Disorders of the Pleura |

Cavernous Pleural Hemangioma: A Rare Cause of Nonhemorrhagic Pleural Effusion FREE TO VIEW

Cherry Ann Dionzon-Serra, MD; Ricardo Salonga, MD; Rodolfo Dizon, MD; Ricardo Beroncal, MD; Jose Edzel Tamayo, MD
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Internal Medicine - Section of Pulmonology, University of Perpetual Help Dalta Medical Center, Las Pinas City, Philippines

Chest. 2014;146(4_MeetingAbstracts):475A. doi:10.1378/chest.1989638
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SESSION TITLE: Pleural Disease Global Case Reports

SESSION TYPE: Global Case Report

PRESENTED ON: Tuesday, October 28, 2014 at 01:30 PM - 02:30 PM

INTRODUCTION: Hemangioma is a compact, abnormal collection of blood vessels. It is a common benign vascular tumor, however, quite rare in some location including the thorax. Gindhart et al reported that it is less than 0.5% of all mediastinal masses and up to 75% of these lesions manifest before the age of 35 (1). Hemangiomas constitute 7% of all benign tumours.

CASE PRESENTATION: A case of a 41 y.o. female, admitted due to chest heaviness. She had been experiencing chest heaviness aggravated by lifting heavy objects for almost a week. Sought consult and Chest Xray showed massive pleural effusion. She underwent thoracentesis, findings were nonspecific, and since tuberculosis is endemic in our country, she was treated empirically with antiKoch’s medication. She was readmitted twice thereafter in the span of three weeks due to reaccumulation of pleural effusion ipsilaterally. Repeat thoracentesis was done and by this time malignancy was highly entertained and yet pleural fluid cytology was inconclusive. She underwent CTT on her third admission and subsequently had Chest CT scan which revealed a heterogenous mass measuring 2.7 x 3.0 x 2.7 cm located at the paravertebral area at the level of T4. Patient underwent Video-assisted Thoracoscopic Surgery (VATS) and the mass was excised and biopsied. Biopsy result showed morphologic findings of large vessel with dilated lumina and thin walls with multiple tiny bubbles filled with blood . The immunohistochemical examination of the tissue showed positivity to Vimentin and CD34. Taken together, these findings led to the diagnosis of Pleural Cavernous Hemangioma with secondary pleural effusion. The patient went home improved without recurrence of pleural effusion up to the present.

DISCUSSION: Pleuropulmonary hemangiomas are extremely rare. They may be congenital or may be as a part of Von-Hippel Lindau disease (capillary hemangiomatosis). The epidemiology is rare and the pathogenesis is unknown. Hemangiomas may be found all over the body but are rare in certain location including those found in the thorax. After exhaustive review of related literatures, we found only 20 cases of mediastinal hemangiomas published from 1952 up to the present, 30% of which, presented with pleural effusion. In our review of hemangiomas of the lungs, we came across four cases available in literature, two of those were asymptomatic, while the other two presented with hemoptysis. Furthermore, we found out that there is only one case of pleural hemangioma that has been published, a case report by S Nanaware, et al. of a pleural hemangioma associated with hemorrhagic pleural effusion. Clinical presentation may involve a wide spectrum of symptoms depending on their size and involvement of adjacent structures. Patient can be asymptomatic in almost half of the case and may have progressive dyspnea. It may present with recurrent pleural effusion, just like in this case, masquerading as advanced malignancy. The most widely used imaging studies include MRI and CT scan. Treatment of hemangiomas should be individualized, depending on the location of the tumor mass, and the depth of its infiltration. Comprehensive treatment modalities are recommended and includes dry ice cryotherapy, radiotherapy, steroid treatment, sclerosing agent injection, vascular ligation, vascular embolism and surgical excision.

CONCLUSIONS: Hemangiomas, due to its nonspecific symptoms may be easily misdiagnosed. Its diagnosis depends primarily on imaging and pathological examinations. As in this case, imaging and cytology was inconclusive, not until we did the immunohistochemical examination did we arrive to a diagnosis.

Reference #1: Ginhardt TD: Cavernous hemangioma of the Superior mediastinum: am J Surg Pathol 1979:3:353-361

Reference #2: S Nanaware*, D Gothi**, JM Joshi*** Hemorrhagic Pleural Effusion Due to Pleural Hemangioma: JAPI • VOL. 51 • JUNE 2003

Reference #3: Naoyuki Yoshino, MD, Daisuke Okada, MD, Hideki Ujiie, MD. Ann Thorac Cardiovasc Surg 2012; 18: 247-250

DISCLOSURE: The following authors have nothing to disclose: Cherry Ann Dionzon-Serra, Ricardo Salonga, Rodolfo Dizon, Ricardo Beroncal, Jose Edzel Tamayo

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