A 52-Year-Old Man Presented With Cough, Chest Pain, and a Mass in the Right Lung.

CASE PRESENTATION: A 52-year-old man, current smoker with a 50 pack-year history, presented to our department with cough, yellow sputum, and localized right chest pain. Chest radiograph revealed a large mass in the right upper lobe. He denied the presence of fever, night sweats, or weight loss. He has a medical history of COPD and anxiety disorder. He was receiving long-acting beta agonists/long-acting muscarinic antagonists as a treatment for COPD and quetiapine 100 mg for anxiety disorder.

Application of endobronchial ultrasound-guided transbronchial needle aspiration in the diagnosis and treatment of mediastinal lymph node tuberculous abscess: a case report and literature review.

BACKGROUND: This study aimed to report the experience of diagnosis and treatment of one rare case of mediastinal lymph node tuberculous abscess (MLNTA) using endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA). CASE PRESENTATION: An 18-year-old female patient was hospitalized in the Affiliated Hospital of Xuzhou Medical University in November 2017, due to intermittent left chest pain. She was suspected of infecting tuberculosis (TB) and thus received anti-TB treatment. Since April 1, 2018, she began to exhibit symptoms of chest distress. The patient was then admitted to Shanghai Pulmonary Hospital and continued receiving systemic anti-TB treatment during the whole course. On April 11, 2018, she received EBUS-TBNA to puncture pus and inject isoniazid. Simultaneously, the pus was sent for cytopathological and bacteriological examination, both supporting the diagnosis of TB in the patient. On April 24 and May 10, she received two times of EBUS-TBNA treatment. The symptoms of chest distress were relieved, but granulomatous neoplasm occurred at the EBUS-TBNA site on the trachea wall. The patient then received local clamp removal and cryotherapy on May 29 and Jul 19, respectively. Chest computed tomography (CT) reexamination on September 28 revealed that the MLNTA lesion had been completely absorbed, and electronic bronchoscopic reexamination on September 30 demonstrated that the granulomatous neoplasm on the trachea wall was entirely invisible. CONCLUSIONS: Using EBUS-TBNA to puncture and aspirate pus and inject drugs can be effectively used to diagnose and treat MLNTA, which provides a new, less invasive, safe and reliable method for diagnosis and treatment of MLNTA.

The first case of multiple pulmonary granulomas with amyloid deposition in a dental technician; a rare manifestation as an occupational lung disease.

BACKGROUND: Occupational lung diseases, such as pneumoconiosis, are one of the health problems of dental workers that have been receiving increasing interest. Pulmonary amyloidosis is a heterogenous group of diseases, and can be classified into primary (idiopathic) and secondary (associated with various inflammatory diseases, hereditary, or neoplastic). To date, the development of pulmonary amyloidosis in dental workers has not been reported. CASE PRESENTATION: A 58-year-old Japanese female presented with chest discomfort and low-grade fever that has persisted for 2 months. She was a dental technician but did not regularly wear a dust mask in the workplace. Chest X ray and computed tomography revealed multiple well-defined nodules in both lungs and fluorodeoxyglucose (FDG)-positron emission tomography revealed abnormal FDG uptake in the same lesions with a maximal standardized uptake value (SUV [max]) of 5.6. We next performed thoracoscopic partial resection of the lesions in the right upper and middle lobes. The histological examination of the specimens revealed granuloma formation with foreign body-type giant cells and amyloid deposition that was confirmed by Congo red staining and direct fast scarlet (DFS) staining that produce apple-green birefringence under crossed polarized light. Because there were no other causes underlying the pulmonary amyloidosis, we performed electron probe X-ray microanalysis (EPMA) of the specimens and the result showed silica deposition in the lesions. Based on these results, we finally diagnosed the patient with pulmonary granulomas with amyloid deposition caused by chronic silica exposure. Afterward, her symptoms were improved and the disease has not progressed for 2 years since proper measures against additional occupational exposure were implemented. CONCLUSIONS: Our case presented three important clinical insights: First, occupational exposure to silica in a dental workplace could be associated with the development of amyloid deposition in lung. Second, EPMA was useful to reveal the etiology of amyloid deposition in the lungs. Last, proper protection against silica is important to prevent further progression of the disease. In conclusion, our case suggested that occupational exposure to silica should be considered when amyloid deposition of unknown etiology is found in the lungs of working or retired adults.

Pulmonary hyalinizing granuloma presenting with dysphagia: a rare presentation.

A 25-year-old man presented with a 2-month history of dysphagia and past history of pulmonary and intestinal tuberculosis. A barium swallow showed a point of constriction 42 mm above the gastroesophageal junction. Computed tomography revealed large opacities in bilateral lung fields, encroaching more on the esophagus. The lesion progressively compressed the esophagus as it moved inferiorly. A right posterolateral thoracotomy was performed for sub-anatomical resection of the mass. A biopsy revealed homogenous whirling hyalinized collagen fibers, highly suggestive of pulmonary hyalinizing granuloma, with no evidence of malignancy. Pulmonary hyalinizing granuloma should be considered in the differential diagnosis of longstanding dysphagia.

Pulmonary manifestation of immunoglobulin G4-related disease in a 7-year-old immunodeficient boy with Epstein-Barr virus infection: a case report.

BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) is a multiorgan fibroinflammatory condition with lymphoplasmacytic infiltrates containing abundant IgG4-positive plasma cells. The immunopathogenesis of the disease and the potential role of triggering autoantigens or infectious factors have not been clearly defined. Immunoglobulin G4-related lung disease is a new and emerging condition in pediatric patients and to date, there have been only two reports regarding pulmonary manifestation of IgG4-RD in children recently published. This is the first report of IgG4-related lung disease in an immunodeficient child with Epstein-Barr virus infection. CASE PRESENTATION: We report on the case of a 7-year old atopic boy who was hospitalized with an initial clinical and radiological diagnosis of pneumonia, positive Epstein-Barr virus (EBV)-DNA in the blood and defective adaptive immunity. The lung CT showed a consolidated mass lesion adjacent to the posterior wall of the chest and the diaphragm. The child underwent surgical resection of the tumor, and the histologic examination of the lung specimens revealed lymphoplasmacytic infiltrates with fibrosis and vasculitis correlating with IgG4-related lung disease. Subsequent monitoring of the patient with lung CT, pulmonary function tests and IgG4 levels did not show signs of active disease. CONCLUSIONS: The diagnosis of IgG4-related lung disease in children is challenging because of its rarity, nonspecific symptomatology and heterogeneous morphological manifestations. Further studies are required in children with pulmonary presentation of IgG4-RD to better understand pathogenesis of this condition, possible immunological or infectious triggering factors, and finally, to determine pediatric patient-targeted therapeutic interventions.

Granulomatous inflammation of pulmonary squamous cell carcinoma: a rare phenomenon.

Some neoplasms are associated with granulomatous inflammation. Granuloma formation in tumor tissue is caused by the cytokines derived from either the main tumor or other cells surrounding the tumor. In other instances, granulomatous inflammation is observed in the lymph nodes draining a tumor. This has been recognized as a sarcoid-like reaction. Herein, we report of a 75-year-old man with pulmonary squamous cell carcinoma (SCC), where granulomatous inflammation was observed extensively at the primary site. The carcinoma seemed to partly regress. In the regressing area, tumor cell debris was surrounded by granuloma. In contrast, no granuloma was identified in the dissected regional lymph nodes. To the best of our knowledge, such a case of SCC had not been described thus far. More case studies are required to determine whether tumor-related granuloma is the main cause of regression or whether it is just a secondary phenomenon caused by the attack and destruction of the tumor by lymphocytes.

[Pulmonary hyalinizing granuloma]. / Pulmonale hyaline Granulomatose.

Benign lesions as pulmonary hyalinizing granuloma may mimic a malign disease. A 63-year old patient complained dyspnea and a weight loss of 30 kg. CT-thorax scans showed a destructive and infiltrative pulmonary process with pleural thickening. Histologic examination of transbronchial and transthoracic biopsies as well as of biopsies taken by minithoracotomy was not conclusive. Due to further progression the patient underwent a left-sided pleuropneumonectomy despite a VO2 peak of 9 ml/kg/min. Histology revealed DIP-like infiltrations, a histiocytic reaction and hyaline granulomas. Among less than 100 published cases of pulmonary hyaline granuloma a comparable rapid progression with a total functional loss of the affected lung is not reported. Mostly hyalinizing granuloma presents with infiltrations, which may mimic lung cancer, or nodular lesions, partly with cavitations or calcifications. The etiology is unknown, a persistent immunologic response to an antigenic stimulus is discussed. Associations with infections, lymphomas, amyloidosis or IgG4-related disease are reported. Some cases have features of multifocal fibrosis. In the case reported none of these associations could be found. The prognosis of pulmonary hyaline granuloma is regarded as benign. There is no effective treatment yet. Once the diagnosis has been established a conservative approach as well as a resection of nodules and a therapeutic attempt with steroids are an option. Extensive resections as pleuropneumonectomy are an exception.

A 40-year-old woman with multiple pulmonary nodules. Pulmonary hyalinizing granuloma.

A 40-year-old woman (a nonsmoker) with history of idiopathic thrombocytopenic purpura and a platelet count > 90,000 cells/µL without specific medication was referred to pulmonary clinic for evaluation of multiple pulmonary nodules. The patient presented to an outside hospital with fatigue, lack of energy, and dyspnea on exertion for 2 years. She denied fever, cough, chest pain, or weight loss. An initial chest radiograph showed bilateral multiple pulmonary nodules. A chest CT scan revealed multiple nodular lesions, varying in size, in all lobes of both lungs. There was no mediastinal lymphadenopathy or pleural effusion. There was no significant hypermetabolic activity on a subsequent fluorodeoxyglucose PET scan/CT scan, and there had been no significant change. She underwent CT scan-guided percutaneous transthoracic biopsy and bronchoscopy with transbronchial biopsies, all of which were inconclusive. An open lung biopsy was considered.

Recurrence of sarcoid granulomas in lung transplant recipients is common and does not affect overall survival.

BACKGROUND: Sarcoidosis represents 2,5% of all indications for lung transplantation and criteria are generally assumed to be the same as for pulmonary fibrosis. Recurrence of granulomas in transplanted lungs has earlier been proved to derive from recipient immune cells, but its role in relation to lung function and overall survival after lung transplantation remains uncertain. OBJECTIVE: To identify recurrent granuloma in transbronchial biopsies in patients receiving lung transplant because of sarcoidosis, and relate the findings to overall survival and lung function. DESIGN: A total of 620 patients were transplanted at this centre from 1992 until august 2012. This study comprised all patients (n=25) transplanted due to pulmonary sarcoidosis. Lung functions, trans-bronchial biopsies, and survival were compared in patients with and without recurrence of granulomas. Granulomas were defined as non-necrotizing epitheloid granulomas with multinucleated giant cells according to standard criteria (formation of epitheloid giant cells) without presence of infection. CONCLUSIONS: Approximately 30% of lung transplant recipients due to sarcoidosis have recurrence of sarcoid granulomas. Recurrence of granulomas does not affect overall survival or lung function.

Pulmonary hyalinizing granuloma detected in a family member after confirmation of tuberculosis in his father.

Pulmonary hyalinizing granuloma (PHG) is an uncommon lung disease that usually presents as bilateral multiple nodules, and more rarely as a solitary nodule. An exaggerated immune response to antigenic stimuli resulting from infection or an autoimmune process has been suggested as the cause of PHG. Here, we describe a rare case of solitary PHG that was detected in a family member after tuberculosis had been confirmed in his father, without any background of infectious disease or autoimmune abnormality.

Bronchocentric granulomatosis in a patient with rheumatoid arthritis.

Bronchocentric granulomatosis is an uncommon entity which has no specific clinical, radiological and immunological features. It is usually diagnosed at morphological examination of biopsy or resected lung material. Aetiology of bronchocentric granulomatosis is unclear. A 49-year-old female patient, who was followed up with diagnosis rheumatoid arthritis in our outpatient clinic, presented with right lobe nodular lesion in chest radiography. Right thoracotomy and wedge resection was performed. Pathological examination revealed bronchocentric granulomatosis. Bronchocentric granulomatosis has been rarely reported in rheumatoid arthritis. This case might be a proof that bronchocentric granulomatosis may be one of the respiratory manifestations of rheumatoid arthritis.

Pulmonary hyalinizing granuloma mimicking multiple lung metastases: report of fluorodeoxyglucose positron emission findings.

Pulmonary hyalinizing granuloma (PHG) is a rare disease characterized by multiple bilateral pulmonary nodules of uncertain etiology. We describe a 71-year-old female patient with thyroid papillary carcinoma in whom bilateral pulmonary nodules were found during a routine chest radiography examination. Subsequent fluorodeoxyglucose positron emission tomography/computed tomography scan gave the impression of multiple pulmonary metastases based on high maximum standardized uptake value. She underwent video-assisted thoracoscopic surgery with wedge resection, and PHG was diagnosed on the basis of histopathologic findings. To our knowledge, this is the first report of PHG developing in a patient as a solid cancer, mimicking multiple pulmonary metastases. We also present the first description of positron emission tomography in PHG, according to a Medline search.

Pulmonary tumorlet: a case report of a diagnostic pitfall in cytology.

BACKGROUND: Pulmonary tumorlets are usually an incidental pathologic curiosity of no clinical importance, but may be mistaken for epithelial and nonepithelial neoplasms. Fine needle aspiration (FNA) of this cell proliferation has rarely been reported. We describe a pulmonary tumorlet associated with bronchocentric granulomatosis presenting as a tumorous consolidation on chest radiograph. CASE: In a hitherto healthy 70-year-old man admitted for acute respiratory infection, a solid consolidation was found on chest radiograph. Medical history was uneventful except right-sided pleurisy in 1949. Computed tomography-guided FNA sample was composed of loose clusters of small columnar cells with cyanophilic cytoplasm and centrally located round to oval nuclei. With a tentative diagnosis of well-differentiated adenocarcinoma, lumpectomy was performed. Intraoperative cytology demonstrated lymphocytes, epithelioid cells, giant cells of Langerhans type and clusters of columnar cells. Definitive histologic examination confirmed the intraoperative diagnosis of necrotizing granulomatosis and tumorlet. Neuroendocrine origin of the cells was confirmed by immunocytochemical and immunohistochemical studies resulting in strong reactivity of the cells to synaptophysin, NSE, chromogranin A and N-Cam. CONCLUSION: Knowledge of the cytomorphologic presentation of tumorlets in FNA and consideration of the appropriate differential diagnoses combined with ancillary studies might have prevented lung resection.

Comparison of fungal culture versus surgical pathology examination in the detection of Histoplasma in surgically excised pulmonary granulomas.

CONTEXT: Granulomatous pulmonary nodules are common in areas endemic for Histoplasma infection, and may require surgical excision to exclude neoplasia. Surgeons may elect to routinely send material directly to the clinical microbiology laboratory for fungal and mycobacterial cultures. OBJECTIVE: To determine if tissue from surgically excised pulmonary granulomatous nodules removed from patients in a geographic area endemic for Histoplasma infection should be routinely submitted for fungal culture. DESIGN: Retrospective review and comparison of surgical pathology histochemical findings and clinical microbiology results of 30 surgical (wedge) lung excisions that demonstrated granulomatous nodule at the time of frozen section. RESULTS: Twenty cases demonstrated fungal organisms consistent with Histoplasma species via histochemical fungal stains. Of these 20 cases, 17 were tested in the microbiology laboratory using direct smear examination and fungal culture; Histoplasma was detected in 1 case (1/17). Eight cases revealed no organisms by surgical pathology. Of these, 6 were tested in the microbiology laboratory, and all 6 were negative by culture and direct smear (0/6). The remaining 2 cases demonstrated organisms other than Histoplasma by surgical pathology examination. CONCLUSIONS: Surgical pathology examination of granulomatous pulmonary nodules detected Histoplasma organisms with greater sensitivity than culture and direct smear. There were no false-negative surgical pathology diagnoses when compared with microbiological results. These findings suggest that it is not necessary to routinely send material from solitary pulmonary granulomas for fungal culture when the material is removed from immunocompetent patients in a geographic area endemic for histoplasmosis.

Lung transplantation in sarcoidosis.

Lung transplantation (LT) is an appropriate therapeutic option for patients with severe, fibrocystic pulmonary sarcoidosis refractory to medical therapy. Survival rates following LT for sarcoidosis are generally comparable to other indications. Timing of transplantation for patients with sarcoidosis is challenging because mortality rates are high (27 to 53%) among sarcoid patients awaiting LT. Deciding when to refer patients for LT is difficult because models predicting mortality have not been validated. Importantly, algorithms or parameters predicting mortality in idiopathic pulmonary fibrosis and other interstitial pneumonias may not apply to sarcoidosis. Pulmonary function tests do not correlate well with mortality risk in patients with sarcoidosis. However, retrospective studies have shown that the presence of pulmonary arterial hypertension in sarcoidosis is an ominous sign and warrants referral for LT. This article reviews indications and contraindications to LT among patients with sarcoidosis, examines risk factors for mortality, and discusses optimal timing of referral for LT. Early referral of patients allows for timely evaluation of patients for possible listing. If the risks specific to transplantation in sarcoidosis are considered and carefully evaluated, outcomes are reasonable and match those of other diagnoses. Recurrent sarcoidosis in the lung allografts can occur but does not affect survival or risk for complications.

[Pulmonary hyalinizing granuloma mimicking pulmonary carcinoma]. / Akciger kanserini taklit eden pulmoner hiyalinize granülom.

Pulmonary hyalinizing granuloma is a rare fibrosing nodular disease of the lung characterized by solitary or multiple pulmonary nodules. They can occur after inflammatory or post-inflammatory changes. A 60 years old asymptomatic patient admitted to our clinic because of a solid mass of 6 cm in his routine chest radiography. A lobectomy was performed and the histological diagnosis was reported as pulmonary hyalinizing granuloma. This case, mimicking pulmonary carcinoma, is rarely found in the literature.

[Necrotizing sarcoid granulomatosis--case report and review of the literature].

The Necrotizing sarcoid granulomatosis is a rarely met angiitis. The disease is predominantly localized in the lung and has a good prognosis. The authors reported about a patient treated in their clinic; the diagnosis is made after histologic examination.

[Bronchocentric granulomatosis]. / Bronchozentrische Granulomatose -- Seltene pulmonale Herdbildungen.

Bronchocentric granulomatosis is a rare disease, which has to be taken into consideration in the differential diagnostic of pulmonary nodules and tumors. Two cases are presented, in which only open lung biopsy made it possible to get the diagnosis of bronchocentric granulomatosis. A 78-year-old woman presented with multiple small pulmonary nodules with aspergillus colonization, whereas an 83-year-old man presented with a large mass in the left upper lobe. Corticosteroid therapy led to sustained remission in both patients. The case reports will underline that bronchocentric granulomatosis is not a uniform clinical entity, but has variable clinical and radiological appearances. Its former subdivision into two forms differing by the occurrence of asthma with the coexistence of an aspergillus colonization, does not represent the many different manifestations. Until now the etiology of bronchocentric granulomatosis is uncertain. But there are hints, that the granulomatous inflammation might be the uniform response to different causes. The definite diagnosis of bronchocentric granulomatosis can only be made histologically. This mostly requires surgical lung biopsy. With therapy -- either surgery or corticosteroid therapy or the combination of both -- the prognosis is good.