scholarly journals Challenges in multidisciplinary medical rehabilitation - Swyer-James-MacLeod Syndrome: case presentation and short literature review

2019 ◽  
Vol 10 (Vol 10 No. 4) ◽  
pp. 457-461
Author(s):  
Adelina ANTON ◽  
Ioana Sinziana GHERGHISAN ◽  
Angela OMER (Gheorghe) ◽  
Elena Valentina IONESCU ◽  
Madalina Gabriela ILIESCU ◽  
...  

Introduction. Swyer-James-MacLeod syndrome (SJMS) or unilateral hyperlucent lung syndrome is a rare disorder caused by infectious bronchiolitis obliterans and pneumonitis occurring in childhood. It is characterized by hypoplasia and/or agenesis of the pulmonary arteries resulting in pulmonary parenchyma hypoperfusion. Materials and methods. We report the case of a 27 years-old female patient who presented with progressive dyspnea, productive cough, fever and chills. Results and discussion. Chest radiography showed unilateral loss of left lung volume with hyperlucency. Unilateral reduction in vascularity with reduced caliber of the left pulmonary artery was revealed on CT scan of the chest, final diagnosis of SJMS being confirmed by angiography. Conclusions. This case strongly supports the recommendation of considering SJMS within the differential diagnosis workup of bronchiectasis, the syndrome being usually underdiagnosed.

1979 ◽  
Vol 46 (4) ◽  
pp. 625-631 ◽  
Author(s):  
W. O. Kan ◽  
J. R. Ledsome ◽  
C. P. Bolter

In chloralose-anesthetized dogs a constant-flow, right atrium-to-left pulmonary artery, right-heart bypass was created. The right lung root was completely occluded and an isolated pouch of the main pulmonary arteries was perfused with venous blood at controlled nonpulsatile pressures between 10 and 90 Torr. An increase in pulmonary arterial pouch pressure caused an increase in systemic arterial pressure and in respiratory drive. In animals with afferent nerves intact, the increase in respiratory drive was indicated by an increase in the amplitude of the integrated phrenic electroneurogram. In animals with the left vagus nerve cut immediately above the left lung root, the increase in respiratory drive was evident by a shortening of the time of expiration. All responses were abolished by bilateral section of the cervical vagosympathetic trunks. It is concluded that in the preparation described pulmonary arterial distension causes a reflex increase in systemic vascular resistance and in respiratory drive, the afferent path of the reflex being in the vagus nerves.


2021 ◽  
Vol 23 (09) ◽  
pp. 614-624
Author(s):  
Dr. Praveen Sharma K MDRD ◽  
◽  
Dr. Samaran M ◽  
Dr. Keerthivatsan Mani ◽  
Dr. Ashwini Govisetty ◽  
...  

SWYER-JAMES-MACLEOD SYNDROME (SJMS) is a rare pulmonary disorder that manifests as unilateral hyperlucent lung. HRCT is a novel imaging modality to diagnose SJMS since Chest radiographs underestimate this condition. We report a case presentation of SJMS in a 54-year-old male and treated with bronchodilators and i.v antibiotics. In conclusion, only a few with SJMS in adulthood has been reported worldwide.


1976 ◽  
Vol 40 (6) ◽  
pp. 883-888 ◽  
Author(s):  
J. C. Newell ◽  
M. G. Levitzky ◽  
J. A. Krasney ◽  
R. E. Dutton

In 16 dogs ventilated with 100% O2, control blood flow to the left lung was 35 +/- 2% of aortic flow. When left lung atelectasis was induced, left pulmonary artery flow fell to 19 +/- 2% of aortic flow. A large retrograde component of flow developed in this pulmonary artery, suggesting that blood flows into the pulmonary arteries of both lungs during systole, but flows back out of the collapsed lung and into the uncollapsed lung during diastole. Systemic PaO2 remained above 78 mmHg. Subsequently, when the ventilation of the right lung was changed from oxygen to room air, systemic PaO2 fell to 64 +/- 3 mmHg and atelectatic left lung flow rose from 19 +/- 2% to 28 +/- 2% f aortic flow. This was associated with a reduction in reflux from the atelectatic lung. These results suggest that the attenuation of flow to an atelectatic lung is more pronounced if systemic normoxemia is maintained by adequate oxygenation of the normal lung.


2016 ◽  
Vol 19 (4) ◽  
pp. 187 ◽  
Author(s):  
Dohun Kim ◽  
Si-Wook Kim ◽  
Hong-Ju Shin ◽  
Jong-Myeon Hong ◽  
Ji Hyuk Lee ◽  
...  

A 10-day-old boy was transferred to our hospital due to tachypnea. Patent ductus arteriosus (PDA), 4.8 mm in diameter, with small ASD was diagnosed on echocardiography. Surgical ligation of the ductus was performed after failure of three cycles of ibuprofen. However, the ductus remained open on routine postoperative echocardiography on the second postoperative day, and chest CT revealed inadvertent ligation of the left pulmonary artery (LPA) rather than the PDA. Emergent operation successfully reopened the clipped LPA and ligated the ductus on the same (second postoperative) day.<br />Mechanical ventilator support was weaned on postoperative day 21, and the baby was discharged on postoperative day 47 with a normal left lung shadow.


2021 ◽  
Vol 14 (1) ◽  
Author(s):  
Kimihiro Nishino ◽  
Eiko Yamamoto ◽  
Yoshiki Ikeda ◽  
Kaoru Niimi ◽  
Toshimichi Yamamoto ◽  
...  

Abstract Background Pure ovarian choriocarcinoma can be gestational or nongestational in origin. Nongestational pure ovarian choriocarcinoma is extremely rare and the prognosis is thought to be worse than that of the gestational type in patients with metastatic disease. We present a case of metastatic pure ovarian choriocarcinoma with poor prognosis in which the origin was identified as nongestational by DNA short tandem repeat (STR) analysis. Case presentation A nulliparous woman in her thirties with metastatic choriocarcinoma was referred to our hospital after initial treatment proved unsuccessful. Two months earlier, she had undergone brain tumor resection and histological examination confirmed choriocarcinoma. Serum human chorionic gonadotropin (hCG) concentration at initial diagnosis was 5030 IU/L. Two cycles of a combination chemotherapy regimen of methotrexate, etoposide, and actinomycin-D (MEA therapy), which is commonly used for gestational choriocarcinoma, was administered. However, the disease could not be controlled. Imaging modalities at presentation revealed tumor present in the left ovary and left lung, but not in the uterus, which led us think that the choriocarcinoma was nongestational. Bleomycin, etoposide, and cisplatin (BEP therapy) which is commonly used for nongestational choriocarcinoma (malignant germ cell tumor) and surgical resection of the uterus, bilateral ovaries, and an affected part of the left lung led to the nadir level of hCG, but the tumor relapsed and levels of hCG again increased. To investigate the origin of choriocarcinoma, we performed DNA STR analysis of tumor cells and oral mucosal cells. Analysis revealed the origin of the choriocarcinoma as nongestational, as the genotype of tumor cells entirely corresponded with that of oral mucosal cells. BEP therapy and chemotherapy regimens administered for nongestational choriocarcinoma and gestational choriocarcinoma proved ineffective, and the patient died 21 months after diagnosis of metastatic choriocarcinoma. Conclusion Metastaic nongestational pure choriocarcinoma of ovary is an extremely rare and an aggressive disease, frequently resulting in poor outcome.


2021 ◽  
Vol 7 (1) ◽  
Author(s):  
Kozue Matsuishi ◽  
Kojiro Eto ◽  
Atsushi Morito ◽  
Hirokazu Hamasaki ◽  
Keisuke Morita ◽  
...  

Abstract Background Solitary fibrous tumor (SFT) is a relatively rare mesenchymal tumor that mainly affects adults. Its prognosis is good after curative resection, but distant recurrences after 10 years or longer have been reported. Recurrent SFT usually arises as a local lesion; distant metastasis is rarely reported. Here, we report lung metastases that recurred a decade after excising a retroperitoneal primary SFT. Case presentation A 44-year-old woman had an SFT resected from her right retroperitoneum at our hospital. Ten years later, at age 54, she underwent a lung resection after CT showed three suspected metastases in her left lung. All three were histologically diagnosed as lung metastases from the retroperitoneal SFT. However, whereas the primary SFT had 1–2 mitotic cells/10 high power fields (HPF), the metastatic lesion increased malignancy, at 50/10 HPF. Conclusion Patients who have had resected SFTs should be carefully followed up, as malignancy may change in distant metastasis, as in this case.


2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Hyun-Hwa Cha ◽  
Hae Min Kim ◽  
Won Joon Seong

Abstract Background Unilateral pulmonary artery discontinuity is a rare malformation that is associated with other intracardiac abnormalities. Cases accompanied by other cardiac abnormalities are often missed on prenatal echocardiography. The prenatal diagnosis of isolated unilateral pulmonary artery discontinuity can also be delayed. However, undiagnosed this malformation would have an effect on further prognosis. We report our case of a prenatal diagnosis of pulmonary atresia with ventricular septal defect and left pulmonary artery discontinuity. Case presentation A 33-year-old Asian woman visited our institution at 24 weeks of gestation because of suspected fetal congenital heart disease. Fetal echocardiography revealed a small atretic main pulmonary artery giving rise to the right pulmonary artery without bifurcation and the left pulmonary artery arising from the ductus arteriosus originating from the left subclavian artery. The neonate was delivered by cesarean section at 376/7 weeks of gestation. Postnatal echocardiography and multidetector computed tomography showed a right aortic arch, with the small right pulmonary artery originating from the atretic main pulmonary artery and the left pulmonary artery originating from the left subclavian artery. Patency of the ductus arteriosus from the left subclavian artery was maintained with prostaglandin E1. Right ventricular outflow tract reconstruction and pulmonary angioplasty with Gore-Tex graft patch was performed 25th day after birth. Unfortunately, the neonate died because of right heart failure 8 days postoperation. Conclusion There is a possibility that both pulmonary arteries do not arise from the same great artery (main pulmonary artery or common arterial trunk). Therefore, clinicians should check the origin of both pulmonary arteries.


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Tomoko Suzuki ◽  
Miwako Saitou ◽  
Yuriko Igarashi ◽  
Satoshi Mitarai ◽  
Katsunao Niitsuma

Abstract Background Mycobacterium (M) talmoniae isolated from a patient with cystic fibrosis was first described in 2017, and cases of M. talmoniae remain exceedingly rare. Case presentation A 51-year-old woman had respiratory symptoms for 10 years. Diffuse panbronchiolitis (DPB) was detected at the first visit at our hospital. A cavity lesion in the apex of the left lung was found, and sputum and bronchoalveolar lavage fluid were acid-fast bacillus (AFB) smear- and culture-positive besides Pseudomonas aeruginosa. M. talmoniae was finally identified, and the standard combination therapy for non-tuberculous mycobacteria (NTM) was administered for 2 y referring to the drug-susceptibility test. Thereafter, the AFB culture was negative, the wall thickness of the lung cavity was ameliorated, and oxygen saturation improved. Conclusions We encountered a rare case of M. talmoniae with DPB, for which standard combination therapy was effective. M. talmoniae may be considered a potential pathogen of lung disease, especially in patients with bronchiectatic lesions.


2014 ◽  
Vol 117 (5) ◽  
pp. 535-543 ◽  
Author(s):  
Justin A. R. Lang ◽  
James T. Pearson ◽  
Arjan B. te Pas ◽  
Megan J. Wallace ◽  
Melissa L. Siew ◽  
...  

At birth, the transition to newborn life is triggered by lung aeration, which stimulates a large increase in pulmonary blood flow (PBF). Current theories predict that the increase in PBF is spatially related to ventilated lung regions as they aerate after birth. Using simultaneous phase-contrast X-ray imaging and angiography we investigated the spatial relationships between lung aeration and the increase in PBF after birth. Six near-term (30-day gestation) rabbits were delivered by caesarean section, intubated and an intravenous catheter inserted, before they were positioned for X-ray imaging. During imaging, iodine was injected before ventilation onset, after ventilation of the right lung only, and after ventilation of both lungs. Unilateral ventilation increased iodine levels entering both left and right pulmonary arteries (PAs) and significantly increased heart rate, iodine ejection per beat, diameters of both left and right PAs, and number of visible vessels in both lungs. Within the 6th intercostal space, the mean gray level (relative measure of iodine level) increased from 68.3 ± 11.6 and 70.3 ± 7.5%·s to 136.3 ± 22.6 and 136.3 ± 23.7%·s in the left and right PAs, respectively. No differences were observed between vessels in the left and right lungs, despite the left lung not initially being ventilated. The increase in PBF at birth is not spatially related to lung aeration allowing a large ventilation/perfusion mismatch, or pulmonary shunting, to occur in the partially aerated lung at birth.


Sign in / Sign up

Export Citation Format

Share Document