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Elasto fibroma Dorsi: a case report of bilateral tumours and excision of the symptomatic lesion in a male patient

Elasto fibroma Dorsi: a case report of bilateral tumours and excision of the symptomatic lesion... Elasto fibroma dorsi is a rare tumour of the shoulder girdle that usually arises at the infra scapular area. We present a 57- year-old male with a soft tissue swelling on his right infra scapular area of 6 months duration. It was a painless lesion which caused him discomfort while sleeping. Preoperative imaging revealed bilateral tumours but the left tumour was impalpable. The surgery itself was uneventful but post-operatively he developed a haematoma which was managed conservatively Elasto fibroma is a benign pseudo tumour of the shoulder girdle. It’s aetiology is tied in to repetitive trauma of the shoulder girdle resulting in a pseudo tumour at the infra scapular area. Magnetic resonance imaging is the diagnostic modality of choice and is pathognomic in the presence of bilateral infra scapular tumours. Treatment is usually conservative and tissue diagnosis is essential as it can mimic a soft tissue sarcoma radiologically. INTRODUCTION when laying down to sleep. There was no history of trauma Elasto fibroma Dorsi (ED) is a benign pseudo tumour of the and clinically he had no pain. There was no significant family shoulder girdle that commonly arises at the infra scapular area history for soft tissue tumours. on the posterior rib cage. It is usually seen in females in the Clinical examination revealed an indurated lesion at the sixth decade of life and is bilateral in 30% of patients [1]. Male right infra scapular area. There was no palpable thrill, bruit or patients make up 10% of all cases and bilateral tumours in a clinical signs of sepsis. The mass was immobile. He had a nor- male patient is extremely rare [1]. Magnetic resonance imaging mal range of movement at his right shoulder joint and his (MRI) is the imaging modality of choice and surgery usually power, tone and reflexes were normal bilaterally. His upper entails a simple excision due to a low risk of recurrence [2]. A limb neurological examination was unremarkable. He had no tissue diagnosis is required to exclude a soft tissue sarcoma or significant medical history. Ultrasound done at the referral if conservative management is being contemplated [3]. hospital indicated a deep soft tissue swelling such as an elas- tofibroma or a sarcoma. MRI confirmed the diagnosis and showed a second lesion on CASE REPORT his left side, also at the infrascapular area (Figs 1–3). The patient A male patient, 57 years of age was referred with a soft tissue had not complained of any symptoms to his left side or his left shoulder joint. After an extensive discussion with the patient swelling of 6 months duration. The lesion was palpable at the right infra scapular area on his back. He was referred to a sur- and his family, we decided on a computerized tomography (CT) geon due to its increase in size that caused him discomfort guided true cut biopsy to confirm the diagnosis (Fig. 4). Received: July 7, 2017. Accepted: October 17, 2017 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 2 Y. Pillay and R. Sabarathnam Figure 1: MRI axial view showing bilateral elasto fibromas (orange arrows) on Figure 3: MRI coronal view of bilateral elastinfibromas (orange arrows). the posterior rib cage. Figure 4: CT scan axial view showing bilateral tumours (orange arrows). Figure 2: MRI sagittal view showing the circumscribed right sided tumour with a pseudocapsule, at the infrascapular region (orange arrow). scapula, usually a rare complication of a level two axillary lymph The tissue sample was inadequate for diagnosis and at this node clearance in breast cancer surgery. Formal pathological stage the patient decided to proceed with surgery in the form results confirmed the diagnosis of an elastofibroma dorsi (Fig. 10). of an excision biopsy rather than a repeat CT guided biopsy. We decided to excise the symptomatic lesion on the right side first and then reevaluate the left side post-operatively. The left DISCUSSION sided lesion remained asymptomatic and clinically impalpable. Elastofibromas are rare benign soft tissue tumours of the shoul- Surgery was uneventful but the tumour was quite deep below the lattisimus dorsi and serratus anterior muscles, and der girdle and are more commonly seen in women than men. Brandser et al. reported an incidence of two percent in patients lying on the posterior rib cage (Figs 5–7). Post-operatively he developed a wound haematoma that was over 60 years of age on CT imaging with a female: male ratio of 11:1 [4]. The tumour is usually located at the infra-scapular managed conservatively and eventually resolved (Figs 8 and 9). He did not develop any neurological sequelae such as an area below the serratus anterior muscle on the posterior ribcage. injury to the long thoracic nerve. This results in winging of the Elasto fibroma Dorsi 3 Figure 5: Tumour exposure(forceps) below the lattisimus dorsi (blue arrow). Figure 7: Posterior rib cage(forceps), post-tumour excision. They are usually singular and bilateral tumours of the shoulder girdle are extremely rare and account for 10 percent of these tumours [5]. This adds to the uniqueness of this case report in that it is a male patient in his fifth decade of life with bilateral tumours. The tumours are usually asymptomatic and if the diagnosis has been confirmed histologically they can be managed conservatively. The pathogenesis remains nebulous though there is some evidence that repetitive trauma results in degeneration of the collagen fibres and the formation of this pseudo tumour at the inferior pole of the scapula [6]. The fibro-elastic tissue at the inferior pole acts as a shock absorber that transmits force from the upper limb to the shoulder girdle [7]. A second theory involves a genetic predisposition as it can occur in patients with a positive family history as well. Surgery is only indicated for symptomatic patients or if there is a doubt about the diagnosis as these tumours can mimic a soft tissue sarcoma radiologically. Tissue diagnosis serves a dual purpose. It reassures the patient and clinician if they decide to continue with conservative management for asymptomatic tumours and secondly that a marginal resection would suffice as opposed to a larger resection for a sarcoma if surgery is contemplated [8]. Histological sections show an admixture of alternating col- lagen bundles with abnormal elastic fibres. The elastic fibres have a degenerate fragmented appearance of petalloid globules or chenille bodies. The Verhoeff stain confirms these abnormal elastic fibres [9] (Fig. 9). The Mason’s Trichrome stain also confirmed the diagnosis by identifying the elastic fibres by negative staining. The elastic Figure 6: Tumour removed off the posterior rib cage (blue arrow). fibres appear brown on the histological slides [9] (Fig. 10). MRI 4 Y. Pillay and R. Sabarathnam Figure 8: (a) Post-operative haematoma (b) Haematoma resolution with conservative management. and we have decided on serial imaging unless the patient becomes symptomatic. CONCLUSION Elastofibromas are rare soft tissue tumours of the shoulder gir- dle and their need for a definite tissue diagnosis belies their ability to mimic a sarcoma radiologically. MRI allows for a presumptive diagnosis especially in the setting of bilateral tumours located infra scapularly. This case report is unique in that it is a male patient with bilateral tumours, which is extremely rare and accounts for 10% of all elastin fibromas. He is also a decade younger than the reported patient cohort. CONFLICT OF INTEREST STATEMENT Figure 9: High Power slide showing elastic fibres (pink areas) which confirms None declared. the diagnosis of elastofibroma. (Altered elastic fibres in a collagenous matrix). REFERENCES 1. Jarvi OH, Lansimies PH. Subclinical elastofibromas in the scapular region in an autopsy series. Acta Pathol Microbiol Scand [A] 1975;83:87–108. 2. Smith HG, Hannay JAF, Thway K, Messiou C, Smith MJF, Strauss DC, et al. Elastofibroma dorsi: the clunking tumour that need not cause alarm. Ann R Coll Surg Engl 2016;98: 208–11. doi10.1308/rcsann.2016.0064. 3. Chandrasekar CR, Grimer RJ, Carter SR, Tillman RM, Abudu A, Davies AM, et al. Elastofibroma dorsi: an uncommon benign pseudotumour. Sarcoma 2008;2008:756565. 4. Brandser EA, Goree JC, El-Khoury GY. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. AJR Am J Roentgenol 1998;171:977–80. 5. Briccoli A, Casadei R, Di Renzo M, et al. Elastofibroma dorsi. Figure 10: Mason’s Trichome stain outlining collagen around the abnormal Surg Today 2000;30:147–52. elastic fibres with the characteristic serrated edges or petalloid globules. It out- 6. Jarvi O, Saxen E. Elastofibroma dorse. Acta Pathol Microbiol lines the elastic fibres by negative staining. Scand Suppl 1961;51:83–4. 7. Hayes AJ, Alexander N, Clark MA, Thomas JM. Elastofibroma: remains the diagnostic imaging modality of choice. It has the a rare soft tissue tumour with a pathognomonic anatomical added advantage of detecting contralateral subclinical lesions location and clinical symptom. Eur J Surg Oncol 2004;30:450–3. which would clinch the diagnosis as in this case. A second 8. Nagamine N, Nohara Y, Ito E. Elastofibroma in Okinawa. A issue is the left sided tumour which was not excised at the ori- clinicopathologic study of 170 cases. Cancer 1982;50: ginal surgery. The patient continues to remain asymptomatic 1794–1805. on the left side and the tumour remains clinically impalpable. 9. Fardisi S, Ashraf MJ, Zarei MR, Azarpira N, Raoof M, The patient has opted for conservative management of this Amanpour S, et al. Elastofibroma of the face: a case report. lesion in the interim. There has been no increase in size to date J Dent (Shiraz) 2015;16:73–5. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Elasto fibroma Dorsi: a case report of bilateral tumours and excision of the symptomatic lesion in a male patient

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017.
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2042-8812
DOI
10.1093/jscr/rjx206
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Abstract

Elasto fibroma dorsi is a rare tumour of the shoulder girdle that usually arises at the infra scapular area. We present a 57- year-old male with a soft tissue swelling on his right infra scapular area of 6 months duration. It was a painless lesion which caused him discomfort while sleeping. Preoperative imaging revealed bilateral tumours but the left tumour was impalpable. The surgery itself was uneventful but post-operatively he developed a haematoma which was managed conservatively Elasto fibroma is a benign pseudo tumour of the shoulder girdle. It’s aetiology is tied in to repetitive trauma of the shoulder girdle resulting in a pseudo tumour at the infra scapular area. Magnetic resonance imaging is the diagnostic modality of choice and is pathognomic in the presence of bilateral infra scapular tumours. Treatment is usually conservative and tissue diagnosis is essential as it can mimic a soft tissue sarcoma radiologically. INTRODUCTION when laying down to sleep. There was no history of trauma Elasto fibroma Dorsi (ED) is a benign pseudo tumour of the and clinically he had no pain. There was no significant family shoulder girdle that commonly arises at the infra scapular area history for soft tissue tumours. on the posterior rib cage. It is usually seen in females in the Clinical examination revealed an indurated lesion at the sixth decade of life and is bilateral in 30% of patients [1]. Male right infra scapular area. There was no palpable thrill, bruit or patients make up 10% of all cases and bilateral tumours in a clinical signs of sepsis. The mass was immobile. He had a nor- male patient is extremely rare [1]. Magnetic resonance imaging mal range of movement at his right shoulder joint and his (MRI) is the imaging modality of choice and surgery usually power, tone and reflexes were normal bilaterally. His upper entails a simple excision due to a low risk of recurrence [2]. A limb neurological examination was unremarkable. He had no tissue diagnosis is required to exclude a soft tissue sarcoma or significant medical history. Ultrasound done at the referral if conservative management is being contemplated [3]. hospital indicated a deep soft tissue swelling such as an elas- tofibroma or a sarcoma. MRI confirmed the diagnosis and showed a second lesion on CASE REPORT his left side, also at the infrascapular area (Figs 1–3). The patient A male patient, 57 years of age was referred with a soft tissue had not complained of any symptoms to his left side or his left shoulder joint. After an extensive discussion with the patient swelling of 6 months duration. The lesion was palpable at the right infra scapular area on his back. He was referred to a sur- and his family, we decided on a computerized tomography (CT) geon due to its increase in size that caused him discomfort guided true cut biopsy to confirm the diagnosis (Fig. 4). Received: July 7, 2017. Accepted: October 17, 2017 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 2 Y. Pillay and R. Sabarathnam Figure 1: MRI axial view showing bilateral elasto fibromas (orange arrows) on Figure 3: MRI coronal view of bilateral elastinfibromas (orange arrows). the posterior rib cage. Figure 4: CT scan axial view showing bilateral tumours (orange arrows). Figure 2: MRI sagittal view showing the circumscribed right sided tumour with a pseudocapsule, at the infrascapular region (orange arrow). scapula, usually a rare complication of a level two axillary lymph The tissue sample was inadequate for diagnosis and at this node clearance in breast cancer surgery. Formal pathological stage the patient decided to proceed with surgery in the form results confirmed the diagnosis of an elastofibroma dorsi (Fig. 10). of an excision biopsy rather than a repeat CT guided biopsy. We decided to excise the symptomatic lesion on the right side first and then reevaluate the left side post-operatively. The left DISCUSSION sided lesion remained asymptomatic and clinically impalpable. Elastofibromas are rare benign soft tissue tumours of the shoul- Surgery was uneventful but the tumour was quite deep below the lattisimus dorsi and serratus anterior muscles, and der girdle and are more commonly seen in women than men. Brandser et al. reported an incidence of two percent in patients lying on the posterior rib cage (Figs 5–7). Post-operatively he developed a wound haematoma that was over 60 years of age on CT imaging with a female: male ratio of 11:1 [4]. The tumour is usually located at the infra-scapular managed conservatively and eventually resolved (Figs 8 and 9). He did not develop any neurological sequelae such as an area below the serratus anterior muscle on the posterior ribcage. injury to the long thoracic nerve. This results in winging of the Elasto fibroma Dorsi 3 Figure 5: Tumour exposure(forceps) below the lattisimus dorsi (blue arrow). Figure 7: Posterior rib cage(forceps), post-tumour excision. They are usually singular and bilateral tumours of the shoulder girdle are extremely rare and account for 10 percent of these tumours [5]. This adds to the uniqueness of this case report in that it is a male patient in his fifth decade of life with bilateral tumours. The tumours are usually asymptomatic and if the diagnosis has been confirmed histologically they can be managed conservatively. The pathogenesis remains nebulous though there is some evidence that repetitive trauma results in degeneration of the collagen fibres and the formation of this pseudo tumour at the inferior pole of the scapula [6]. The fibro-elastic tissue at the inferior pole acts as a shock absorber that transmits force from the upper limb to the shoulder girdle [7]. A second theory involves a genetic predisposition as it can occur in patients with a positive family history as well. Surgery is only indicated for symptomatic patients or if there is a doubt about the diagnosis as these tumours can mimic a soft tissue sarcoma radiologically. Tissue diagnosis serves a dual purpose. It reassures the patient and clinician if they decide to continue with conservative management for asymptomatic tumours and secondly that a marginal resection would suffice as opposed to a larger resection for a sarcoma if surgery is contemplated [8]. Histological sections show an admixture of alternating col- lagen bundles with abnormal elastic fibres. The elastic fibres have a degenerate fragmented appearance of petalloid globules or chenille bodies. The Verhoeff stain confirms these abnormal elastic fibres [9] (Fig. 9). The Mason’s Trichrome stain also confirmed the diagnosis by identifying the elastic fibres by negative staining. The elastic Figure 6: Tumour removed off the posterior rib cage (blue arrow). fibres appear brown on the histological slides [9] (Fig. 10). MRI 4 Y. Pillay and R. Sabarathnam Figure 8: (a) Post-operative haematoma (b) Haematoma resolution with conservative management. and we have decided on serial imaging unless the patient becomes symptomatic. CONCLUSION Elastofibromas are rare soft tissue tumours of the shoulder gir- dle and their need for a definite tissue diagnosis belies their ability to mimic a sarcoma radiologically. MRI allows for a presumptive diagnosis especially in the setting of bilateral tumours located infra scapularly. This case report is unique in that it is a male patient with bilateral tumours, which is extremely rare and accounts for 10% of all elastin fibromas. He is also a decade younger than the reported patient cohort. CONFLICT OF INTEREST STATEMENT Figure 9: High Power slide showing elastic fibres (pink areas) which confirms None declared. the diagnosis of elastofibroma. (Altered elastic fibres in a collagenous matrix). REFERENCES 1. Jarvi OH, Lansimies PH. Subclinical elastofibromas in the scapular region in an autopsy series. Acta Pathol Microbiol Scand [A] 1975;83:87–108. 2. Smith HG, Hannay JAF, Thway K, Messiou C, Smith MJF, Strauss DC, et al. Elastofibroma dorsi: the clunking tumour that need not cause alarm. Ann R Coll Surg Engl 2016;98: 208–11. doi10.1308/rcsann.2016.0064. 3. Chandrasekar CR, Grimer RJ, Carter SR, Tillman RM, Abudu A, Davies AM, et al. Elastofibroma dorsi: an uncommon benign pseudotumour. Sarcoma 2008;2008:756565. 4. Brandser EA, Goree JC, El-Khoury GY. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. AJR Am J Roentgenol 1998;171:977–80. 5. Briccoli A, Casadei R, Di Renzo M, et al. Elastofibroma dorsi. Figure 10: Mason’s Trichome stain outlining collagen around the abnormal Surg Today 2000;30:147–52. elastic fibres with the characteristic serrated edges or petalloid globules. It out- 6. Jarvi O, Saxen E. Elastofibroma dorse. Acta Pathol Microbiol lines the elastic fibres by negative staining. Scand Suppl 1961;51:83–4. 7. Hayes AJ, Alexander N, Clark MA, Thomas JM. Elastofibroma: remains the diagnostic imaging modality of choice. It has the a rare soft tissue tumour with a pathognomonic anatomical added advantage of detecting contralateral subclinical lesions location and clinical symptom. Eur J Surg Oncol 2004;30:450–3. which would clinch the diagnosis as in this case. A second 8. Nagamine N, Nohara Y, Ito E. Elastofibroma in Okinawa. A issue is the left sided tumour which was not excised at the ori- clinicopathologic study of 170 cases. Cancer 1982;50: ginal surgery. The patient continues to remain asymptomatic 1794–1805. on the left side and the tumour remains clinically impalpable. 9. Fardisi S, Ashraf MJ, Zarei MR, Azarpira N, Raoof M, The patient has opted for conservative management of this Amanpour S, et al. Elastofibroma of the face: a case report. lesion in the interim. There has been no increase in size to date J Dent (Shiraz) 2015;16:73–5.

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Journal of Surgical Case Reports – Oxford University Press

Published: Nov 7, 2017

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