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A 10 years old girl presented with fever, cough, haemoptysis, dyspnoea on exertion of two months duration. Chest skiagram and CT-scan of thorax revealed well defined mass lesion of varying density occupying right upper and middle lobes with areas of calcification. On thoracotomy, a huge mass in the right upper lobe was resected and hispathological examination proved it to be a case of plasma cell granuloma. [J Indian Med Assoc 2007; 105: 92-3 & 98]
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Key words : Plasma cell granuloma.
Plasma cell granuloma is uncommon benign lesion of lung that may show local aggressive behaviour. It occurs
predominantly in children and young adults and majority of the patients are symptomless and chest radiography in majority of cases show solitary well circumscribed nodule. Pre-operative diagnosis is rarely confirmed. Treatment of choice is surgical resection. In general, recurrences and metastases are not detected.
Case Report
A 10-year-old girl presented with complaints of fever, cough with haemoptysis, dyspnoea on exertion for last two months.
Examination — She was thin built, febrile, preferred to lie in propped up position. Her facies showed prominent malar eminence with depressed nasal bridge. Conjunctiva was muddy. There were mild pallor and clubbing. Systemic examination revealed accessory muscles of respiration were working. There was mild bulging of right side of the anterior chest wall, trachea shifted to left side, apex beat lied on left fourth intercostal space on midclavicular line and there was diminished movement, dullness and decreased breath sound in the right infraclavicular, mammary and inframammary region. Liver was palpable 2cm in midclavicular line, margin was smooth and non-tender.
Investigations — Her haemoglobin was 10.6 g/dl, RBC 2.8 million/cmm, WBC 9300/cmm with neutrophils 48%, lymphocytes 44%, eosinophils 7% and monocytes 1%.; ESR 160mm/hour, platelets adequate and reticulocytes < 1%. The levels of blood sugar, urea, creatinine were within normal limit. Her serum protein was 11.8 g/dl with albumin and globulin ratio was 1:3.5. Aldehyde test was positive. Bone marrow examination showed hypercellular marrow, adequate erythroblasts, granulocytes relatively increased with slight increase in lymphoid cells, megakaryocytes were increased but active, plasma cell increased to 8-10%; M:E was 6:1. Immunoprofile study showed IgG level as 395mg/dl, IgA 359 mg/dl, IgM 295mg/dl. Urine for Bence Jones protein was negative.
Chest skiagram revealed a well defined mass in right upper and middle hemithorax (Fig 1). CT-scan of thorax revealed well defined mass lesion of varying density occupying right upper and middle lobes with areas of calcification.
Surgery — On thoracotomy a huge mass was seen in right upper lobe engulfing azygos vein and superior vena cava. Debulking of the mass done through right posterior intercostal space. Mass could not be removed totally because of adherence to those structures. Few lymph nodes were also excised. Chest was closed after two intercostal drains. The postoperative course was uneventful.
Histopathology — There was infiltration by non-specific mononuclear and plenty of plasma cells within fibrous stroma which was suggestive of plasma cell granuloma (Fig 2, H&E, x 400).
Follow-up — She has been on regular follow-up over an year with no complaints.
Discussion
Plasma cell granuloma is not a true neoplasm and is a rare lesion that was first described in the lung in 19391. It has been subsequently reported in various locations including liver, spleen, lymph node, salivary gland, breast, soft tissue, skin2. The pathogenesis is unclear but a dual concept of their pathogenesis has arisen, either representing an unusual chronic inflammatory reaction to an unidentified agent or a reparative process secondary to a pulmonary infection. In general neither recurrences nor metastases are detected following surgical excision but, in some cases, however, there is evidence of local aggressive behaviour.
It occurs predominantly in children and young adults. Although it constitutes less than 1% of all lung tumours, it is reported to be the most common cause of solitary lung masses in children3. It occurs with equal incidence in men and women and 1/3rd of the patients has history of upper respiratory tract infection or pneumonia2,4. Most of the patients are asymptomatic, the lesion being discovered on a screening chest roentgenogram. Symptoms when present include cough, haemoptysis, chest pain; signs and symptoms of bronchial or tracheal obstruction may present in some patients. Deaths have been reported due to extrinsic compression of mediastinal structure5.
Roentgenographic manifestation consists of either a solitary pulmonary nodule or homogenous area of consolidation that can mimic primary or metastatic neoplasm. Calcification may present occasionally and cavitation rarely present. Endobronchial tumour can cause collapse and obstructive pneumonitis. It is uncommonly manifested as multiple pulmonary nodules. Hilar or mediastinal adenopathy, small pleural effusion are also reported.
Histologically it is composed of predominantly plasma cells and lesser number of lymphocyte, giant cells, histiocyte in a fibrous stroma. Various other tumour like fibrous histiocytoma, histiocytoma or xanthoma, fibroma, etc, are described depending upon predominance of above mentioned cells. While recognising practical difficulty in making a distinction in some cases, it is considered that majority of these tumours are inflammatory, nevertheless recurrence and local tissue infiltration and destruction suggest that some may represent a true neoplastic proliferation. Plasma cell granulomas often locally invade mediastinum, diaphragm, chest wall, vertebral bodies and major vessels6. The natural course is usually either a slow growth or spontaneous resolution7.
Because pre-operative imaging is indistinguishable from that of malignancy and diagnosis by transthoracic fine needle aspiration or transbronchial biopsy is difficult as tumour has variable cellular composition and because it can demonstrate aggressive behaviour and has overlapping clinical and pathological features with inflammatory sarcoma, complete surgical excision is general treatment of choice1,8. Recurrence after resection is rare but have been reported7. In general, neither recurrences nor metastases are detected following surgical excision; in a minority of cases, however, there is evidence of local aggressive behaviour.
References
1 Copin MC, Gosselin BH, Ribet ME — Plasma cell granuloma of the lung: difficulties in diagnosis and prognosis. Ann Thorac Surg 1996; 61: 1477-82.
2 Anthony PP — Inflammatory pseudotumour (plasma cell granuloma) of lung, liver and other organs. Histopathology 1993; 23: 501-3.
3 Cohen MC, Kaschula RO — Primary pulmonary tumors in childhood : a review of 31 years experience and the literature. Pediatr Pulmonol 1992; 14: 222-32.
4 Matsubara O, Tan-Liu NS, Kenney RM, Mark EJ — Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous hitiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988; 19: 807-14.
5 Shochat S — Tumours of the lung . In: Oneill JA, Rowe MI, editors. Pediatric Surgery. Vol 1.5th ed. St Louis: Mosby, 1998: 921-5.
6 Singh RS, Dhaliwal RS, Puri D — Inflammatory pseudotumour of the lung: report of a case and of literature. Indian J Chest Dis Allied Sci 2001; 43: 231.
7 Balachandran A, Shivbalan S — Plasma cell granuloma of lung. Indian Pediatr 2004; 41: 292-3.
8 Bahadori M, Liebow AA — Plasma cell granuloma of lung. Cancer 1973; 31: 191-208.
Department of Chest Medicine, RG Kar Medical College, Kolkata 700004
*MD, Assistant Professor
**DCH, MD, RMO cum Clinical Tutor
***MD, Assistant Professor, Department of Pathology, SSKM Hospital, Kolkata 700020
****MS, MCh, Professor, Department of Cardiothoracic and Vascular Surgery, Medical College, Kolkata 700073
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