Intercostal Neuralgia Caused by a Parosteal Lipoma of the Rib

Intercostal Neuralgia Caused by a Parosteal Lipoma of the Rib

Ann Thorac Surg 2006;81:1901–3 CASE REPORT KIM ET AL PAROSTEAL LIPOMA OF THE RIB 1901 1. Albuquerque KV, Deshpande RK, Desai PB. Cervical approach ...

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Ann Thorac Surg 2006;81:1901–3

CASE REPORT KIM ET AL PAROSTEAL LIPOMA OF THE RIB

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1. Albuquerque KV, Deshpande RK, Desai PB. Cervical approach for repair of congenital tracheo-esophageal fistula presenting in an adult. J Postgrad Med 1993;39(4):216 –7. 2. Holman WL, Vaezy A, Postlethwait RW, Bridgman A. Surgical treatment of H-type tracheoesophageal fistula diagnosed in an adult. Ann Thorac Surg 1986;41:453– 4. 3. Zacharias J, Genc O, Goldstraw P. Congenital tracheoesophageal fistulas presenting in adults: presentation of two cases and a synopsis of the literature. J Thorac Cardiovasc Surg 2004;128:316 – 8. 4. Azoulay D, Regnard JF, Magdeleinat P, Diamond T, RojasMiranda A, Levasseur P. Congenital respiratory-esophageal fistula in the adult. J Thorac Cardiovasc Surg 1992;104(2): 381– 4. 5. Rothenberg S. Thoracoscopic repair of tracheoesophageal fistula in newborns. J Pediatr Surg 2002;37(6):869 –72. 6. Bax NMA, van Der Zee DC. Feasibility of thoracoscopic repair of esophageal atresia with distal fistula. J Pediatr Surg 2002; 37(2):192– 6. 7. Hazelrigg SR, Boley TM, Naunheim KS, et al. Effect of bovine pericardial strips on air leak after stapled pulmonary resection. Ann Thorac Surg 1997;63(6):1573–5.

Intercostal Neuralgia Caused by a Parosteal Lipoma of the Rib Hyun Koo Kim, MD, PhD, Young Ho Choi, MD, PhD, Yang Hyun Cho, MD, Young-sang Sohn, MD, PhD, and Hark Jei Kim, MD, PhD Department of Thoracic and Cardiovascular Surgery, Korea University Medical Center, Seoul, Korea

Parosteal lipoma is an extremely rare benign tumor that is composed mainly of mature adipose tissue, and it has an intimate relationship to the underlying periosteal bone. We believe that only three cases have been previously reported that have described parosteal lipoma of the rib. Although parosteal lipoma is asymptomatic, motor and sensory function deficits have been reported that were caused by the tumor compressing the neuromuscular bundles in the proximal forearm and the sciatic nerve. We present here an exceedingly rare case of intercostal neuralgia caused by a parosteal lipoma of the rib. (Ann Thorac Surg 2006;81:1901–3) © 2006 by The Society of Thoracic Surgeons

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arosteal lipoma is a rare benign neoplasm that is mainly composed of mature adipose tissue that has an intimate relationship to the underlying periosteal bone [1]. The incidence of this tumor is 0.3% of all lipomas. The most common locations for this tumor are the femur, proximal radius, humerus, tibia, clavicle, and pelvis [2]. We believe that only three previously reported cases of parosteal lipoma of the rib have been described. We report here on an exceedingly rare case of intercostal neuralgia that was caused by parosteal lipoma of the rib.

Accepted for publication May 19, 2005. Address correspondence to Dr Choi, 97 Guro-Dong, Guro-Ku, Seoul, 152-703 Korea; e-mail: [email protected].

© 2006 by The Society of Thoracic Surgeons Published by Elsevier Inc

Fig 1. Computed tomographic scan demonstrating a fatty mass lesion in the intramuscular layer of the left posterior chest wall at the fifth to eighth rib level with an internal protruding osseous masslike portion from the sixth rib.

A 57-year-old woman was referred to us for a palpable mass of 3 years duration that was located on the left posterior chest wall. The mass had slowly increased in size and was associated with progressive intercostal neuralgia for the past 5 weeks. The patient denied any history of trauma. The physical examination revealed a soft, oval-shaped, fixed mass on the left paravertebral area. Computed tomographic scan demonstrated a welldefined elongated mass with a fat density signal in the left chest wall from the fifth to the eighth posterior rib levels (Fig 1). The mass was mainly located in posterior paraspinal back muscle layer with a local insinuation into the sixth intercostal space and the intrathoracic region. The outer cortex of the attached sixth rib showed a cauliflower-like osseous lesion protruding into the fatty mass lesion, and the inner rib cortex showed a slight diffuse erosive change. The operation was performed on the patient through a left posterolateral thoracotomy. The mass was strongly adhered to the underlying sixth rib, and it was displacing the intercostal nerve posteriorly and compressing it. The parietal pleura was opened to examine the inner surface of the rib, which had a hard irregularity, but there was no adhesion to the visceral pleural. The patient underwent en bloc resection of the mass and the left sixth rib. The distance from the resection margin of the rib was 6 cm and 3 cm, respectively. A final diagnosis of parosteal lipoma was rendered. On gross examination, the mass measured 9 ⫻ 6 ⫻ 4 cm and was relatively well demarcated, yellowish, homogenous, and soft (Fig 2). Histopathologically, the lesion was composed of mature lipocytes that had an intimate relationship with the periosteum. No cellular atypia or lipoblasts were seen (Fig 3). 0003-4975/06/$32.00 doi:10.1016/j.athoracsur.2005.05.079

FEATURE ARTICLES

References

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CASE REPORT KIM ET AL PAROSTEAL LIPOMA OF THE RIB

Ann Thorac Surg 2006;81:1901–3

The patient had an uneventful postoperative course and she complained of some minimal wound pain at 2 months after surgery.

Comment

FEATURE ARTICLES

Lipoma is a soft tissue tumor, but osseous localization of a lipoma is rare [1]. There are three types of osseous lipomas with respect to their relation to the parent bone: (1) interosseous, (2) cortical, and (3) parosteal. Parosteal lipomas are rare and usually asymptomatic benign lesions consisting of mature adipose tissue that is intimately associated with the periosteum of bone. Since their original description by Seering in 1836, various reviews in the English literature have been published during the years regarding this rare entity [3]. Patients with parosteal lipoma range in age from 40 to 60 years of age, and they usually present with a history of a slow growing, large, painless, and nontender immobile mass that is not fixed to the skin. The most common sites of origin for parosteal lipomas are the femur and the radius [2]. Disturbances in the motor and sensory function of the adjacent nerves may occasionally occur due to local pressure, and this can result in muscle atrophy [4]. In our patient, having intercostals neuralgia, the mass intruded into the intercostal space and this compressed the intercostal neurovascular bundle lying beneath the rib. On roentgenograms, the most characteristic feature demonstrated is a well-defined fat-containing mass adjacent to the cortical bone, and this is commonly associated with reactive changes in the underlying cortex. On computed tomographic scan, parosteal lipoma usually presents as a well-defined mass almost entirely composed of mature adipose tissue with an osseous excrescence within it or erosion at the attachment of the soft-tissue mass to the subjacent cortex, or both [5]. Computed tomographic scan can also confirm the indolent nature of this lesion by showing the mechanical displacement rather than the invasion of the adjacent structures.

Fig 3. Microscopic examination shows the well-circumscribed pedunculated bony proliferation with an overlying mass of mature adipose tissue. (Hematoxylin and eosin; ⫻100.)

Pathologically the lesion is usually a multi-lobulated yellowish mass composed of mature adipocytes, and it is well encapsulated with a broad base of attachment to the underlying bone [6]. Microscopically, the fat cells of a parosteal lipoma appear histochemically identical to the adipocytes that are found in the subcutaneous tissues [7]. There has been no indication to date that this tumor undergoes malignant degeneration, although minimal cellular pleomorphism may occasionally occur. The treatment of parosteal lipoma is complete surgical resection. In the case with nerve entrapment, the tumor must be removed before irreversible muscle atrophy occurs so as to maintain function. The nerve must also be separated from the parosteal lipoma and care must be taken to spare it during surgical excision [4]. In our case, the intercostal neuromuscular bundle was secured by carefully dissecting it from the mass. Adequate surgical removal of a parosteal lipoma requires either subperiosteal dissection, an osteotome to separate the lesion from the underlying bone or segmental resection of bone; this is in contrast to the relatively easy dissection for a soft tissue lipoma lying adjacent to bone. Local recurrence is unusual, but it has been reported. There are no reports of malignant transformation. In summary, we have described an extremely rare case of parosteal lipoma of the rib that induced intercostal neuralgia.

References

Fig 2. The gross finding shows the relatively well demarcated yellowish homogenous adipose tissue attached to the rib. The mass measured 9 times; 6 ⫻ 4 cm.

1. Murphey MD, Johnson DL, Bhatia PS, Neff JR, Rosenthal HG, Walker CW. Parosteal lipoma: MR imaging characteristics. AJR Am J Roentgenol 1994;162:105–10. 2. Fleming RJ, Alpert M, Garcia A. Parosteal lipoma. AJR Am J Roentgenol 1962;87:1075– 84. 3. Kawashima A, Magid D, Fishman EK, Hruban RH, Ney DR. Parosteal ossifying lipoma: CT and MR findings. J Comput Assist Tomogr 1993;17:147–50. 4. Fitzgerald A, Anderson W, Hooper G. Posterior interosseous nerve palsy due to parosteal lipoma. J Hand Surg 2002;27: 535–7.

Ann Thorac Surg 2006;81:1903– 4

CASE REPORT PALIMENTO AND PICCHIO MENINGIOMA CAUSING HEMOTHORAX

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5. Imbriaco M, Ignarra R, De Rosa N, Lambiase G, Romano M, Ragozzino A. Parosteal lipoma of the rib. CT findings and pathologic correlation. Clin Imaging 2003;27:435–7. 6. Yu JS, Weis L, Becker W. MR imaging of a parosteal lipoma. Clin Imaging 2000;24:15– 8. 7. Miller MD, Ragsdale BD, Sweet DE. Parosteal lipomas: a new perspective. Pathology 1992;24:132–9.

Meningioma of the Mediastinum Causing Spontaneous Hemothorax Domenico Palimento, MD, and Marcello Picchio, MD

Ectopic meningiomas are extremely rare. Mediastinal tumors may rarely cause spontaneous hemothorax. We report a case of angioblastic meningioma of the posterior mediastinum causing spontaneous hemothorax. (Ann Thorac Surg 2006;81:1903– 4) © 2006 by The Society of Thoracic Surgeons

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pontaneous hemothorax, a collection of blood in the pleural cavity not related to trauma, is a rare disease manifestation. Hemothorax due to mediastinal tumors is extremely rare. We report a case of angioblastic meningioma of the posterior mediastinum, presenting as spontaneous hemothorax. A 45-year-old woman was admitted with left thoracic pain, dyspnea, and easy fatigability for 24 hours. At admission, general conditions were satisfactory, blood pressure was 120/80 mm Hg, and the pulse rate was 75/min and regular. Physical examination of the thorax showed a decrease in tactile fremitus, dullness to percussion, and diminution of breath sounds all over the left hemithorax. Laboratory tests showed a red cell count of 3,770,000/mm3, hemoglobin of 12.0 gr/dL, and hematocrit of 31.7%, and an arterial blood gas analysis revealed a pH of 7.34 with Pao2 of 82 mm Hg and Paco2 of 32 mm Hg. Chest roentgenogram showed the presence of a posterior paravertebral mass in the left hemithorax; the mass had smooth margins and it was 10 cm wide. Basal pleural effusion was also present. Two hours after admission, the patient had hypotension (blood pressure, 50/60 mm Hg) with bradycardia (heart beat, 44/min) develop, and her hemoglobin was 6.5 gr/100 mL and her hematocrit was 19%. In spite of appropriate volume replacement with crystalloid solutions and packed red cells, restoration of hemodynamic stability was not achieved (blood pressure, 80/60 mm Hg). A computed tomographic scan of the thorax demonstrated the presence of an 11 cm wide left paravertebral mass. After contrast agent injection the mass showed hemorrhagic areas. A massive pleural effusion was also Accepted for publication July 13, 2005. Address correspondence to Dr Picchio, via Stefano Boccapaduli, nr. 51, Rome, 00151 Italy; e-mail: [email protected].

© 2006 by The Society of Thoracic Surgeons Published by Elsevier Inc

Fig 1. Computed tomographic scan of the chest showing the left hemothorax with collapsed lung and after contrast enhancement an 11 cm wide left paravertebral mass.

present with rightward shift of the mediastinum and hilum and collapse of the left lung (Fig 1). Paracentesis of the left hemithorax returned blood. The patient underwent emergency left posterolateral thoracotomy. When the pleural cavity was opened, 2 L of blood were drained. The paravertebral mass as seen on computed tomographic scan was 12 cm wide, elastic, and off white in color. The mass was completely excised. The thoracic wound was closed with drainage of the left pleural cavity. Histologic examination of the mass showed the presence of monomorphic spindle cells with a low mitotic index (5 ⫻ 10 high power view), an abundant vascular network, and focal hemorrhagic and necrotic areas (Fig 2). Immunohistochemical examination showed focal positivity to CD34, CD68, and vimentin. The morphologic and immunohistochemical patterns were compatible with the diagnosis of angioblastic meningioma. The postoperative course was uneventful and the patient was discharged 7 days after operation. One month after operation the patient had magnetic resonance imaging of the head and spine, which excluded possible central nervous system lesions.

Comment Meningiomas are common and usually benign, slowgrowing tumors of the central nervous system. Ectopic meningiomas are uncommon and arise through metastatic spread of malignant meningiomas. Primary ectopic meningiomas are extremely rare extracranial and extraspinal tumors and have been reported sporadically in the head and neck region as well as in the lung, finger, skin, eyelid, retroperitoneum, thigh muscle, and foot [1]. The histogenesis of ectopic menigiomas is still unclear. They are believed to arise from ectopic arachnoid tissue or from mesenchymal cells or Schwann cells that have been differentiated into meningothelial cells [2, 3]. Only 0003-4975/06/$32.00 doi:10.1016/j.athoracsur.2005.07.045

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Department of Surgery, Civil Hospital “S. Paolo,” Naples; Department of Surgery, Civil Hospital “A. Fiorini,” Terracina, Italy