A case of primary Sj?gren's syndrome complicated with lung adenocarcinoma]

A 69-year-old woman presented with headache. Her chest radiograph and computed tomographic scans showed a mass shadow causing superior vena cava syndrome. Bronchofiberscopic examination was nonproductive. The serum value of carcinoembryonic antigen was highly elevated, so we made a presumed diagnosis of primary non-small lung cancer. She also complained of dry eyes and mouth. The elevated values of serum antibodies against SS-A and SS-B and further examinations resulted in a definitive diagnosis of primary Sj?gren's syndrome. Chemotherapy was not effective and she died 14 months later. Autopsy revealed that the mass shadow was a primary lung adenocarcinoma. At the age of 66 she suffered a refractory pneumothorax and her pulmonary cysts or bullae were surgically resected. Those lesions had bullae, emphysema, and alveolar septae thickened by infiltration of lymphoplasmacytic cells. Because she had complained of xerostomia for the last few decades, we associated the cysts with Sj?gren's syndrome. Thoracic CT scans at that time showed a nodule next to a cystic lesion. We raise a possibility that lung cancer might derive from cystic lesions associated with Sj?gren's syndrome.     

Multiple evanescent white dot syndrome following vaccination for COVID-19: A case report

Rationale:Multiple evanescent white dot syndrome (MEWDS) is an acute, usually unilateral, retinal disorder of unknown etiology that predominantly occurs in healthy young women. We report a case of bilateral asymmetric MEWDS that developed following the first vaccination for coronavirus-19 and worsened after a second vaccination.Patient concerns:A 30-year-old Japanese woman was examined in an eye clinic for blurred vision in her left eye for 1 week duration. Thirteen days before her examination, she had received her first BNT162b2 mRNA SARS-CoV-2 vaccination. Her best-corrected visual acuity was 20/20 in both eyes. Fundus examination revealed multiple yellowish-white spots in the perifoveal area of both eyes. Visibility of the spots gradually decreased during the following week. She was then vaccinated with a second dose, and 3 days later, her vision worsened in her left eye. She was then referred to our hospital because of worsened vision and the appearance of white spots on other parts of the retina. Ophthalmological examination revealed a best-corrected visual acuity of 30/20 both eyes.Diagnosis:The flare value in the anterior chamber was elevated in both the eyes. Fundus examination showed multiple white spots in the perifoveal area of both eyes, but they were more prominent in the left eye. Fundus fluorescein angiography revealed early hyperfluorescent spots located circumferentially around the fovea in both eyes. We concluded that the patient had MEWDS, which was most likely due to mRNA COVID-19 immunization.Interventions:The patient was treated with topical betamethasone sodium phosphate/fradiomycin sulfate 0.1% thrice daily for 2 months.Outcomes:Two months after treatment, her blurry vision resolved with the disappearance of the fundus lesions.Lesson:Clinicians should be aware of potential adverse ocular events following similar vaccinations.     

A case of small-cell lung cancer during treatment for active pulmonary tuberculosis after surgery for non-small-cell lung cancer]

A 76-year-old man taking anti-tuberculous drugs for active pulmonary tuberculosis was admitted to our hospital requiring further examination of a pulmonary mass in the left upper lobe. He had had right lower lobectomy and partial middle lobectomy for non-small-cell lung cancer two years before admission. Retrospectively, the left upper lobe mass had been gradually growing for about 2 months. We performed a transbronchial lung biopsy of the pulmonary mass, and diagnosed small-cell lung cancer. This report describes a rare case of small-cell lung cancer appearing during treatment for active pulmonary tuberculosis after non-small-cell lung cancer surgery.     

Video assisted thoracic surgery for Mycobacterium avium complex pulmonary disease spread over multiple pulmonary lobe within one lung]

A 75-year-old woman with Mycobacterium avium complex (MAC) pulmonary disease who had been treated by drug therapy for 7 years was admitted on an emergency basis for hemoptysis. Through the previous 7 years, her sputum cultures had been positive for MAC, and her clinical symptoms and examinations, such as chest X-ray and computed tomography, revealed that her condition had worsened. The lesions spread over the right middle lobe, segment 3 (S3) in the right upper lobe, and segment 6 (S6) in the right lower lobe, however, no lesions were present in the left lung. Since we believed that the disease was localized unilaterally, surgical treatment was selected. The surgical procedures that were employed included right middle lobectomy, right S3 segmentectomy and partial resection of right S6 by thoracoscopy. Sixteen months postoperatively, the sputum culture is negative for MAC. Our procedure enabled the preservation of effective lung function and was successful in controlling MAC pulmonary disease. It is believed that surgery for MAC pulmonary disease should be evaluated in more patients to decide the appropriate surgical indication and procedure.     

The challenging management of lung choriocarcinoma

The purpose of this paper is to highlight the existence and the management of lung choriocarcinoma (CCA), a rare category of lung tumors. We present a 42-year-old female that presented to our department with a PET positive lesion in the left upper lobe and a history of pregnancy 6 months prior to onset of symptoms. CT guided biopsy was inconclusive for diagnosis and the patient underwent a left thoracotomy and lingula sparing upper lobectomy. Histology revealed CCA of the lung and subsequently blood results confirmed the elevated b-HCG. CCA of the lung is a clinical entity that should be considered in the differential diagnosis of lung lesions in women after pregnancy.     

Bronchioloalveolar carcinoma complicated by a lesion resembling bronchiolitis obliterans organizing pneumonia in the opposite lung]

A 50-year-old woman with abnormal shadows in her chest radiograph was admitted for a more detailed examination in April 2000. A chest CT scan showed infiltrates surrounded by ground-glass opacities in the right upper lobe, and ground-glass opacities with partial infiltrative changes in the left lower lobe. A diagnosis of adenocarcinoma like bronchioloalveolar carcinoma (BAC) was made for the lesion in the right upper lobe by means of a transbronchial biopsy. However, the specimen from the left lower lobe showed nonspecific changes, and bronchiolitis obliterans organizing pneumonia (BOOP) or a metastatic lesion from the BAC was suspected. We prescribed steroids for the short term. Since the lesion in the left lobe disappeared, we performed a right upper lobectomy. In cases of primary lung carcinoma, BOOP in the opposite lung is rare and it was difficult to decide on the operability. We concluded that steroid therapy is useful for distinguishing between inflammatory and tumorous lesions in cases in which a detailed examination is difficult.     

Presumed primary malignant melanoma of the liver: a case report

A 55-year-old woman was hospitalized with a palpable and painful mass in her upper abdomen. Abdominal computed tomography revealed a 10 × 7-cm tumor extending into the abdominal cavity from the left hepatic lobe and multiple metastatic lesions in the right hepatic lobe. A left hepatic lobectomy was performed for debulking and palliative resection. Histopathological examination of the resected specimen diagnosed the large hepatic tumor in the left hepatic lobe as a malignant melanoma. Physical and radiological examinations were performed on dermatological, ophthalmic, gynecological, and central nervous system areas, and endoscopic examinations were performed on the upper digestive tract and colon. No other lesions were disclosed as possible primary tumors for the disease. This result suggested that the tumor might arise from the left hepatic lobe.     

A case of endobronchial actionomycosis evaluated by FDG-PET

In April 2005, a 78-year-old man was admitted to our hospital because an abnormal chest shadow had been noted on a medical examination. Our investigation revealed primary squamous cell lung carcinoma in the right lower lobe (stage IIIA). Right middle and lower lobectomies including resection of the chest wall were performed. 18Fluorodeoxyglucose-positron emission tomography (FDG-PET) conducted 18 months later demonstrated nodular lesions with intense FDG activity in the right upper lobe and the presence of a post-resection positive bronchial stump. Fiberoptic bronchoscopic examination revealed a pus-coated mass located in the endobronchial lesion, and Actinomyces was identified in a biopsy specimen of the mass. Endobronchial actinomycosis was diagnosed. An FDG-PET examination conducted after the patient was treated with amoxicillin (AMPC) for 2 months, did not indicate any FDG activity in the endobronchial lesion.     

A case of psittacosis with wandering infiltrates developing to acute respiratory distress syndrome]

A 52-year-old woman visited a local hospital with a high fever, non-productive cough and general fatigue. Her chest X-ray showed infiltrate in the right middle lung field. Computed tomography scans revealed ground-glass opacity and surrounding ring-shaped air-space consolidation, the "reversed halo sign". Cefpirom was administered, but her symptoms persisted and the infiltrate migrated to the left upper lobe. As cryptogenic organizing pneumonia was suspected, she was then treated with intravenously pulsed methylprednisolone followed by prednisolone. Despite these therapies, acute respiratory failure occurred and she was therefore transferred to our hospital. On admission, severe hypoxemia and diffuse bilateral infiltrates on chest images suggested acute respiratory distress syndrome. As we obtained information that a parakeet had recently died at her home, minocycline was administered, resulting in prompt improvement of the symptoms, respiratory insufficiency and pulmonary infiltrates. Finally, elevated antibody titers against Chlamydophila psittasi confirmed a diagnosis of Psittacosis. Sequential chest computed tomography scans in this case indicate that absorption of marginal air-space consolidation with extended central ground glass attenuation in concordance with a new infiltrate on another lung field appeared to create wandering infiltrate. Wandering infiltrate on chest X-ray in psittacosis may be a sign of disease progression.     

A case of multidrug-resistant (MDR) tuberculosis with collapse of the left lung after hemoptysis

An 82-year-old female was admitted to our hospital with multidrug-resistant (MDR) tuberculosis, defined as resistance to both isoniazid and rifampicin. Chest X-ray showed massive infiltrates with a large cavitary lesions in the left lung field. No antituberculous agents were useful in improving her clinical condition and at 6th months after admission, she exhibited sudden onset of massive hemoptysis, which was successfully treated by bronchial artery embolization. After hemoptysis, her chest X-ray showed collapse of the left lung and computed tomography showed a coagula-like shadow in the left main bronchus, and sputum examination revealed no Mycobacterium tuberculosis colonies. The patient was discharged 5 months after the onset of hemoptysis.     

Tongue metastasis as an initial presentation of a lung cancer

Metastasis to the tongue seldom occurs, and lingual metastasis as an initial sign of cancer occurs even less frequently. We report a case of lung cancer in which the patient's initial symptom was related to the tongue metastasis. A 63-year-old man had a submucosal tumor on the left posterolateral aspect of the tongue and a biopsy specimen of the tongue tumor showed poorly differentiated squamous cell carcinoma. A chest X-ray showed a mass in the right lung and cytological examination of the specimen obtained by bronchial brushing showed poorly differentiated squamous cell carcinoma, whose appearance was similar to that of the tongue. Based on these findings, the tongue lesion was diagnosed a metastatic tumor from the lung cancer. The patient received radiation therapy combined with systemic chemotherapy, however, he died 5 months after the diagnosis of lung cancer. An autopsy revealed a lung cancer in the right lower lobe with metastatic tumors in the tongue, right middle lobe, left upper lobe, liver, adrenal gland, pericardium, heart, and subcutaneous tissues. No other possible primary cancer that may have been the cause of the metastases was identified.     

A case of invasive thymoma displaying endobronchial polypoid growth

A 58-year-old woman was admitted due to an abnormal shadow on chest X-ray film and two episodes of hemoptysis during the past two years. Chest radiography showed a mass with calcification in the left upper lobe of the lung. A white polypoid lesion in left B3b + c bronchus was discovered by fiberoptic bronchoscopy. Biopsy specimen of the polypoid lesion demonstrated fungi like aspergilli in the necrotic tissue. We suspected pulmonary aspergillosis combined with old tuberculosis and treated with antifungal and antituberculous drugs. After nine months, however, we recognized that the mass had become larger radiologically. A polypoid tumor with a brown, glossy surface in the left B3 bronchus was again noted endoscopically. Histologic findings of the biopsy specimen obtained from this tumor were suggestive of thymoma. Thymectomy and left upper lobectomy were performed. The cross-section of the surgical specimen revealed that the tumor extended not only in the mediastinum but also in the pulmonary parenchyma with polypoid growth into the lumina of bronchi. Microscopically, the tumor was an invasive thymoma composed of both epithelial and lymphocytic elements.     

Patient with advanced intrahepatic cholangiocarcinoma with long-term survival successfully treated with a combination of surgery and chemotherapy

Intrahepatic cholangiocarcinoma (ICC) is a relatively rare malignancy arising from the biliary epithelium. Prognosis is typically poor. Currently, aggressive surgical resection is the only treatment modality that offers patients any chance of long-term survival. Here, we present the case of a 57-year-old woman in whom we diagnosed ICC, with the tumor occupying the entire left and caudate hepatic lobes and daughter nodules in the right lobe. She underwent hepatectomy of segments I to VI, combined with intraoperative microwave coagulation therapy for nodules in the residual liver. Three months after the surgical resection, she had recurrence of the disease. The patient subsequently received weekly intraarterial chemotherapy with irinotecan (CPT-11), and a partial response was observed which persisted for 18 months. Subsequent computed tomography revealed the regrowth of three tumors, and she therefore underwent a repeat resection 24 months after the first surgical operation. In postoperative-month (POM) 32, she received systemic chemotherapy with tegatur/gimeracil/oteracil potassium (S-1)/cisplatin for multiple small nodules in her lung. Following three cycles of chemotherapy with a stable disease response, partial resections of the lung were performed. Third and fourth hepatectomies were performed in POMs 46 and 59, respectively. Five years and 5 months after the first hepatectomy, she is alive with small lesions in her lung. This multimodal approach may be effective for ICC.     

Malignant Perivascular Epithelioid Cell Neoplasm of the Mediastinum and the Lung: One Case Report

A perivascular epithelioid cell neoplasm (PEComa) in the chest is rare, let alone in the mediastinum and lung.A 63-year-old man was admitted to our hospital with chest pain for more than 2 months and was found to have an opacity in his mediastinum and lung for 3 weeks. Enhanced chest computed tomography (CT) revealed a mass in both the left upper lobe and central anterior mediastinum. To identify the disease, a CT-guided percutaneous transthoracic needle biopsy of the upper left lung lesions was performed. The pathology result was consistent with epithelioid angiomyolipoma/PEComa. After a standard preparation for surgery, the neoplasms in the mediastinum and left lung were resected. The operative findings revealed extensive mediastinal tumor invasion in parts adjacent to the pericardium, including the mediastinal pleura, left pulmonary artery and vein, and phrenic nerve. The left lung tumor had invaded the lung membranes. The final pathologic diagnosis was malignant epithelioid angioleiomyoma in the left upper lung and mediastinum. Later, the mediastinal tumor recurred. The radiography of this case resembles left upper lobe lung cancer with mediastinal lymph node metastasis. Because this tumor lacks fat, the enhanced CT indicated that it was malignant but failed to identify it as a perivascular epithelioid cell neoplasm.This case reminds clinicians that, although most PEComa are benign, some can be malignant. As the radiology indicated, chest PEComas lack fat, which makes their preoperative diagnosis difficult. Therefore, needle biopsy is valuable for a definitive diagnosis.     

A case of Sj?gren's syndrome with dermatomyositis who died of rapidly progressive interstitial pneumonia]

We report a case of 55 year-old woman with six year history of Sj?gren's syndrome developed fatal rapidly progressive interstitial pneumonia. She had been well until February 1999. She developed swelling and erythematous lesions in the cheek and hands in spring 1999. She was admitted to our hospital for investigations of skin lesions in May 1999. Physical examination on admission revealed small hemorrhagic lesions in the nailfold. Serum CK level was slightly elevated. Electromyogram and MRI suggested mild myositis in the proximal upper extremities. She was suspected to have dermatomyositis along with Sj?gren's syndrome. Prednisolone 10 mg/day had been given for her skin problems since March 1999. Suddenly, dyspnea on exertion was appeared on 34th day of admission. Chest X-ray film showed an acute worsening of interstitial pneumonia. Methylprednisolone pulse therapy (1000 mg for 3 days) and cyclophosphamide pulse therapy (500 mg for a day) were started, and she was subsequently treated with 60 mg/day of prednisolone and 250 mg/day of Cyclosporin A. However, interstitial pneumonia did not respond to the treatment, and pneumomediastinum and pneumothorax have developed. She died of respiratory failure on 55th day. We consider that most likely explanation for fatal interstitial pneumonia is concomitantly occurred dermatomyositis.     

Familial recurring cardiac myxoma

A 34-year-old woman presented with her fourth occurrence of an atrial myxoma and a right lower lung field mass. Her surgical history was extensive. In addition to resection of three previous atrial myxomas, she had undergone resection of her adrenal glands as a teenager because of Cushing's syndrome, and a hysterectomy at age 26 revealed a myxoid leiomyoma. Family history was remarkable as a maternal uncle and daughter died from embolic complications of left atrial myxomas and her sister previously had a left atrial myxoma resected. The patient underwent uncomplicated removal of the myxoma, and resection of the lung mass revealed a granuloma. A review of typical and atypical aspects of cardiac myxomas is provided including a rare and recently described syndrome of familial cardiac myxoma associated with Cushing's syndrome, spotty skin pigmentation and other myxoid tumors.     

Eosinophilic myocarditis due to Churg–Strauss syndrome mimicking reversible dilated cardiomyopathy

A 41-year-old woman with a history of asthma arrived at the emergency room of our hospital with dyspnea. The electrocardiogram showed no specific results. Echocardiography defects revealed an obvious decrease in the left ventricular systolic function and enlargement of the left chamber. We initially considered her condition to be dilated cardiomyopathy. However, she had eosinophilia in the peripheral blood and elevated cardiac enzymes. The coronary angiography showed normal coronary arteries. Single photon emission computed tomography (SPECT) showed infiltrative myocardial disease. She was then diagnosed with eosinophil infiltrations. Combined with peripheral nerve injury and lung involvement, she was diagnosed as having Churg–Strauss syndrome. After initiating prednisone treatment, her eosinophilia and rising cardiac enzymes recovered to normal, and both her echocardiographic abnormalities and symptoms noticeably improved.     

Antiphospholipid syndrome with cortical blindness resulting from infarction around the posterior cerebral artery in an elderly woman

An 87-year-old woman with antiphospholipid syndrome accompanied by cortical blindness and thalamic syndrome resulting from infarction of the posterior cerebral artery is reported. She was hospitalized because of laceration of the head. Two months later, she complained of loss of visual acuity, sharp pain and numbness involving the left half of the body except her face. New right posterior lobe infarction and the existence of old left infarctions were confirmed by serial CT scans. Helical CT scan revealed embolization of the posterior cerebral artery with atherosclerotic stenosis. Serological examination showed biologically false-positive and positive findings for lupus anticoagulant. She was treated with warfarin potassium and clonazepam.     

A case of adenocarcinoma of the lung presenting symptoms of choroidal metastasis as the initial clinical manifestation]

A 51-year-old woman was referred to our hospital with a complaint of disturbance in vision. Ophthalmologic examination revealed multiple choroidal tumors. High-resolution CT showed a nodular shadow in the left lower lobe. Transbronchial biopsy and right supraclavicular lymph node biopsy specimens showed a poorly-differentiated adenocarcinoma. We concluded that the choroidal tumors had metastasized from the lung. Combined chemotherapy (CDDP + CPT-11) followed by irradiation of both eyes and brain were performed. Nevertheless, she died 6 months after the initial presentation. It is important to notice ophthalmologic symptoms because lung cancer may metastasize to the choroids.     

Intramedullary spinal cord metastasis of lung adenocarcinoma causing Brown-Séquard Syndrome]

A 36-year-old woman admitted to our hospital because of numbness in the left limbs and weakness in the right arm, and was subsequently given a diagnosis of intramedullary spinal cord metastases from lung cancer. The patient had lung adenocarcinoma with metastases to the brain, spine and lymph nodes. Occipital craniotomy, radiation therapy and chemotherapy were performed on the lesions in the year following June 1994. In June 1995, however, she complained of numbness in the left limbs and weakness in the right arm. Compatible with her neurological manifestation, MRI demonstrated tumors in the right side of the cord at the spinal level of C3-4 and C7-Th1, both of which were of high density in T2-enhanced conditions with enhancement by gadolinium-diethylenetriamine pentaacetic acid. No invasion from spinal metastasis was detected by CT, scintigraphy or MRI. We therefore diagnosed her manifestation as Brown-Séquard syndrome caused by intramedullary spinal cord metastatic tumors of lung adenocarcinoma. In order to avoid paraplegia and dysfunction of the bladder and bowel, radiation therapy of the cord lesions with total dose of 44 Gy was performed. Her neurologic manifestation was improved, restoring her quality of life, as the tumor size estimated by MRI decreased. Four months later, however, she died of lung adenocarcinoma that developed accompanied with severe peritonitis carcinomatosa and multiple metastases.