Antoni the original authors and original publisher are properly B areas (fewer cells, organized, with great myxoid credited. (Please see www.ijcasereportsandimages.com/ component). They are positive for S-100 [6] and negative copyright-policy.php for more information.) for CD-34. Detecting a malignant retroperitoneal schwannoma is crucial for effective management as these carry a much poorer prognosis. Its clinical, radiological, REFERENCES and histological features usually clinch the diagnosis. Radiologically, a malignant schwannoma has irregular 1. Enzinger FM, Weiss SM. Soft tissue tumors, St. Louis: margins and infiltrates the adjacent structures. Distant Mosby, 1983: 5–7. metastases via perineural and intra-neural routes are 2. Schindler SO, Dixon JH, Case P. Retroperitoneal giant characteristic features. Histologically, an infiltrative schwannomas: Report of two cases and review of the literasture. J Orthopeadic Surg. 2002; 10: 77–84 margin with nuclear palisading is a striking feature. In 3. Maleux G, Brys P. Sampson I, Sciot R, Baert AL. terms of treatment, surgery is the modality of choice Giant schwannoma of the lower leg. Eur. Radio. 1997; for retroperitoneal schwannomas. Chemotherapy and 7:1031–34. radiotherapy have only limited roles. In malignant 4. Gubbay AD, Moschille G, Gray BN, Thompson I. schwannomas, adjuvant chemotherapy or radiotherapy Retroperitoneal schwannoma: A case series and has marginal added benefit; some authors advocate review. ANZ J Surg. 1995; 65: 197–200. induction chemotherapy for this condition. Our 5. Guz BV, Wood DP Jr, Montie JE, Pontes JE. patient had benign neurilemmoma with already cystic Retroperitoneal nerve sheath tumors: Cleveland degeneration which could not be removed fully due to clinic experience. J Urol. 1989; 142: 1434–7. 6. Harkin I. Pathology of nerve sheath tumors. Ann NY severe hemorrhage and close proximity to duodenum and Acad Sci. 1986; 486: 147–54. inferior vena cava. Postoperatively patient was closely

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7. Melicow MM. Primary tumors of the retroperitoneum: A clinicopathologic analysis of 162 cases: Review of the literature and tables of classification. J Int Coll Surg 1953; 19: 401–49.

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CASE REPORT OPEN ACCESS Frank hematuria as sole manifestation of acute myeloid leukemia: A case report

ML Patel, Rekha Sachan, Apul Goel

Abstract International Journal of Case Reports and Images 2013;4(10):575–577. Introduction: Unexplained hematuria may be presenting feature of various systemic illnesses ********* such as coagulation disorder, anticoagulation therapy, leukemia, disseminated intravascular doi:10.5348/ijcri-2013-10-383-CR-12 coagulation and multiorgan dysfunction. Bleeding is a common presenting feature of acute leukemia. This may occur in any mucosal surface of body. Case Report: A 52-year-old male presented with complaints of low grade fever, Introduction and gross hematuria for last three days. He was diagnosed as a case of acute myeloid leukemia Usually, frank hematuria is a clinical manifestation of on the basis of bone marrow aspiration and various systemic illnesses such as coagulation disorders, cytochemistry. Conclusion: Presentation of acute anticoagulation therapy, leukemia, disseminated leukemia with gross hematuria is uncommon. intravascular coagulation and multiorgan dysfunction Gross hematuria as the sole presenting feature (severe liver function derangement) [1]. Hemorrhagic in such cases is unusual and only few cases have cystitis due to viral infection like Adeno virus, BK/JC been described in literature. virus reported in immunocompromised patients [2]. Acute leukemia particularly acute myeloid leukemia can Keywords: Frank hematuria, Acute myeloid result in leukemic infiltration of many organs including leukemia, Chemotherapy, Cystoscopy. urinary tract resulting in manifestations like hematuria [1]. Though leukemic infiltration of urinary tract is ********* clinically not evident but autopsy studies have been shown involvement of urinary system [3]. Gross hematuria is an ML Patel, Sachan R, Goel A. Frank hematuria as sole uncommon presentation of acute leukemia. Bleeding in manifestation of acute myeloid leukemia: A case report. acute leukemias usually results from thrombocytopenia, involving the skin, mucous membrane and may or may not correlate with the degree of thrombocytopenia as platelet dysfunction may also be present. Another cause ML Patel1, Rekha Sachan2, Apul Goel3 Affiliations: 1MD (Medicine), Assistant Professor, Department of bleeding in some patients with acute myeloid leukemia of Medicine, CSM Medical University, Lucknow; 2MS (Obs & is coagulopathy due to disseminated intravascular Gynae), Associate Professor, Department of and coagulation (DIC), which is commonly seen in patients , CSM Medical University, Lucknow; 3MS, MCh with acute promyelocytic leukemia [4]. (), Professor, Department of Urology, CSM Medical University, Lucknow. Corresponding Author: Dr. ML Patel, MD, Assistant CASE REPORT Professor, Department of Medicine, CSM Medical University, Lucknow-226003, Uttar Pradesh, India; Tel: +919839007000; A 52-year-old male patient was admitted in Department Email: [email protected] of Medicine, Chhatrapati Shahuji Maharaj Medical University, (Erstwhile KGMC) Lucknow in January 2011 Received: 21 January 2012 with complaints of low grade fever, and gross hematuria Accepted: 06 May 2012 for last three days. On general examination patient was Published: 01 October 2013 febrile 100.6°F, blood pressure 130/80 mmHg, pulse

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86/min, mild pallor present and no signs of dehydration anticoagulation therapy, leukemia, disseminated was present. General examination revealed there was intravascular coagulation and multiorgan dysfunction no lymphadenopathy, icterus, edema, hemorrhagic spot (severe liver function derangement). or bleeding from any other site on the body. Systemic Hematuria as the only or main presenting feature in examination was within normal limit no palpable lump acute leukemia is rare and to best of our knowledge only 17 was present in abdomen. There was no history of intake of cases have been reported till now [5, 6]. In this case blood medication such as anticoagulation therapy, antiplatelet investigation reports were suggestive of acute leukemia. drug, nonsteroidal anti-inflammatory drug or steroid in Although any organ system in the body can be infiltrated the past. by leukemic cells but involvement of renal system is On routine investigation hemoglobin 7.8 g/dL, unusual. If the urinary tract is infiltrated by leukemic leukocyte count 20.6x103/µL, MCV 90 fL, MCH 30 pg, and cells, kidney is the most common site of involvement [5, platelet count was 60.0x103/µL. General blood picture 7]. Leukemic infiltration of the urinary bladder is very revealed 22% blast cells with auer rods. Liver function tests rare, till date only few cases have been reported [5–8]. and coagulation profile were normal. Serum creatinine In this case though cystoscopy was normal but it could was 1.2 mg/dL and blood urea was 24 mg/dL. Mid not ruled out occult leukemic infiltration, this could be stream urine examination showed protein in traces, 2–4 confirmed only by biopsy but patient refused for the pus cells/high power field, and numerous red blood cells same. along with amorphous deposit of crystals. Urine culture Although clinically evident involvement of the and sensitivity were sterile, there was no evidence of urinary system and hematuria as the presenting glomerulonephritis. FDP and D-dimer were normal. symptom is rare, leukemic infiltration was observed in Patient refused PCR test for detection of viral etiology. more than 50% of cases in an autopsy study [7]. Our Ultrasonography of kidney and computed tomography case was unusual, because patient initially presented (CT) scan of abdomen revealed no abnormality of urinary with gross hematuria, later on diagnosed as a case of tract region. Cystoscopic examination of the bladder acute myeloid leukemia on the basis of bone marrow was normal (Figure 1). The bone marrow aspirate and aspiration and cytochemistry [9]. However, malignancy cytochemistry were consistent with the diagnosis of of kidney or urinary bladder could be another possible acute myeloid leukemia of M4FAB subtype. The patient cause of hematuria, as per review based on the articles was kept on induction chemotherapy (daunorubicin 50 mentioned bladder cancer usually causes no frank mg/m2/day on day 1, 2, 3 and cytarabine 150 mg/m2/ hematuria until it reaches an advance stage. Mainly day, intravenous infusion for seven days) and achieved large exophytic urinary bladder carcinoma causes frank remission. Hematuria subsided within three days after hematuria. In our reported case patient who initially treatment. presented with gross hematuria, diagnosed as case of systemic disease (acute myeloid leukemia). In this case since hematuria did not recur after the treatment with chemotherapy for acute myeloid leukemia despite the drop in platelet count to very low level, the possibility of occult leukemic infiltration of urinary tract appears to be the likely cause of hematuria. The disappearance of hematuria also suggests that disease responded with the treatment and this hematuria might be due to acute myeloid leukemia. Hemorrhagic cystitis due to viral infection like Adeno virus, BK/JC virus usually occurs between 15– 60 days following bone marrow transplantation and within the first three months of kidney transplantation in immunocompromised patients [9, 10]. Our patient was not immunocompromised so the possibility of hemorrhagic cystitis was ruled out [11].

Figure 1: Cystoscopy of urinary bladder of patient showing normal healthy wall and ureteric orifices. CONCLUSION

There are so many causes of hematuria such as viral hemorrhagic cystitis, kidney or urinary bladder DISCUSSION malignancy and systemic illness. When patient presented with hematuria as the main symptom, acute leukemia Frank hematuria is a clinical manifestation of should be kept in mind. various systemic illnesses such as coagulation disorders,

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********* 1972;107(6):1073–7. 2. David Ghez, Eric Oksenhendler, Catherine Scieux, Acknowledgements Kaiss Lassoued. Haemorrhagic cystitis associated with Adenovirus in a patient with AIDS treated for We acknowledge Dr. Ravi Uniyal, Senior Resident, a non-Hodgkin’s Lymphoma. American journal of Department of Medicine, CSMMU, Lucknow for helping 2000;63(1):32–4. in collection of data and investigation. 3. Vidana E, Bross ID, Pricken JW. An autopsy study of metastatic patterns of human leukemia. Author Contributions 1978;35(2):87–96. ML Patel – Substantial contributions to conception and 4. Tallman MS, Kwaan HC. Reassessing the hemostatic design, Acquisition of data, Analysis and interpretation disorder associated with acute promyelocytic leukemia. Blood 1992 Feb 1;79(3):543–53. of data, Drafting the article, Revising it critically for 5. Watson EM, Sauer HR, Sadugor MG. Manifestation important intellectual content, Final approval of the of the lymphoblastomas in the genitor-urinary tract. J version to be published Urol 1949;61(3):626–45. Rekha Sachan – Acquisition of data, Revising it critically 6. Chang CY, Chiou TJ, Hsieh YL, Cheng SN. Leukemic for important intellectual content, Final approval of the infiltration of the urinary bladder presenting as version to be published uncontrollable gross hematuria in a child with acute Apul Goel – Acquisition of data, Revising it critically lymphoblastic leukemia. J Pediatr Hematol Oncol for important intellectual content, Final approval of the 2003 Sep;25(9):735–9. version to be published 7. Kirshbaum JD, Preuss FS. Leukemia: a clinical and pathologic study of one hundred and twenty-three fatal cases in a series of 14400 necropsies. Arch Intern Guarantor Med 1943;71:777. The corresponding author is the guarantor of submission. 8. Pentecost CJ, Pizzolato P. involvement of the genitourinary tract in Leukemia. J Urol 1945;53:725. Conflict of Interest 9. Owais M. Suriya, Aamer Aleem. Frank Hematuria as Authors declare no conflict of interest. the Presentation feature of Acute leukemia. Saudi J Kidney Dis Transpl 2010;21(5):940–2. Copyright 10. Koga S, Shindo K, Matsuya F, Hori T, Kanda S, © ML Patel et al. 2013; This article is distributed under Kanetake H. Acute hemorrhagic cystitis caused by adenovirus following renal transplantation: Review of the terms of Creative Commons attribution 3.0 License the literature. J Urol 1993;149(4):838–9. which permits unrestricted use, distribution and 11. Miyamura K, Takeyama K, Kojima S, et al. reproduction in any means provided the original authors Hemorrhagic cystitis associated with urinary and original publisher are properly credited. (Please see excretion of adenovirus type 11 following allogeneic www.ijcasereportsandimages.com/copyright-policy.php bone marrow transplantation. Bone Marrow for more information.) Transplant 1989;4(5):533–5.

REFERENCES

1. Persky L, Newman AJ, Tucker AS. Urologic manifestations of childhood Leukemia. J Urol

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CASE REPORT OPEN ACCESS Spontaneous regression of lumbar disc herniation: Conservative treatment in a case with motor deficit

Saliha Eroğlu Demir, Nihal Özaras, Ebru Aytekin

Abstract *********

Introduction: Conservative healing of lumbar Demir SE, Özaras N, Aytekin E. Spontaneous regression disc herniation (LDH) exists. Most surgical of lumbar disc herniation: Conservative treatment in a studies have followed a minimum six-week case with motor deficit. International Journal of Case trial of conservative therapy before surgical Reports and Images 2013;4(10):578–581. intervention. Patients who do not present with emergency surgery indications may be treated ********* initially with conservative methods. Case Report: A case of 32-year-old female with motor deficit doi:10.5348/ijcri-2013-10-384-CR-13 caused by an extruded herniated disc. Physical and medical therapy was given because she refused to undergo surgery. After the treatment, her motor deficits improved. Magnetic resonance imaging scan showed totally regression of the Introduction extruded disc. Conclusion: Although a single case cannot confirm the validity of conservative Lumbar disc herniation (LDH) is a common health management of LDH with motor deficits, patient problem. There have been some reports on spontaneous preference and severity of the disability from pain regression of LDH at different levels and with various are important factors when choosing treatment clinical presentations such as myelopathy, neurological modalities. deficits, lumbar radiculopathy ascertained by magnetic resonance imaging (MRI) scan or computed tomography Keywords: Intervertebral disk displacement, (CT) scan [1–9]. Since spontaneous resorption of LDH Lumbosacral region, Low back pain, Neurologic is well-known, most patients including with neurologic manifestations, modalities. deficits are reluctant to disc surgery. A case of spontaneous regression of extruded disc herniation with severe motor Saliha Eroğlu Demir1, Nihal Özaras2, Ebru Aytekin3 deficit (manual muscle test ≤3) is rare. In this report, Affiliations: 1MD, Associated Professor, Physical medicine totally regression of herniated material in a case with and Rehabilitation Specialist, Bezmialem Vakif University, severe motor deficit caused by an extruded herniated disc Physical Medicine and Rehabilitation Department, at the L4-L5 level is presented. Istanbul, Turkey; 2MD, Associated Professor, Bezmialem Vakif University, Physical Medicine and Rehabilitation Department, Istanbul, Turkey; 3MD, Physical medicine and Rehabilitation Specialist, Ministry of Health, Istanbul CASE REPORT Research and Training Hospital, Physical Medicine and Rehabilitation Clinic, Istanbul, Turkey. A 32-year-old female presented for the physical Corresponding Author: Saliha Eroglu Demir, MD, Physical medicine and rehabilitation department in March Medicine and Rehabilitation Department Bezmialem Vakif 2008 with a two-month history of low back and right University Medical Faculty, Department of Physical Medicine leg radiating pain. The patient’s history was notable for and Rehabilitation, Istanbul, Turkey; Tel: +90 212 523 37 19; intermittent low back pain over the previous nine months Fax: +90 212 533 23 26; Email: [email protected] after normal vaginal delivery in July 2007. Her pain had started to radiate to right leg after she lifted her baby two months ago. The patient was able to ambulate in sideways Received: 03 August 2012 bending position. Neurological examination showed a Accepted: 07 December 2012 Published: 01 October 2013 positive straight-leg raising test (SLRT) at 40° on the right

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):578–581. Demir et al. 579 www.ijcasereportsandimages.com side. The SLRT was negative on the left side. On manual After 18 months, the patient was still complaining muscle test, muscle weakness was found in the right leg: intermittent right leg radiating pain. The SLRT was tibialis anterior was 3/5, extensor hallucis longus was 2/5. negative and there was weakness of extensor hallucis The right patella tendon reflex was normal. Hypoesthesia longus muscle (4/5). Hypoesthesia was noted over the was noted over the medial calf and dorsum of the right dorsum of the right foot. A second lumbosacral MRI foot. Magnetic resonance imaging (MRI) scan showed scan was done at this time. It revealed total regression large extruded disc herniation on the right side at the L4– of the extruded disc fragment at the L4–L5 level without L5 intervertebral disc level compressing the spinal nerve compression of dural sac and L5 root, but a protruded L5- and the dural sac (Figure 1). The patient was a medical S1 herniated disc (Figure 2). doctor and aware of the risk of neurological impairment. At the end of the second year, she had no pain at She refused surgical intervention and preferred to take resting or with daily activities. She described intermittent conservative treatment. She accepted to be prescribed radiating pain with or without back pain associated with ibuprofen, but refused any other medication because she seasonal factors such as rainy days. insisted on breastfeeding her child. Patient was appointed 14 sessions of physical therapy including conventional transcutaneous electrical nerve stimulation (TENS) (20 minutes), a 1-Mhz ultrasound (10 minutes, intensity 1W/cm2, continue mode), interference current vacuum therapy (10 minutes, 50 Hz frequency, pulse mode), hot pack (20 minutes), back exercises and prescribed lumbosacral corset. The patient wore lumbosacral corset for three weeks and stayed active with it. She continued working and taking care of her child after the work. She was very careful about possible triggering movements for pain because she knew biomechanics of low back. She tried to do every movement without triggering pain. After 14 physical therapy session, the patient was able to ambulate normally. Straight-leg raising test was positive at 60° and muscle strength of tibialis anterior and extensor hallucis longus were 4/5 and 3/5 on the right leg, respectively. She still had right leg radiating pain and low back pain, but not the same magnitude after the therapy (Visual analog scale for pain: before treatment 9, Figure 2: Follow-up magnetic resonance imaging (sagittal and after treatment 4). She was offered surgical intervention axial) showing regression of L4–L5 disc herniation. again, but she refused. She was followed with serial neurological examinations. During this period, her pain gradually improved. DISCUSSION

The natural course of LDH is benign in many cases as herniated nucleus pulposus regresses spontaneously in time and symptoms will improve in most patients with conservative management alone [10, 11]. There are three hypotheses explaining spontaneous regression. The first hypothesis is dehydration of herniated disc. This states that herniated fragment would disappear due to gradual dehydration and shrinkage. The second hypothesis, ‘retraction of herniated disc’, means that herniated disc retracts back into the intervertebral space. The third hypothesis, ‘inflammatory reaction and neovascularization’, states that extruded material is recognized as a foreign body and induces an inflammatory reaction by autoimmune system resulting neovascularization, enzymatic degradation, and macrophage phagocytosis [1–4, 6, 12]. It is possible that Figure 1: Sagittal and axial images of extruded disc hernia all three mechanisms play a role in regression of herniated demonstrated in magnetic resonance imaging of the lumbar tissue. Many factors related to resorption process spine. have been recognized: penetration through posterior

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):578–581. Demir et al. 580 www.ijcasereportsandimages.com longitudinal ligament, size of herniation and existence of CONCLUSION cartilage and anulus fibrosus tissue in herniated material [10]. Some authors reported that patients younger than Although a single case cannot confirm the validity 40 years old and migrating or extruding type herniations of nonsurgical management of lumbar disc herniation had a higher spontaneously regression potential [2, 6, with severe motor deficits; patient preference, symptom 13]. In their follow-up of disc herniation resorption on duration and the severity of the disability from the pain are MRI scan, Autio et al. were reported that higher baseline important factors when choosing treatment modalities. scores for enhancement thickness, higher migration according to the Komori classification, and age category ********* 41–50 years were associated with a higher resorption rate [10]. Author Contributions Motor and sensory deficits are present in 50–90% Saliha Eroğlu Demir – Substantial contributions to of patients with LDH [14]. Patients with paresthesias conception and design, Acquisition of data, Drafting the or motor weakness should be observed very closely. article, Revising it critically for important intellectual There seems to be consensus that surgery is indicated content, Final approval of the version to be published in patients with persistent neuromotor deficit, or Nihal Özaras – Substantial contributions to conception severe radiculopathy with a positive SLRT and imaging and design, Acquisition of data, Drafting the article, demonstrating LDH at the nerve root level correlating Revising it critically for important intellectual content, with the patient’s examination findings [11, 15]. Most Final approval of the version to be published surgical studies have followed a minimum six-week trial Ebru Aytekin – Substantial contributions to conception of conservative treatment before surgical intervention and design, Acquisition of data, Drafting the article, [11]. Surgical treatment may result in faster relief of Revising it critically for important intellectual content, symptoms and earlier return to function than conservative Final approval of the version to be published treatments. However, long-term results appear to be equally effective [16, 17]. Guarantor Conservative treatment for LDH and radiculopathy The corresponding author is the guarantor of submission. is primarily aimed at pain reduction by analgesics or by reducing pressure on the nerve root [15]. It includes Conflict of Interest medications, physical therapy, corsets and lumbar Authors declare no conflict of interest. injection. Physical therapy is an important conservative treatment choice of LDH. Many physical modalities Copyright including TENS, Ultrasound, laser, traction, exercise, © Saliha Eroğlu Demir et al. 2013; This article is massage could be chosen. The effects of physical distributed under the terms of Creative Commons modalities are various such as enhanced microcirculation, attribution 3.0 License which permits unrestricted use, local release of neurotransmitters such as serotonin, distribution and reproduction in any means provided increased activity of small non-myelinated C-fibers, the original authors and original publisher are properly muscle relaxation, increased local blood flow [18] In a credited. (Please see www.ijcasereportsandimages.com/ systematic review, Hahne et al. reported that many of copyright-policy.php for more information.) the intervention and comparison treatments including advice, medication, traction, stabilization exercises, physical therapy, manipulation, laser, ultrasound, corsets REFERENCES and multimodal inpatient program were equivalent [15]. Significant pain relief and improvement of function 1. Ryu SJ, Kim IS. Spontaneous regression of a large was reported by a combination treatment of physical lumbar disc extrusion. J Korean Neurosurg Soc modalities [18]. 2010;48(3):285–7. The efficacy of conservative management of LDH in 2. Chang CW, Lai PH, Yip CM, Hsu SS. Spontaneous the presence of severe motor deficit is reported in the regression of lumbar herniated disc. J Chin Med Assoc 2009;72(12):650–3. present case. Patient was referred to a spinal surgeon. 3. Gezici AR, Ergun R. Spontaneous regression of a But her choice for the treatment was conservative and huge subligamentous extruded disc herniation: short we planned combined physical therapy program and report of an illustrative case. Acta Neurochir (Wien) observed her neurologic deficits closely. The potential 2009;151(10):1299–300. for the regression was higher because she was young 4. Sabuncuoglu H, Ozdogan S, Timurkaynak E. and extruding type herniation was revealed. At the end Spontaneous regression of extruded lumbar disc of physical therapy, her pain was tolerable and there herniation: report of two illustrative case and review was obvious neurological improvement. At that period, of the literature. Turk Neurosurg 2008;18(4):392–6. we considered that the mechanism of this improvement 5. Maigne JY, Rime B, Deligne B. Computed tomographic follow-up study of forty-eight cases of nonoperatively could be the dehydration of herniated material. treated lumbar intervertebral disc herniation. Spine

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(Phila Pa 1976) 1992;17(9):1071–4. Konservatif Tedavi Etkinliginin Klinik Parametreler 6. Komori H, Shinomiya K, Nakai O, Yamaura I, Takeda ve Manyetik Rezonans Görüntüleme Yöntemi ile S, Furuya K. The natural history of herniated nucleus Degerlendirilmesi (Evaluation of the Efficacy of pulposus with radiculopathy. Spine (Phila Pa 1976) Conservative with Clinical Parameters 1996;21(2):225–9. and Magnetic Resonance Imaging in Lumbar Disc 7. Gurkanlar D, Aciduman A, Kocak H, Gunaydin H. Herniations). Turk J Phys Med Rehab 2007;53:108– Spontaneous regression of lumbar disc herniations at 12. different levels and times in a patient: A case report. 14. Weinstein JN, Lurie JD, Tosteson TD, et al. Surgical Turk Neurosurg 2005;15(1):18–22. vs nonoperative treatment for lumbar disk herniation: 8. Kasimcan O, Kaptan H. Lomber Disk Hernisinde the Spine Patient Outcomes Research Trial (SPORT) Spontan Regresyon (Spontaneous Regression of observational cohort. JAMA 2006;296(20):2451–9. Lumbar Disc Hernia: Original Image). Turkiye 15. Jacobs WC, van Tulder M, Arts M, et al. Surgery Klinikleri J Med Sci 2008;28(3):422–4. versus conservative management of sciatica due to a 9. Torun F. Spontaneous Regression of an Extruded lumbar herniated disc: a systematic review. Eur Spine Lumbar Disc Herniation: A Case Report. J Neurol Sci J 2011;20(4):513–22. [Turk] 2007;24(10):88–90. 16. Hahne AJ, Ford JJ, McMeeken JM. Conservative 10. Autio RA, Karppinen J, Niinimaki J, et al. management of lumbar disc herniation with Determinants of spontaneous resorption of associated radiculopathy: a systematic review. Spine intervertebral disc herniations. Spine (Phila Pa 1976.) (Phila Pa 1976) 2010;35(11):E488–504. 2006;31(11):1247–52. 17. Schoenfeld AJ, Weiner BK. Treatment of lumbar disc 11. Gregory DS, Seto CK, Wortley GC, Shugart CM. Acute herniation: Evidence-based practice. Int J Gen Med lumbar disk pain: navigating evaluation and treatment 2010;3:209–14. choices. Am Fam 2008;78(7):835–42. 18. Pieber K, Herceg M, Kienbauer M, et al. Combination 12. Slavin KV, Raja A, Thornton J, Wagner FC Jr. treatment of physical modalities in the treatment Spontaneous regression of a large lumbar disc of musculoskeletal pain syndromes: a prospective- herniation: report of an illustrative case. Surg Neurol controlled study. European Journal Translational 2001;56(5):333–6. Myology 2010;1(4):157-165. 13. Meydan Ocak F, Karaaslan M, Tutar I, Konuralp N, Güzelant A, Ozguzel H. Lomber Disk Hernilerinde

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CASE in images OPEN ACCESS Treatment with closed manipulation and functional bracing of a humeral diaphysis refracture with implant failure: A case report

Ali Ersen, Atakan Guvendiren, Ozgur Yazici

Abstract Keywords: Humerus diaphysis non-union, Bracing, close reduction Introduction: Implant failures and loss of the reduction are the main complications of open ********* reduction and internal fixation with plate and screws. If those complications occur, revision Ersen A, Guvendiren A, Yazici O. Treatment with closed surgery is the standard treatment. We would manipulation and functional bracing of a humeral like to present a case where implant failure and diaphysis refracture with implant failure: A case report. loss of reduction of humerus diaphyseal fracture International Journal of Case Reports and Images is treated with close reduction and Sarmiento 2013;4(10):582–585. bracing. Case Report: A 28-year-old male was diagnosed with closed humeral diaphysis ********* fracture without neuromuscular injury due to traffic accident. He was treated with open doi:10.5348/ijcri-2013-10-385-CII-14 reduction and internal fixation with plate and screws. After three months, he was presented to outpatient clinic with severe pain after minor trauma. Implant failure with screw breakage and loss of reduction with angulation were obvious Introduction on plane X-ray. Instead of revision surgery, it was treated with closed reduction and Sarmiento Most of the humeral fractures heal uneventfully with bracing. Conclusion: Functional bracing can be conservative methods. Although the operative treatment appropriate for humeral diaphysis non-unions of fractures has improved the care of many patients, it with implant failure when the patient is not may also produce undesirable consequences. Loss of suitable for surgery. fixation and non-union are two major complications of humeral fractures treated with open reduction and plate fixation.

1 2 2 Non-union rates of humeral shaft fractures are Ali Ersen , Atakan Guvendiren , Ozgur Yazici reported 10–15% with surgical treatment methods [1, 2]. Affiliations: 1Istanbul University Medical Faculty, Department of Orthopedics and Traumatology, Istanbul; 2Mardin Open fractures, segmental fractures, highly comminuted Kiziltepe State Hospital Department of Orthopedics and fractures, transvers fractures and impaired host healing Traumatology, Mardin. (diabetes, smoking and malnutrition) are the basic risk Corresponding Author: Dr. Ali Ersen, Istanbul University factors of humerus shaft non-union. İf a humeral shaft Medical Faculty, Department of Orthopedics and non-union is diagnosed, conservative treatment methods Traumatology, ISTANBUL; Ph: +905357409795; Email: ali_ are not recommended because of poor results reported [email protected] [3]. İf previous surgery has been performed hardware removal, debriding the fibrous tissue, bone grafting and fixation should be done to achieve a solid union. External Received: 27 April 2013 fixation should be necessary in cases of infected non- Accepted: 23 May 2013 Published: 01 October 2013 unions where internal devices are not useful for fixation.

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Loss of fixation is the result of inadequate surgical technique. To achieve stability with plate osteosynthesis, the plate must have adequate thickness and length. By compression plating at least four screws holes must overlie each major distal end proximal fragments and equal length of plate and number of screws should be on both sides of fracture [4]. Obvious loss of stability on radiographs or clinical examination is a clear evidence of non-union and should be re-evaluated for revision surgery. We would like to present a case where a humeral shaft fracture treated with inadequate plating technique resulted with loss of fixation and treated with close manipulation and bracing without hardware removal.

CASE REPORT

A 28-year-old male diagnosed with closed humerus shaft fracture due traffic accident without neurovascular injury. He was treated with open reduction and plate fixation at another hospital four months ago (Figure 1A–B). Postoperative period was uneventful. However, there was a lack of union after 12 weeks (Figure 2). He was presented to our outpatient clinic with severe pain after a minor trauma. The plain radiographs revealed a non- union of humerus shaft fracture with loss of reduction and implant failure with a broken screw (Figure 3). There was no evidence of infection on clinical examination and C-reactive protein level was also normal. The patient was diagnosed with atrophic non-union with implant failure Figure 1: (A, B) Inappropriate plate fixation of humeral and offered revision surgery with implant removal, bone diaphysis fracture. grafting and fixation with a longer and thicker plate. However, the patient did not accept revision surgery demanded conservative treatment. Closed manipulation (Figure 4A–B) under sedation and a Sarmiento type brace cast was applied. Although the reduction was good, the patient was informed of high risk of ineffectiveness of this treatment method. During monthly follow-up, there was no loss of reduction again, and at the fourth month follow-up after closed manipulation solid bony union was achieved without any deformity (Figure 5A–B).

DISCUSSION

Humerus non-unions can be severely disabling and the treatment should be different then acute Figure 2: After 12 weeks lack of healing. fracture treatment. Although many authors suggested debridement, open reduction and plate fixation for non- unions of the humeral shaft inadequate plating technique is the main reason of failure [5, 6]. In order to have patients with low demands functional bracing could be enough stability at the non-union site, double plating, considered as an option to support the limb but on active one on the lateral side and one on the posterior side is young patient bony union must be achieved in order recommended [7]. Bone grafting is offered to enhance the to restore function [9]. We could not find any paper in biological environment [8]. literature about the treatment of previously surgically Functional bracing for the humeral shaft provides intervened humerus diaphyseal non-union with implant high union rates but it is not the recommended treatment failure treated with closed reduction and functional option for humeral diaphyseal non-unions. On elderly bracing.

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Figure 3: Implant failure with loss of reduction and broken screw.

Figure 5: (A, B) After four months solid union was achieved.

*********

Author Contributions Ali Ersen – Substantial contributions to conception and design, Acquisition of data, Drafting the article revision it critically for important intellectuel content, Final approvel of the version to be published Atakan Gunevdiren – Substantial contributions to conception and design, Acquisition of data, Drafting Figure 4: (A, B) Implant after close reduction. the article revision, Final approvel of the version to be published Ozgur Yazici – Substantial contributions to conception and design, Acquisition of data, Drafting the article, Final approvel of the version to be published CONCLUSION Guarantor The case of our patient illustrates that functional The corresponding author is the guarantor of submission. bracing can be appropriate for humerus diaphyseal non-unions with implant failure when the patient is not Conflict of Interest suitable for surgery. Authors declare no conflict of interest.

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Copyright 4. Anglen JO, Archdeacon MT, Cannada LK, Herscovici © Ali Ersen et al. 2013; This article is distributed under D Jr, Ostrum RF. Avoiding complications in the the terms of Creative Commons attribution 3.0 License teratment of humeral fractures. Instr Course Lect which permits unrestricted use, distribution and 2009;58:3–11. 5. Ring D, Perey BH, Jupiter JB. The functional outcome reproduction in any means provided the original authors of operative treatment of ununited fractures of the and original publisher are properly credited. (Please see humeral diaphysis in older patients. J Bone Joint www.ijcasereportsandimages.com/copyright-policy.php Surg Am 1999;81(2):177–90. for more information.) 6. Foster RJ, Dixon GL Jr, Bach AW, Appleyard RW, Green TM. Internal fixation of fractures and non- unions of the humeral shaft. Indications and results REFERENCES in a multi-center study. J Bone Joint Surg Am 1985;67(6):857–64. 1. Sarmiento A, Zagorski JB, Zych GA, Latta LL, Capps 7. Murray WR, Lucas DB, Inman VT. Treatment of non- CA. Functional bracing for the treatment of fractures union of fractures of the long bones by the two-plate of the humeral diaphysis. J Bone Joint Surg Am method. J Bone Joint Surg Am 1964;46:1027–48. 2000;82(4):478–6. 8. Hierholzer C, Sama D, Toro JB, Peterson M, Helfet 2. Marti RK, Verheyen CC, Besselaar PP. Humeral shaft DL. Plate fixation of ununited humeral shaft fractures: nonunion: evaluation of uniform surgical repair in effect of type of bone graft on healing. J Bone Joint fifty-one patients. J Orthop Trauma 2002;16(2):108– Surg Am 2006;88(7):1442–7. 15. 9. Rosen, H. Management of nonunions and malunions 3. Anglen JO. Enhancement of fracture healing with in long bone fractures. In Comprehensive Care of bone stimulators. Failed internal fixation. Tech Orthopaedic Injuries in the Elderly, pp. 489-511. Orthop 2002;17:506-14. Edited by J. D. Zuckerman. Baltimore, Urban and Schwarzenberg, 1990.

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clinical images OPEN ACCESS Otogenic pneumocephalus secondary to recurrent cholesteatoma and associated temporal bone cerebrospinal fluid leak

Johannes Christiaan Oosthuizen, Fintan Wallis, John Fenton

CASE REPORT significant amount of intra-ventricular free air (Figure 1) as well as the previous posterior craniotomy defect (arrow). A 79-year-old diabetic male was presented to the High resolution images of the temporal bone confirmed emergency department with a one day history of a mild extensive soft tissue opacification of the middle ear cavity frontal headache and watery discharge from his right and a defect in the tegmen tympani. ear. His past medical history included previous mastoid Magnetic resonance imaging (MRI) scan was surgery, performed more than 50 years ago. The performed which on the coronal images (Figure 2) procedure was complicated by postoperative intracranial demonstrated extension of the lateral ventricle to abscess formation which required neurosurgical the temporal bone and localized ventriculitis and intervention and unfortunately resulted in loss of right meningeal enhancement. The diagnosis of otogenic sided visual acuity. Regrettably, the patient did not attend pneumocephalus, secondary to recurrent cholesteatoma for continued follow-up and the last contact with the with associated temporal bone CSF leak was made. Otolaryngology service had been more than 25 years ago. Unfortunately, the patient developed fulminant Examination revealed clear pulsatile otorrhea from the meningitis whilst being prepared for surgery, which right ear and a sample sent for Beta 2 transferrin testing resulted in multiorgan failure despite maximal medical confirmed the presence of a cerebrospinal fluid (CSF) care and ultimately proved fatal. leak. Microscopic examination of the external auditory canal demonstrated an inflamed external auditory canal with a significant amount of keratin debris, consistent with likely recurrent cholesteatoma, visible beyond a narrow isthmus. A contrast computed tomography (CT) scan was requested emergently and demonstrated a

Johannes Christiaan Oosthuizen1, Fintan Wallis2, John Fenton3 Affiliations: 1MBChB, MRCSI, DOHNS, Department of , Head and Neck Surgery, Limerick University Hospital, Dooradoyle, Limerick, Co. Limerick, Republic of Ireland; 2MB BCh, FRCR, Department of Radiology, Limerick University Hospital, Dooradoyle, Limerick, Co. Limerick, Republic of Ireland; 3Department of Otorhinolaryngology, Head and Neck Surgery, Limerick University Hospital, Dooradoyle, Limerick, Co. Limerick, Republic of Ireland. Corresponding Author: Mr. Johannes Christiaan Oosthuizen, MBChB, MRCSI, DOHNS, Department of Otorhinolaryngology, Head and Neck Surgery, Limerick University Hospital, Dooradoyle, Limerick, Co. Limerick, Republic of Ireland; Tel: +353 (061) 301111; Fax: +353 (061) 301165; Email: [email protected]

Received: 11 April 2013 Accepted: 28 May 2013 Figure 1: Computed tomography brain, pneumocephalus Published: 01 October 2013 (Stars), right posterior craniotomy defect (arrow).

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CSF leak which results in a relatively negative intracranial pressure which allows the lost CSF to be replaced by air and is also referred to as the ‘inverted bottle’ effect [2–4, 6]. A history of mastoid surgery, trauma or clinical findings suggestive of CSF otorrhea (which should be confirmed with a Beta 2 transferrin assay) should raise the suspicion of pneumocephalus [1]. Computed tomography scan is the first line investigation of choice and is capable of identifying as little as 0.5 mL of air within the cranial cavity [1, 4]. The use of MRI scan serves as a useful adjunct in the investigation as demonstrated by the case in question and is particularly helpful in the localization of the defect [2]. The management of pneumocephalus is dependent upon the etiology. However, surgical intervention is indicated in patients that fail conservative management with persistent CSF leak, progression of the intracranial collection or a tension pneumocephalus [3]. Conservative measures include bed rest, elevation of the patients head by 20–30 degrees and intravenous antibiotics [1]. Various surgical techniques have been described in the management of these cases and include either an extra or intracranial approach [4]. Where feasible these defects should preferentially be approached via the extracranial Figure 2: Magnetic resonance imaging, T2-weighted fluid route as this technique is associated with lower morbidity attenuated inversion recovery sequence. and mortality rates.

DISCUSSION CONCLUSION

Pneumocephalus, also referred to as aerocoele is Otogenic pneumocephalus is an infrequently defined as the present of air within the cranial cavity. It encountered clinical entity that can have vague presenting was first described by Chiari in 1884 during a postmortem symptoms and a high index of clinical suspicion is of a patient with ethmoiditis [1]. It was not, however, until necessary to diagnose this condition. 1926 that the first case of otogenic pneumocephalus was described [2]. The development of pneumocephalus is ********* reliant upon a defect in the cranial cavity which allows air to enter and typically occurs between the temporal bone Oosthuizen JC, Wallis F, Fenton J. Otogenic and the middle or posterior cranial fossa [3]. Etiological pneumocephalus secondary to recurrent cholesteatoma factors associated with the development of otogenic and associated temporal bone cerebrospinal fluid leak. pneumocephalus include trauma, otitis media, surgical International Journal of Case Reports and Images intervention, congenital defects and either benign or 2013;4(10):586–588. malignant neoplastic processes [1, 3]. The presenting features of these patients can often be vague and include ********* headache, lethargy, disorientation and meningism [4]. In the presence of raised intracranial pressure secondary doi:10.5348/ijcri-2013-10-386-CI-15 to pneumocephalus; nausea, vomiting and papilledema are commonly encountered and surgical decompression ********* is required as a matter of urgency in the instance of a tension pneumocephalus [1, 5]. Entry of air into the Author Contributions cranial cavity occurs through one of two mechanisms Johannes Christiaan Oosthuizen – Substantial [3, 6]. The first is secondary to a ball valve effect where contributions to conception and design, Acquisition of raised nasopharyngeal pressure, secondary to straining data, Analysis and interpretation of data, Drafting the or nose blowing, forces air through the defect where after article, Revising it critically for important intellectual the subsequent increase in intracranial pressure results in content, Final approval of the version to be published trapping of the air within the intracranial space [3]. The Fintan Wallis – Substantial contributions to conception second mechanism is due to a significant and continuous and design, Drafting the article, Revising it critically

IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198] IJCRI 2013;4(10):586–588. Oosthuizen et al. 588 www.ijcasereportsandimages.com for important intellectual content, Final approval of the REFERENCES version to be published John Fenton – Substantial contributions to conception 1. Lee DH, Cho HH, Cho YB. Pneumocephalus secondary and design, Drafting the article, Revising it critically to mastoid surgery: a case report. Auris Nasus Larynx for important intellectual content, Final approval of the 2007;34(1):91–3. version to be published 2. Chee NW, Niparko JK. Imaging quiz case 1. Otogenic pneumocephalus with temporal bone cerebrospinal fluid (CSF) leak. Arch Otolaryngol Head Neck Surg Guarantor 2000;126(12):1499,503. The corresponding author is the guarantor of submission. 3. Lefantzis D, Triantos S, Vontetsianos H, Dokianakis G. An unusual case of otogenic pneumocephalus. J Conflict of Interest Laryngol Otol 1998;112(12):1179–80. Authors declare no conflict of interest. 4. Ciorba A, Berto A, Borgonzoni M, Grasso DL, Martini A. Pneumocephalus and meningitis as a Copyright complication of acute otitis media: case report. Acta © Johannes Christiaan Oosthuizen et al. 2013; This article Otorhinolaryngol Ital 2007;27(2):87–9. 5. Dubey SP, Jacob O, Gandhi M. Postmastoidectomy is distributed under the terms of Creative Commons pneumocephalus: case report. Skull Base attribution 3.0 License which permits unrestricted use, 2002;12(3):167–3. distribution and reproduction in any means provided 6. Villa RA, Capdevila A. Images in clinical medicine. the original authors and original publisher are properly Spontaneous otogenic pneumocephalus. N Engl J credited. (Please see www.ijcasereportsandimages.com/ Med 2008;358(12):e13. copyright-policy.php for more information.)

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IJCRI – International Journal of Case Reports and Images, Vol. 4 No. 10, October 2013. ISSN – [0976-3198]