Ann Thorac Surg 2007;84:2091-2093. doi:10.1016/j.athoracsur.2007.06.042
© 2007 The Society of Thoracic Surgeons
Case Reports
Radiofrequency Thermoablation in Chest Wall Mesenchymal Hamartoma of an Infant
Arianna Bertocchini, MDa,
Piergiorgio Falappa, MDb,
Antonella Accinni, MDa,
Rita Devito, MDc,
Alessandro Inserra, MDa,*
a General and Thoracic Surgery Unit, Rome, Italy
b Vascular and Interventional Radiology Unit, Rome, Italy
c Pathology Unit, Bambino Gesù Children’s Hospital, Rome, Italy
Accepted for publication June 13, 2007.
* Address correspondence to Dr Inserra, Bambino Gesù Children’s Hospital, Piazza S. Onofrio 4, Rome, 00165, Italy (Email: inserra{at}opbg.net).
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Abstract
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We report on an infant presenting with a chondroid hamartoma managed with a combined conservative surgical treatment and radiofrequency thermoablation.
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Introduction
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Mesenchymal hamartomas of the chest wall are rare benign lesions usually discovered at birth or in early infancy. When symptoms are present, they are respiratory in nature, and they result from the extrinsic compression on the adjacent pulmonary parenchyma. Their severity depends on the lesion size and location. Although spontaneous regression has been documented, the more common outcome is its progressive growth [1], and in the literature the treatment of choice is "en-bloc" excision of the involved portion of the chest wall, even if severe postoperative orthopedic problems might occur. Authors [2] emphasize the importance of early and complete excision of the lesion (even in minimally symptomatic patients) to avoid lethal respiratory and orthopedic complications or malignant transformation [1].
A 6-month-old girl was admitted to our institute to evaluate a mass in her right chest wall, together with respiratory distress and deficiency of growth.
A first radiological evaluation (Figs 1A,
1B) revealed a mass extending into the thorax arising from the right third and fourth ribs (T3 to T4) and another smaller one developing from the eighth and ninth (T8 to T9). A right thoracotomy was performed with excision of the upper mass, leaving the involved ribs behind so as not to affect the column stability. For its limited size the second mass has not been removed.
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Fig 1. (A) Chest x-ray film showing a right thoracic mass that dislocates the mediastinum leftward. (B) Middle thorax computed tomographic scan showing a disomogeneous mass arising from the chest wall. (C) Chest x-ray film showing an upper right thoracic recurrent mass 4 months after surgical treatment. (D) Upper thorax computed tomographic scan showing a disomogeneous recurrent mass similar to the first one.
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At macroscopic examination (Figs 2A,
2B) the specimen on cut sections showed cyst-like spaces filled with blood, intermingled with soft brownish and hard whitish areas. At histopathologic examination the solid areas were composed of a proliferation of primitive mesenchymal cells without atypia, which focally showed maturation into cartilagineous islands with enchondral ossification and immature bone surrounded by plump fibroblasts. The cystic spaces were separated by fibrous septa containing fibroblasts, multinucleated giant cells, and hemosiderin-laden macrophages. Endothelial-like cells lined the septa and showed positivity for vimentin, (cluster differentiation) CD68 (clone PGM1), and CD163, and negativity for CD31 and CD34. The diagnosis was mesenchymal hamartoma of the chest wall with secondary aneurysmal bone cyst.
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Fig 2. (A, B) Histopathological findings of chondroid islands and vascular aneurysmal bone cyst-like wall. ([A] Hematoxylin and eosin, magnification 20x; [B] Hematoxylin and eosin, magnification 10x.) (C) Radiofrequency thermoablation under computed tomography control in the lateral decubitus after debulking the fluid content. (D) One-year follow-up X-ray film of a remarkable reduction of the upper right thoracic mass. The eighth and ninth ribs show a dysplastic lesion caused by the hamartoma localization.
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Four months later a follow-up computed tomographic scan demonstrated a recurrent mass that was both cystic and solid (Figs 1C, 1D). The lesion was localized in the upper right thorax, arising from the same ribs. It extended to the anterior thoracic wall compressing the right bronchus. A modest dimensional increase of the lower mass was documented. After suitable informed consent, radiofrequency thermoablation (RFT) treatment was planned out exclusively for the bigger mass to treat the local disease and avoid radical chest wall resection.
After a needle biopsy, two needle electrodes with a 3-cm exposed tip were sequentially inserted percutaneously into the lesion under computed tomographic guidance (Cool-tip RF ablation system; Valleylab, Boulder, CO) (Fig 2C), with the former tangent to the involved third rib and the latter into the middle of the mass. The lesion was treated for 6 minutes at each level with 100 Watts of power. The temperature at the thermocoagulation electrode tip was 55°C, which was assessed at the end of the procedure. After removal of the needle system, a computed tomographic scan was obtained to check for possible complications.
For its large extension, a new RFT was performed on the reduced mass 1.5 months later.
Two months after the RFT, the treated upper lesion appeared unvaried to magnetic resonance and roentgenogram follow-up. On the contrary the lesion at the T8 to T9 level that was never treated, because it has always been asymptomatic, looked slightly enlarged.
At a 1-year roentgenogram follow-up, the treated mass even showed a volumetric decrease (Fig 2D).
Presently, 12 months after the RFT, the patient is asymptomatic, in good clinical condition, with no evidence of scoliosis, and at the right height and ponderal growth.
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Comment
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Approximately 80 cases of cartilaginous hamartoma have been described in the literature to date, and the reported incidence is 1 in 3,000 (0.03%) among primary bone tumors. Recommendation for treatment of chest wall hamartoma varies. One tumor was reported to have been spontaneously regressed, supporting the observation that it may be appropriate for conservative management in asymptomatic children whose tumor is not incompatible with growth or life [3]. Nevertheless, most cases have been treated with surgical excision, which is necessary in respiratory involvement with a distress syndrome. Surgery however is not without complications, including hemorrhage and post-surgical scoliosis, especially if an en-bloc excision has been performed. The curvature degree is related directly to the number of resected ribs, with the greater curvature being found to follow the more estensive resections. Due to these observations, we decided to use a more conservative surgery along with RFT. Radiofrequency thermoablation is a relatively noninvasive percutaneous and repeatable technique, performed under computed tomographic guidance, which can ablate areas of pathologic tissue.
In particular, as RFT has been effectively used in osteoid osteoma and epiphyseal chondroblastoma, although in older children, we decided to also use the same technique to treat chondroid hamartomas because it is histologically similar [4, 5].
However, in younger children, RFT is used to treat cardiac diseases [6]. Radiofrequency thermoablation has been performed on the basis of this evidence to avoid new surgery, which would have been highly radical considering the mass localization and dimensions.
This type of approach has allowed us to effectively treat a rather extended lesion in a minimally invasive way, localized in a site that is difficult to reach in a child who is less than 1 year of age.
The upper mass response to the treatment and growth of the lower one, although slight, demonstrates the success of our procedure.
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References
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- Dounies R, Chwals WJ, Lally KP, et al. Hamartomas of the chest wall in infants Ann Thorac Surg 1994;57:868-875.[Abstract]
- Andiran F, Ciftci AO, Senocak ME, Akcoren Z, Gogus S. Chest wall hamartoma: an alarming chest lesion with a benign course J Pediatr Surg 1998;33:727-729.[Medline]
- Cameron D, Ong TH, Borzi P. Conservative management of mesenchymal hamartomas of the chest wall J Pediatr Surg 2001;36:1346-1349.[Medline]
- Rosenthal DI, Hornicek FJ, Wolfe MW, Jennings CL, Gebhardt MC, Mankin HJ. Percutaneous radiofrequency coagulation of osteoid osteoma compared with operative treatment J Bone Joint Surg Am 1998;80:815-821.[Abstract/Free Full Text]
- Erickson JK, Rosenthal DI, Zaleske DJ, Gebhardt MC, Cates JM. Primary treatment of chondroblastoma with percutaneous radio-frequency heat ablation: report of three cases Radiology 2001;221:463-468.[Abstract/Free Full Text]
- Vaksmann G, D’Hoinne C, Lucet V, et al. Permanent junctional reciprocating tachycardia in children: a multicentre study on clinical profile and outcome Heart 2006;92:101-104.[Abstract/Free Full Text]
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Abstract
Introduction
