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Follicular B-Cell Lymphoma Embedded in a Pulmonary Chondroid Hamartoma: A Case Report

Background: Here we report a rare case of concomitant pulmonary chondroid hamartoma and follicular non Hodgkin lymphoma in a 65-year-old woman with a history of 4-month nonspecifi c symptoms like fever, sweating and weight loss. Methods: Chest CT scan revealed a 12-mm nodule with relatively regular margins in the right middle lung fi eld, adhering to the chest wall, with no involvement of hilar or mediastinal lymph nodes. The patient underwent thoracoscopy and resection of the lesion. Following histological exam and immunohistochemical staining, the patient was diagnosed as having follicular b-cell lymphoma embedded in a pulmonary chondroid hamartoma. FISH analysis was performed and showed the presence of typical follicular lymphoma chromosomal translocation t (14;18) (q32;q21). Conclusions: Following the fi nal diagnosis, we decided not to refer the patient to chemotherapy and she enter watch and wait regimen with imaging control every 6 months. After 18 months, the chest X-ray and abdominal ultrasound are still negative with no evidence of recurrence. Case Report Follicular B-Cell Lymphoma Embedded in a Pulmonary Chondroid Hamartoma: A Case Report Lara Mannelli1*, Sofi a Kovalchuk1, Benedetta Puccini1, Gemma Benelli1, Catia Dini2, Alberto Bosi1 and Luigi Rigacci1 1Department of Hematology, AOU Careggi General and Academic Hospital, Florence, Italy 2Department of Radiodiagnostic, AOU Careggi, General and Academic Hospital, Florence, Italy Dates: Received: 17 February, 2017; Accepted: 08 March, 2017; Published: 09 March, 2017 *Corresponding author: Lara Mannelli, Department of Hematology, AOU Careggi General and Academic Hospital, Florence, Italy, Tel: 055 794 5296; E-mail:


Introduction
Pulmonary chondroid hamartoma (PCH) is the most common benign neoplasm of the lung with an incidence of 0.25% [1], and accounts for 7-14% of all solitary lung nodules [2].
PCH are usually well circumscribed from the lung parenchyma or protude into the bronchial lumen when located in the bronchus [3].
Primary pulmonary lymphomas (PPL) are extremely rare, representing less than 1% of primary malignant lung cancers, fewer than 1% of malignant lymphomas, and accounting for only 3.6% of extranodal lymphomas [4].
Here we report a rare case of pulmonary chondroid hamartoma containing grade I-II follicular lymphoma in a 65-yr-old female patient.

Case Description
A 65 year-old woman presented with dry cough and progressive dyspnea. She experienced 10 kg of weight loss during 4 months and reported nocturnal fever and sweating.
The patient was a chain-smokers with prior history of asthma.
She was admitted to a hospital and received broad-spectrum antibiotics with no improvement. In her second hospital admission, no focal fi ndings were noted on examination, especially no palpable lymphnodes and no hepatosplenomegaly.
A complete blood test showed a white cell count of 6.21x109 cells/l and the percentage of neutrophil was 54.9%. The renal and hepatic function and serum lactate dehydrogenase levels were unremarkable.
Chest CT scan revealed a 12-mm nodule with regular margins in the right middle lung fi eld, adhering to the chest wall. No hilar or mediastinal lymph nodes were signifi cantly visible (Figures 1a,b,2).
According to thoracic surgeon, the nodular lesion was enucleated, with the rest of the lung being considered normal and not resected. After 18 months, the chest X-ray and abdominal ultrasound are still negative with no evidence of recurrence.
The onset of the tumor is generally in the adulthood (peak age incidence in the sixth decade) (2) with a male: female ratio being 2:1 to 3:1 [5].
Chondroid hamartomas are usually solitary and well circumscribed, with dimensions usually ranging from 1 to 4 cm [5]. Rarely, they may be larger and multiple. Based on the localization, PCH may be distinguished into two types, peripheral parenchymal type and central endobronchial type.
The former comprises over 90% of all chondroid hamartomas, arises from small bronchi and is generally asymptomatic. The latter arises from large bronchi, is less frequent but is often symptomatic [5].
PCHs are frequently discovered on routine X-ray chest and they appear as solitary coin lesions. Less commonly, they may show up as multiple coin lesions [2] or as cavitary lesions in case of cystic PCH.
Some cases show popcorn pattern of calcifi cation. Transthoracic FNAC can be used to establish the diagnosis [5]. The cytologic diagnosis is based on the recognition of mesenchymal components with fi bromyxoid and chondroid elements.
PCH histologically consists of islands of cartilage, fat, fi bromyxoid stroma, and narrow spaces lined by respiratory epithelium. [5].
Recently, cytogenetic studies have demonstrated an abnormal karyotype in several instances. Thus, it was proposed that PCH represents a true benign mesenchymal neoplasm instead of a developmental abnormality [1].
Recurrence of chondroid hamartoma is extremely unusual [5] and malignant transformation is exceptional.
Primary pulmonary lymphomas are extremely rare and represent <1% of primary malignant lung tumors, <1% of lymphomas, and only 3%-4% of extranodal lymphomas [6] PPL is defi ned as clonal lymphoid proliferation affecting one or both lungs [7], lobes or primary bronchus, with or without mediastinal involvement, and no evidence of extrathoracic lymphoma at the time of diagnosis or for 3 months thereafter.
The most common PPL type is mucosa-associated lymphoid tissue (MALT) lymphoma, An extranodal marginal zone lymphoma that accounts for 80%-90% of PPL cases.
DLBCL is the second most common type of PPL  is a localized alveolar opacity [6], less than 5 cm-diameter and blurred or well-defi ned contours (according to the series); PPL is associated in nearly 50% of cases with an air bronchogram [6], peribronchial disease with a halo of ground-glass shadowing and proximal bronchiectasis.
PPL has low FDG involvement, so the benefi t of FDG-PET in the clinical assessment of this kind of lymphoma is still unclear [6].
Although the diagnostic procedure of this disease has not been well defi ned, open thoracotomy or chest CT-guided percutaneous biopsy are the preferred methods used [8].
The positive value of open thoracotomy biopsy is high, but it is associated with high medical cost and clinical risk [8].
By contrast, the diagnostic value of chest CT-guided percutaneous biopsy has a low medical cost and less serious complications. However, the procedure is limited for the diagnosis of masses that are in or adjacent to the hilum or mediastinum [8].
Although there is no consensus on the treatment of PPL, surgical resection is generally performed in cases of localized tumors. Because the rate of local recurrence after complete resection is reported to be high, chemotherapy or radiation therapy should be considered for non-indolent lymphomas [6].
In the present case, a nodular opacity with regular margins was detected by CT-scan in a patient symptomatic for dyspnea, cough, chest pain, as well as fever and weight loss.
The pathologist confi rms the presence of follicular b-cell lymphoma embedded in a chondroid hamartoma, and we didn't fi nd a similar cases in the previous reported studies.
The patient was considered a surgical candidate because of the neoplasm localization.
Following the fi nal diagnosis, we decided not to refer the patient to chemotherapy and she enter watch and wait regimen with imaging control every 6 months.
After 18 months, the chest X-ray and abdominal ultrasound are still negative with no evidence of recurrence.