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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 4  |  Issue : 1  |  Page : 13-14

Pulmonary hyalinizing granuloma involving the diaphragm and the pericardium


1 Department of Pathology, JSS Medical College, JSS University, Mysore, Karnataka, India
2 Department of Forensic Medicine, JSS Medical College, JSS University, Mysore, Karnataka, India

Date of Web Publication28-Jul-2014

Correspondence Address:
Haranahally Raghavan Vanisri
Department of Pathology, JSS Medical College, JSS University, Mysore, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2230-7095.137615

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  Abstract 

Pulmonary hyalinizing granuloma (PHG) is a rare disorder and is a pertinent differential for lung diseases with multiple pulmonary nodules. Natural history of this disease is not known. Although the usual course is benign, a close follow-up of these cases is necessary. We herein report a case of PHG involving the diaphragm and the heart, detected on autopsy in a 57-year-old male who suffered a road traffic accident.

Keywords: Diaphragmatic nodules, pericardium, pulmonary hyalinizing granuloma


How to cite this article:
Vanisri HR, Suchitha S, Chandrakanth H, Manjunath GV. Pulmonary hyalinizing granuloma involving the diaphragm and the pericardium. Int J Stud Res 2014;4:13-4

How to cite this URL:
Vanisri HR, Suchitha S, Chandrakanth H, Manjunath GV. Pulmonary hyalinizing granuloma involving the diaphragm and the pericardium. Int J Stud Res [serial online] 2014 [cited 2019 Jun 20];4:13-4. Available from: http://www.ijsronline.net/text.asp?2014/4/1/13/137615


  Introduction Top


Pulmonary hyalinizing granuloma (PHG) is a rare disease with distinct fibrosing lesions of the lung characterized by central whorled deposits of lamellar collagen. It has been reported that PHG is accompanied by extra-pulmonary fibrous lesions at various sites including the kidney, tonsils and thyroid glands.[1] PHG presents as pulmonary nodules with non specific symptoms of cough, hemoptysis, chest pain and shortness of breath. When asymptomatic, it is usually detected on routine chest radiograph. An immune response to the antigenic stimuli by infection or autoimmune process has been postulated in the pathogenesis but the precise etiology remains obscure [2]. The lesion can be situated in the lung parenchyma or sub pleura. Due to their behavior, a biopsy is required to establish the primary diagnosis of PHG [3]. To the best of our knowledge, this is the first encounter with a case of pulmonary hyalinizing granuloma with involvement of the pleural surface of the diaphragm and heart without involvement of the lung parenchyma.


  Case Report Top


A medico legal autopsy was performed at our institute on a 57-year-old male who died following a road traffic accident. History obtained from his relatives and medical records revealed him to be a chronic smoker for past 30 years and a known hypertensive since 3 years, and on single drug antihypertensive therapy. There was no history suggestive of any infection or autoimmune disease. Autopsy revealed multiple, bilateral, well-circumscribed, rubbery white nodules on the diaphragm, largest measuring 3 × 4 cm and the smallest 1 × 1 cm. There was no pulmonary involvement [Figure 1]a] on gross appearance. In addition to the diaphragmatic nodules, careful examination of the pericardium also showed presence of two such similar nodules [Figure 1]b]. Histopathological examination of these individual nodules showed bundles of lamellar hyalinized collagen arranged in parallel and whorl configuration [Figure 2]a], admixed perivascular lymphoplasmacytic infiltration was noted [Figure 2]b]. There was no evidence of granulomas or areas of necrosis in the multiple sections that were studied.
Figure 1 (a) Bilateral irregular multiple white nodules on the diaphragm with normal lung below (b) Irregular white nodules on the pericardial surface of the heart

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Figure 2 (a) Homogenous hyalinized lamellae of collagen arranged in parallel formation (H and E, ×100) (b) Perivascular lymphoplasmacytic aggregates around hyalinized collagen bands (H and E, ×200)

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Masons' trichrome stain and van Gieson's stain were done, which confirmed the presence of collagen. The nodular section was also subjected to special stains like acid fast and Congo red to rule out mycobacterial involvement and amyloidosis respectively. A final diagnosis of PHG was made that invariably involved the diaphragm and the pericardium.


  Discussion Top


PHG is a rare benign condition first described in 1977, which usually manifests as multiple bilateral pulmonary nodules of lamellar hyaline collagen deposits.[4] It usually affects people of age 19-77 years with a mean age of 44 years at the time of presentation and has no gender predilection [4]. Size of the tumor varies from several millimeters to 15 cm in greatest dimensions and 73% of such patients have multiple lesions [5]. Majority of the patients are asymptomatic, which correlated with the present case.

The etiology of PHG is unknown, but it has been associated with immunologic or infectious diseases such as rheumatoid arthritis, sclerosing mediastinitis, retroperitoneal fibrosis, uveitis, occulopapillitis, tuberculosis, histoplasmosis and aspergillosis [3]. Neoplastic diseases have rarely been reported which include abdominal lymphoma, multiple myeloma, Paget's disease of breast and astrocytoma [6]. The present case did not have any previous history of autoimmune or infective diseases or precisely, no medical records were available for the same. The lesion can be situated in the lung parenchyma or subpleura [3]. PHG is sometimes accompanied by extra-pulmonary fibrous lesions at other sites, and coexistence of PHG along with laryngeal and subcutaneous nodules have been reported [1]. The present case did not show extra pulmonary fibrous lesions at other sites.

The disease follows a relatively benign course with the nodules showing increasing in size over a period of years. There are two reported cases of PHG complicated by lymphoma. Therefore, a follow-up is utmost essential in such cases [2]. Prognosis of PHG is excellent with no significant impact on longevity [4]. There have been reports of patients who responded well to corticosteroid therapy [7]. Surgical resection is the treatment of choice [8].


  Conclusion Top


Diagnosis of PHG requires a histological examination, and therefore, a biopsy is essential. In previously reported cases, most lesions occurred in the lung with extra-pulmonary manifestations. This is a rare case of PHG involving the diaphragm and the heart without involvement of the lung, discovered incidentally at autopsy.

 
  References Top

1.Shinohara T, Kaneko T, Miyazawa N, et. al. Pulmonary hyalinizing granuloma with laryngeal and subcutaneous involvement: report of a case successfully treated with glucocorticoids. Intern Med 2004;43(1):69-73.  Back to cited text no. 1
    
2.Khilnani GC, Kumar A, Gupta SD, Surendranath A, Sharma S. Pulmonary hyalinizing granuloma presenting with dysphagia. J Assoc Physicians India 2003;51(5) : 519-21.  Back to cited text no. 2
    
3.Esme H, Ermis SS, Fidan F, Mehmet U, Fatma HD. A case of pulmonary hyalinizing granuloma associated with posterior uveitis. Tohoku J Exp Med 2004;204(1):93-7.  Back to cited text no. 3
    
4.Zeiden A, Adal-El-Badrawy. Pulmonary hyalinizing granuloma is a possible cause of lung mass. EJB 2011;5(1):43-5.  Back to cited text no. 4
    
5.Agrawal D, Deshpande R, Maheshwari S, Patel A, Udwadia ZF. Pulmonary hyalinizing granuloma with ureteric fibrosis: a case report and review of relevant literature. Indian J Chest Dis Allied Sci 2006;48(4):283-5.  Back to cited text no. 5
    
6.Ren Y, Raitz EN, Lee KR, Pinglrton SK, Tawfik O. Pulmonary small lymphocytic lymphoma (mucosa-associated lymphoid tissue type) associated with pulmonary hyalinizing granuloma. Chest 2001;120(3):1027-30.  Back to cited text no. 6
    
7.Na KJ, Song SY, Kim JH, Kim YC. Subpleural pulmonary hyalinizing granuloma presenting as a solitary pulmonary nodule. J Thorac Oncol 2007;2(8):777-9.  Back to cited text no. 7
    
8.Yousem SA, Hochholzer L. Pulmonary hyalinizing granuloma. Am J Clin Pathol 1987;87(1):1-6.  Back to cited text no. 8
    


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  [Figure 1], [Figure 2]



 

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