A case of chronic necrotizing pulmonary aspergillosis due toAspergillus nidulans

A 55-year old man without immunosuppression clinically showed a coin lesion in the right lower lung on the chest radiographs.Aspergillus nidulans was isolated and identified in both trans-bronchial lung biopsy specimen and resected tissue. The specimens revealed characteristics of chronic necrotizing pulmonary aspergillosis pathologically. Very few reports on cases of pulmonary aspergillosis due toA. nidulans exist, and we were not able to find any reports of similar cases. This case may be the first reported case of chronic necrotizing pulmonary aspergillosis due toA. nidulans.     

A pulmonary paragonimiasis case mimicking metastatic pulmonary tumor

Pulmonary paragonimiasis is a relatively rare cause of lung disease revealing a wide variety of radiologic findings, such as air-space consolidation, nodules, and cysts. We describe here a case of pulmonary paragonimiasis in a 27-year-old woman who presented with a 2-month history of cough and sputum. Based on chest computed tomography (CT) scans and fluorodeoxyglucose positron emission tomography (FDG-PET) findings, the patient was suspected to have a metastatic lung tumor. However, she was diagnosed as having Paragonimus westermani infection by an immunoserological examination using ELISA. Follow-up chest X-ray and CT scans after chemotherapy with praziquantel showed an obvious improvement. There have been several reported cases of pulmonary paragonimiasis mimicking lung tumors on FDG-PET. However, all of them were suspected as primary lung tumors. To our knowledge, this patient represents the first case of paragonimiasis mimicking metastatic lung disease on FDG-PET CT imaging.     

慢性阻塞性肺疾病合并肺曲菌病一例及文献复习

目的:提高对慢性阻塞性肺疾病合并侵袭性肺曲菌病(COPD合并IPA)临床特点、诊断及治疗的认识.方法:回顾性分析2011年4月收治的一例COPD合并IPA患者的临床资料及诊治经过,并复习相关文献.结果:男患,“咳嗽、咳痰30余年,气短3年,加重1月余”入院,肺部CT示双肺多发结节影、空洞影,经抗炎、抗念珠菌治疗无效,CT下肺结节病灶活检病理示肺曲菌.抗曲菌治疗后症状好转、肺部影像明显吸收.结论:COPD合并IPA正逐渐引起重视,临床特征无明显特异性,肺部影像以结节影、空洞影多见,早期常规治疗无效时应积极抗曲菌治疗,可明显改善症状,降低死亡率,病理活检是确诊的依据.     

High cellular atypia in a pulmonary tumorlet. Report of a case with cytologic findings

BACKGROUND: Pulmonary tumorlets are localized lesions of neuroendocrine cell proliferation, usually found in association with chronic pulmonary inflammation. Since they are mostly incidental histologic or radiologic discoveries, they have received little attention, and there have been no reports on their detailed cytology. We describe for the first time the cytologic features of a pulmonary tumorlet and discuss its differential diagnosis. CASE: An abnormal nodule in the right lung field was discovered on a regular checkup by chest roentgenogram in a 70-year-old, nonsmoking female. Intraoperative aspiration cytology demonstrated cohesive, spindle-shaped cells arranged in fascicles or singly. Since these cells showed nuclear atypia, such as hyperchromasia, a coarsely granular chromatin pattern and nuclear grooving, a nonepithelial malignant lesion was suspected and upper lobectomy performed. The final diagnosis was a pulmonary tumorlet on the basis of histologic examination of the resected material. CONCLUSION: This is the first cytologic report of a pulmonary tumorlet. In this case, differential diagnosis was made of a tumor consisting predominantly of spindle-shaped cells. Although cytologic findings included nuclear atypia, the lesion was not malignant.     

Challenges in Diagnosing Invasive Pulmonary Aspergillosis in Patients With COPD and Other Chronic Lung Disorders

Invasive pulmonary aspergillosis (IPA) is a necrotizing pneumonia caused by airborne opportunistic fungi of Aspergillus species. Patients with chronic obstructive pulmonary disease (COPD) or other chronic lung disorders (CLD) have emerged to be at risk for IPA, with a related mortality rate greater than 70%. Host factors playing a role in the occurrence of IPA in CLD patients differ from those in patients with hematologic disorders and may be largely responsible for a distinct pattern of the disease. Furthermore, these host factors affect the results of standard diagnostic procedures recommended for IPA. Therefore, the diagnosis of IPA in patients with COPD and other CLD remains challenging for physicians. This review puts into perspective the host factors contributing to the pathogenesis of IPA in CLD patients and discusses the use and interpretation of the main diagnostic tools currently available.     

慢性坏死性肺曲霉病18例临床分析

目的 探讨慢性坏死性肺曲霉病（CNPA）的临床特征、诊断与治疗,以提高对该病的认识。方法 对2003年1月-2006年3月广州呼吸疾病研究所经病理证实的18例慢性坏死性肺曲霉病住院病例进行回顾性研究,结合文献分析其临床表现、影像学、诊断和治疗方法。结果CNPA症状无特异性,主要表现为咳嗽、咳痰18例（100.0％）,咯血9例（50.0％）,咳血痰1例（5．6％）,发热7例（38．9％）,气促5例（27．8％）,盗汗3例（16．7％）,胸痛3例（16.7％）,消瘦3例（16．7％）,疲乏无力2例（11.1％）。右肺病变5例（27．8％）,左肺病变11例（61．1％）,双肺受累2例（11.1％）。病灶位于下肺6例（33．3％）,上中肺9例（50.0％）,双肺弥漫病变1例（5．6％）,双肺多发空洞1例（5．6％）,左侧毁损肺1例（5．6％）,不常见的胸膜（腔）受累共4例。“空气新月征”有提示诊断意义,在本组中发现8例（44.4％）。全部病例均经病理确诊,其中经支气管肺组织活检确诊7例。结论CNPA临床表现无特异性,影像学检查在诊断中起重要作用,而诊断需要病理依据。存在肺部基础疾病,出现发热、血痰及空气新月征三联征时,应高度警惕CNPA。系统抗真菌治疗无效、合并大咯血或肺部阴影与肿瘤不能鉴别时,可考虑手术切除。     

The role of muscle flaps in pulmonary aspergillosis

Pulmonary invades the lung parenchyma and vessels, causing necrotizing pneumonia and massive hemoptysis in immunocompromised patients. Medical treatment alone often fails to clear the organism. Early surgical intervention is advocated in localized disease to remove infection near pulmonary vessels. The resection is limited in an attempt to preserve as much lung function as possible. However, preexisting cavitations and lung disease predispose to postoperative space problems, including prolonged air leak, bronchopleural fistula, and empyema. Muscle flaps provide a solution to these problems by obliterating residual space and providing protective coverage to the bronchial stump. The authors present four cases of pulmonary aspergillosis treated by multimodality therapy and extrathoracic muscle flap transposition. Factors that may contribute to successful treatment include underlying condition of the host and history of cancer, radiation therapy, and great vessel involvement. Despite aggressive medical and surgical therapy, pulmonary aspergillosis has a poor prognosis.     

Reversibility of complete unperfusion in a patient with recurrent hemoptysis

We present a case of a 68-year-old woman with a history of mild smoking and chronic bronchitis who showed recurrent hemoptysis. She presented with a nearly normal chest roentgenogram, a non-diagnostic fiberoptic bronchoscopy and a computed tomography and lung scanning both of which were highly suggestive for malignancy. In fact, the former showed obstruction of the main left bronchus, of the superior bronchus for the left upper lobe and of the apical bronchus for the left lower lobe, the latter showed a total cessation of blood flow through the left lung. Pulmonary angiography, however, was normal and aortography showed dilatated and twisted left bronchial arteries. Computed tomography and lung scanning came back to normal after bronchoscopic aspiration of endobronchial clots and a nonspecific antibiotic therapy were carried out. Although very infrequent, bronchial stenosis on CT and complete monolateral unperfusion on lung scintigraphy may occur in patients with hemoptysis of benign origin. We recommend the use of pulmonary arteriography in patients with the above pattern when diagnostic doubt remains after bronchoscopy.     

Erlotinib-Associated Exacerbation of Hypothyroidism with Pericardial Tamponade

ObjectiveTo report a case of erlotinib-associated exacerbation of hypothyroidism complicated by pericardial tamponade.MethodsWe describe the patient’s clinical presentation, biochemical workup, and clinical course.ResultsNon-small cell lung cancer was diagnosed in a 54-year-old woman. After cisplatin and radiation therapy, she was noted to have subclinical hypothyroidism that did not necessitate treatment. The tyrosine kinase inhibitor erlotinib, 150 mg once daily, was prescribed. Three months later, the patient was documented to have severe hypothyroidism. Levothyroxine was prescribed, but she continued to experience shortness of breath, fatigue, and chest and back pain, which resulted in an emergency department visit. Inpatient workup revealed cardiac tamponade with a large pericardial effusion and a right ventricular diastolic collapse. Pericardiocentesis was performed.ConclusionsThis is the first case report linking erlotinib use and thyroid disease. (Endocr Pract. 2012; 18:e111-e113)     

Iatrogenic Cushing Syndrome in Patients Receiving Inhaled Budesonide and Itraconazole or Ritonavir: Two Cases and Literature Review

ObjectiveTo present two cases of iatrogenic Cushing syndrome caused by the interaction of budesonide, an inhaled glucocorticoid, with ritonavir and itraconazole.MethodsWe present the clinical and biochemical data of two patients in whom diagnosis of Cushing syndrome was caused by this interaction. We also reviewed the pertinent literature and management options.ResultsA 71-year-old man was treated with inhaled budesonide for a chronic obstructive pulmonary disease and itraconazole for a pulmonary aspergillosis. The patient rapidly developed a typical Cushing syndrome complicated by bilateral avascular necrosis of the femoral heads. Serum 8:00 AM cortisol concentrations were suppressed at 0.76 and 0.83 µg/dL on two occasions. The patient died 4 days later of a massive myocardial infarction. The second case is a 46-year-old woman who was treated for several years with inhaled budesonide for asthma. She was put on ritonavir, a retroviral protease inhibitor, for the treatment of human immunodeficiency virus (HIV). In the following months, she developed typical signs of Cushing syndrome. Her morning serum cortisol concentration was 1.92 µg/dL. A cosyntropin stimulation test showed values of serum cortisol of <1.10, 2.65, and 5.36 µg/dL at 0, 30, and 60 minutes, respectively, confirming an adrenal insufficiency. Because the patient was unable to stop budesonide, she was advised to reduce the frequency of its administration and eventually taper the dose until cessation.ConclusionClinicians should be aware of the potential occurrence of iatrogenic Cushing syndrome and secondary adrenal insufficiency due to the association of inhaled corticosteroids with itraconazole or ritonavir. (Endocr. Pract. 2013;19:e138-e141)     

Atypical squamous cells as a diagnostic pitfall in pulmonary Wegener's granulomatosis. A case report

BACKGROUND: Wegener's granulomatosis (WG) is characterized by systemic, necrotizing, granulomatous inflammation accompanied by vasculitis. It classically involves the triad of the upper respiratory tract, lungs and kidneys. Isolated pulmonary lesions of WG may present in some patients as pulmonary masses, simulating neoplasms. The features of WG can be suggested by cytologic study. Atypical epithelial cells associated with WG have previously been reported as a cause of a false positive diagnosis of bronchoalveolar carcinoma. CASE: In this case the cytologic findings included atypical squamous cells in a background of acute, chronic and granulomatous inflammation. In several respiratory specimens the atypical squamous cells were incorrectly interpreted as diagnostic of squamous cell carcinoma. The correct diagnosis of WG was confirmed with open lung biopsy, which demonstrated necrotizing granulomatous inflammation with geographic necrosis and associated vasculitis. CONCLUSION: Markedly atypical squamous cells mimicking squamous cell carcinoma can be found accompanying the inflammatory process associated with WG and are a possible diagnostic pitfall. The possibility of WG as well as other inflammatory processes should always be considered in the differential diagnosis of squamous cell carcinoma of the lung. This case is the only reported case of WG in which atypical squamous cells were a diagnostic pitfall, initially suggesting a diagnosis of squamous cell carcinoma.     

Pulmonary sclerosing hemangioma. Report of a case with diagnosis by fine needle aspiration.

Sclerosing hemangioma is a rare but well-recognized benign lesion of the lung. We report a case of pulmonary sclerosing hemangioma correctly diagnosed by fine needle aspiration (FNA) cytology. The sharp and smooth contour of the discrete mass in the left lower zone of the chest roentgenogram raised the possibility of a benign lesion, including pulmonary sclerosing hemangioma. The characteristic "blood spaces" with surrounding regular, bland polygonal tumor cells in the FNA smears provided an essential clue to the diagnosis of sclerosing hemangioma. It was confirmed by Surecut biopsy of the lesion. The patient remained well one year after the investigation and was spared an unnecessary diagnostic thoracotomy. The cytologic features and differential diagnoses of pulmonary sclerosing hemangioma are discussed. Besides delineating the cytologic characteristics of pulmonary sclerosing hemangioma, this case illustrates the importance of a careful clinicopathologic correlation, which should be exercised by the cytopathologist in all instances.     

UNUSUAL CASE OF PULMONARY ARTERIOVENOUS FISTULA CONCEALED BY THE CARDIAC SILHOUETTE ON CHEST ROENTGENOGRAM

A large pulmonary arteriovenous fistula was discovered in a patient with long-standing cyanosis, clubbing and dyspnea, with no other cardiovascular signs or symptoms and a normal chest roentgenogram at the time of cardiac catheterization and pulmonary angiography. The fistula was overshadowed by the cardiac silhouette. Surgical resection was successful. Although rarely undetected on the chest roentgenogram, this potentially lethal malformation should be considered in the differential diagnosis of cyanosis unaccompanied by other cardiovascular signs or symptoms.     

Fatal diphtheria in an older woman

A previously healthy 68-year-old woman presented with fever and sore throat. Her condition was initially diagnosed as necrotizing streptococcal tonsillitis and was treated with penicillin G, given intravenously. A swab of her throat taken for culture at the time of admission yielded Corynebacterium diphtheriae 48 hours later. At that time an electrocardiogram showed new T-wave inversion — evidence of diphtheritic myocarditis. She was immediately given 60 000 units of equine diphtheria antitoxin (following a test dose), but later that day she began choking, became apneic and died. The patient had not received any immunizing agents as a child, and no antitoxin was detected in a blood sample obtained prior to administration of the antitoxin. Her death re-emphasizes the seriousness of diphtheria, an infection to which many elderly people are susceptible.     

Pulmonary adenocarcinoma, Lambert--Eaton myasthenic syndrome, and voltage-gated calcium channels: a case report

ABSTRACT: INTRODUCTION: Lambert--Eaton myasthenic syndrome is a rare disorder and it is known as a paraneoplastic neurological syndrome. Small cell lung cancer often accompanies this syndrome. Lambert--Eaton myasthenic syndrome associated with lung adenocarcinoma is extremely rare; there are only a few reported cases worldwide. CASE PRESENTATION: A 75-year-old Japanese man with a past history of chronic rheumatoid arthritis and Sjogren syndrome was diagnosed with Lambert--Eaton myasthenic syndrome by electromyography and serum anti-P/Q-type voltage-gated calcium channel antibody level preceding the diagnosis of lung cancer. A chest computed tomography to screen for malignant lesions revealed an abnormal shadow in the lung. Although a histopathological examination by bronchoscopic study could not reveal the malignancy, lung cancer was mostly suspected after the results of a chest computed tomography and [18F]-fluorodeoxyglucose positron emission tomography. An intraoperative diagnosis based on the frozen section obtained by tumor biopsy was adenocarcinoma so the patient underwent a lobectomy of the right lower lobe and lymph node dissection with video-assisted thoracoscopic surgery. The permanent pathological examination was the same as the frozen diagnosis (pT2aN1M0: Stage IIa: TNM staging 7th edition). Immunohistochemistry revealed that most of the cancer cells were positive for P/Q-type voltage-gated calcium channel. CONCLUSIONS: Our case is a rare combination of Lambert--Eaton myasthenic syndrome associated with lung adenocarcinoma, rheumatoid arthritis and Sjogren syndrome, and to the best of our knowledge it is the first report that indicates the presence of voltage-gated calcium channel in lung adenocarcinoma by immunostaining.     

Asymptomatic Presentation of Chronic Pulmonary Paracoccidioidomycosis: Case Report and Review

Asymptomatic presentations of chronic pulmonary paracoccidiodomycosis have been reported since 1959, usually published in case series of paracoccidiodomycosis. Incidental radiographic findings on chest roentgenogram led to diagnostic evaluation in these cases. An unusual case in a female patient is described, and 24 previously reported cases are reviewed.     

Spontaneous remission from acute exacerbation of chronic adult T-cell leukemia

Spontaneous remission without any anti-cancer therapy in a 57-year-old woman with adult T-cell leukemia (ATL) is reported. The patient was referred to our department because of persistent cough and appearance of abnormal lymphocytes in the peripheral blood, and she was diagnosed as having chronic ATL. Eight months later, she was re-admitted because of cystitis, watery diarrhea and worsening of respiratory symptoms with an increase of ATL cells (WBC 31 x 10(9)/l with 56% ATL cells). Acute exacerbation of ATL was diagnosed. Interestingly, antibiotic therapy for the pulmonary and urinary tract infections brought about spontaneous reduction of the ATL cell count. Spontaneous remission of ATL continued for one year without chemotherapy. The role of infection as a trigger of acute exacerbation and spontaneous remission of ATL is discussed.     

Primary lymphoma of the heart

The authors describe the case of a 59-year-old woman, treated for breast cancer by mastectomy and chemotherapy 13 years before her death. Eleven years later she was treated successfully by gastric resection and chemotherapy for gastric cancer. In the last five months, the patient presented dyspnoea, leucopenia, hydropericardium and thoracic fluid both sides. In vivo the origin of these symptoms has not been discovered, neither by cytology nor by pleural biopsy.     

Tuberculous thyroiditis and miliary tuberculosis manifested postpartum in a patient with thyroid carcinoma

Right nodular goiter with diffuse miliary shadow on chest roentgenogram was found in a postpartum febrile woman. Transbronchial lung biopsy revealed tuberculous granuloma and acid-fast bacilli were found by aspiration cytology of the thyroid. Although chemotherapy was effective, the thyroid nodule remained palpable and the serum thyroglobulin level remained high. Subtotal thyroidectomy revealed papillary carcinoma associated with tuberculosis and lymph nodes metastasis. This seems to be the first case report of a patient with tuberculous thyroiditis, coexisting with thyroid carcinoma, diagnosed by aspiration cytology and treated prior to surgery.     

Case report of acute necrotizing pancreatitis associated with combination treatment of sitagliptin and exenatide

ObjectiveTo report the first postmarketing case of necrotizing pancreatitis in a patient on combination therapy of sitagliptin and exenatide.MethodsWe describe the patient’s clinical presentation, laboratory test results, imaging, and autopsy findings.ResultsA 76-year-old woman with a history of type 2 diabetes mellitus presented with severe abdominal pain, vomiting, and fever requiring hospital admission. She had been treated with exenatide for 3 years to manage her diabetes mellitus. A few weeks before presentation, sitagliptin was added, presumably to further optimize her glycemic control. Acute pancreatitis was diagnosed during hospital admission. At initial presentation, her serum amylase concentration was 1136 U/L (reference range, 10-130 U/L) and her lipase concentration was greater than 3500 U/L (reference range, 0-75 U/L). In addition, computed tomography of the abdomen and pelvis demonstrated extensive pancreatic parenchymal necrosis. She had undergone previous cholecystectomy, reported no alcohol consumption, and had a normal lipid profile. Although she had a long-standing history of diabetes mellitus, she had no history of pancreatitis or other risk factors that would have caused her to develop the underlying condition. After initial brief improvement, her symptoms worsened, and despite aggressive care, her clinical state deteriorated and she died. Autopsy findings demonstrated acute necrotizing pancreatitis with complete digestion of the pancreas.ConclusionsConsidering the temporal relationship of her symptoms to the addition of sitagliptin to her existing exenatide regimen, this case strongly suggests a possible causal link between exenatide or sitagliptin (or the combination of the 2 drugs) and the etiology of pancreatitis in this patient. (Endocr Pract. 2012;18:e10-e13)