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We report on the case of a 3-year-old child presenting bilateral bronchiectasis due to recurrent pneumonia with esophageal achalasia. The final diagnosis was triple A syndrome. This presentation is particularly atypical and rare at this age.

A 65-year-old man with a lifelong history of paranoid schizophrenia had been treated with 50 mg of risperidone microspheres every 2 weeks for the past 10 years and had tolerated this therapy well. Approximately 1 year ago some drooling and a modest resting tremor developed, but there were no other

Tracheo-esophageal fistula (TEF) without associated esophageal atresia or H-type fistula is a rare congenital anomaly. H-type fistula is usually missed in the neonatal period as the presenting symptoms are either of recurrent pneumonia or gastro-esophageal reflux which always lead to delay in diagnosis and infant undergoes unnecessary treatment. We report a case of H-type of TEF, diagnosed within 12 days of birth based upon choking and cyanosis on the first trial of spoon feeds. Diagnosis was confirmed with contrast esophagogram. The infant was operated for it and was successfully discharged.

A 35-year-old lady was seen in the outpatient clinic owing to fever, cough with mucopurulent expectoration, and breathlessness for the duration of 1 month. She had history of similar episodes treated with antibiotics four times during last 2 years. There was no history of recurrent sinusitis, diarrhea, and skin or soft tissue infection. She had no history of diabetes mellitus or steroid intake. She denied any history of facial trauma or dental infection in the past. There was no history of tuberculosis in her or in the family. Radiograph and CT scan of the chest revealed right upper lobe consolidation. Flexible fibreoptic bronchoscopy revealed multiple nodules at opening of right upper lobe bronchus. This clinicopathological conference describes the details of differential diagnoses, difficulties in achieving the final diagnosis and management of such patient.

A 56-year-old man with a history of total gastrectomy for gastric cancer at age 44 visited the internal medicine department of our hospital because of a slight fever (> 37℃ ), chills and cough since the previous night. His white blood cell count was 44,200 cells/μl and C-reactive protein level was 8.4 mg/dl. Plain chest radiography showed opacity in the right upper lung field. Based on these findings, he was diagnosed with pneumonia and hospitalized. He responded to intravenous ceftriaxone sodium hydrate (2 g/day) for 8 consecutive days, and was discharged on hospital day 9. Coughing started again 2 weeks after discharge and he visited our department again 10 days after noticing the cough. Plain chest radiography showed opacity in the right lower lung field and he was diagnosed and hospitalized for the second time with pneumonia 48 days after discharge. He was discharged on hospital day 9 but visited our department again with a complaint of nighttime heartburn 9 days after the second discharge. Bile reflux after total gastrectomy was suspected as the cause of the recurrent pneumonia and oral administration of camostat mesilate (300 mg/day) was started. Although this treatment alleviated the condition, his adherence to the regimen gradually declined 9 months after the second discharge. He visited our department for a third time complaining of fever and coughing 15 months after the second discharge. Pneumonia in the left middle lung field was treated by a 7-day oral course of levofloxacin hydrate (500 mg/day). The importance of oral camostat mesilate was stressed to the patient and instructions on lifestyle changes were given to prevent reflux esophagitis. The patient has not had heartburn or cough since, and recurrence of pneumonia appears to have been prevented. This case suggests that silent aspiration due to esophageal reflux and aspiration pneumonia should be suspected in cases of recurrent fever without cough or sputum after total gastrectomy.

Morgagni hernia is the rarest form of congenital diaphragmatic hernia and is commonly found either in the first few hours of life or in antenatal period. It is less common in adult and is mostly diagnosed incidentally in an asymptomatic patient. Symptomatic adult cases are even rarer with a wide variety of symptoms. Here we report a patient with a one year history of chronic recurrent cough and dyspnea, who had been misdiagnosed as recurrent pneumonia before being recognized and treated as Morgagni hernia.

Background and aims Pneumonia is the most important cause of death in children in many countries. The use of inappropriate antibiotics in the treatment of pneumonia is associated with multi drug resistant microorganisms that lead to increased morbidity and mortality. Therefore, the causative microorganism should be detected for avoidence of unnecessary antibiotic usage. Underlying diseases are predisposing factors for recurrent pneumonia. The choice of pneumonia treatment should be based on causative microorganisms and underlying diseases. In this study we aimed to evaluate the frequency of recurrent pneumonia and underlying disease in hospitalised children, to identify the distribution of causative microorganisms and to compare our data with previous studies. Methods Between January 2003 and December 2015 children hospitalised due to pneumonia were retrospectively owerviewed. The patients who met criteria for recurrent pneumonia were included in this study. Demographic, clinical, laboratory and imaging results were collacted from the data system. Results During 13 years, 129 (9.2%) hospitalised children met criteria of recurrent pneumonia. Underlying diseases were detected in 95 (73.6%) patients. Rhinovirus, adenovirus and respiratory synstial virus were the most frequent causative agents. Conclusion The majority of patients who were hospitalised due to recurrent pneumonia had an underlying disease. The most common cause was aspiration syndrome and viruses were leading causative agents for recurrent pneumonia. As a result; It should kept in mind that viruses can be commonly seen in recurrent pneumonia. Appropriate treatment choice should be made according to the underlying disease, clinical condition, laboratory and radiological data of the patient.

A 50-year-old woman was admitted to the emergency center with dyspnea, cough, and fever symptoms. She had a medical history of diabetes mellitus type II, rheumatoid arthritis, as well as several admission records due to aspiration pneumonia. The primary diagnosis was diabetic ketoacidosis and pneumonia. Normal breath sounds were reduced on the lower posterior right side of the thorax. A standard chest x-ray and lung Computed Tomography (CT) scan revealed collapse consolidation in the Right Lower Lobes (RLL) and Right Middle Lobes (RML). We here presented a case of unilateral diaphragmatic paralysis with a history of recurrent pneumonia.

A 58-year-old female with a history of recurrent pneumonia was evaluated for fevers, right lower back pain, and hematuria. A noncontrast abdominal computed tomography (CT) scan showed air and fluid-filled area in the right lower lobe for which a contrast-enhanced CT chest was performed. The CT of the chest revealed the cystic mass was supplied by an anomalous artery from the descending aorta. The patient was then diagnosed with a superinfected bronchopulmonary sequestration which was treated with surgical resection.

A 14-month-old male child presented with a history of recurrent pneumonia of the right upper lobe of the lung. Computed tomography scan showed an accessory bronchus proximal to the carina on the right side with collapse consolidation of the lobe.

A 50-year-old former smoker presented with recurrent pneumonia, fever and atypical chest pain on and off for a year and was treated with antibiotics several times. Meanwhile, her lung function deteriorated with an obstructive pattern and therefore she was treated with inhalation medication without effect. Tests of the heart, bronchoscopy, gastroscopy and a CT-scan of the lungs did not reveal the cause. Finally, an FDG-PET scan showed inflammation of the tissue on the backside of a 21-year-old silicone chest implant, which was intact but encapsulated. The chest implants were removed after which the patient has remained free of infections. However, the lung function impairment did not improve, and a new HRCT-scan demonstrated widespread emphysema. Keywords: Breast implants, Recurrent pneumonia, Smoking, Atypical chest pain

Tracheo-oesophageal fistula with oesophageal atresia is common life-threatening congenital malformations with an incidence of 1 in 3500 to 1 in 4500 births. ‘H’ type tracheo-oesophageal fistula is an isolated type with an incidence of 4–5% and least association with congenital anomalies. Its diagnosis is often delayed .We report two cases, one presented as a 3 months female child with ‘nasal regurgitation of feeds, recurrent pneumonia and choking on feeding since 15 days of life, while the other immediately after birth with classic triad of symptoms. A review of the literature is presented, with a focused discussion of presentation and treatment options. Confirmation of diagnosis was done by compiling clinical presentation, high index of suspicion and rigid tracheobronchoscopy findings. Management involved surgery by right transcervical approach. High index of clinical suspicion and early diagnosis can provide a better prognosis.

Cystic hygroma is a benign congenital malformation of lymphatic system, common location being cervico-facial region and axilla. We report a child with cystic hygroma, who had recurrent attacks of lower respiratory tract infection for surgery and the eventual anesthetic challenges associated with it.

Simultaneous occurrence of intracerebral hemorrhage (ICH) in different arterial territories is an uncommon event. We report on two cases of multiple spontaneous simultaneous ICH for which we could find no specific cause. A 73-year-old man, with no related medical history, was admitted to the hospital with simultaneous bithalamic ICH, and subsequently died of recurrent pneumonia. Second patient was a 60-year-old man who presented with simultaneous ICH in the pons and thalamus; he died of recurrent bleeding. We review the possible pathological mechanisms, clinical and radiologic features of simultaneous multiple ICH.

We present a 7.8-month-old girl with recurrent pneumonia, growth retardation, and tachypnea. Computed tomography revealed a particularly unusual esophageal lung, in association with pulmonary vascular anomalies of right pulmonary veins atresia and right pulmonary artery hypoplasia.

Introduction Cri du chat syndrome is a rare genetic disorder due to deletion of variable length of short arm of chromosome 5(5p). It mainly presents with typical cat like cry, facial dysmorphism, poor growth with feeding problems and severe cognitive, speech, and motor delays. Case Report We present here a one year old child who did not presented with typical features but presented with recurrent pneumonia with chronic stridor since early infancy. On evaluation we found short flabby epiglottis and we did karyotyping which showed 5P deletion syndrome. Conclusion Cri du chat syndrome can be presented with atypical features like chronic stridor and recurrent pneumonia without having typical cat like cry. Early diagnosis is necessary for proper counselling of parents and treatment of the patient.

Amongst the lung tumors, granular cell tumor (GCT), also known as granular cell myoblastoma, or Abrikossoff is one of the rarest tumors. Herein, we present a case of a 6-year-old male child who presented with a history of cough, fever and recurrent pneumonia due to a 8 × 5 × 3 cm GCT.

Aim . This study was performed in order to investigate clinical spectrum of immune deficiency syndrome (IDS) and its influence on the course of tuberculosis in HIV mono - infected or co - infected children. Methods . We analyzed historical data and carried out a randomized prospective study involving 125 children. Results . Infection - related IDS was diagnosed in all HIV - mono - infected children, children with HIV - associated tuberculosis and children with tuberculosis, i.e. in one third of the cases. Children with HIV and tuberculosis co - infection frequently had a history of respiratory diseases, such as recurrent tracheobronchitis and recurrent pneumonia, and extended and / or complicated pulmonary tuberculosis. Conclusion . Therefore, infection - related IDS could be a predictor of tuberculosis in children and should be considered in diagnosis of HIV monoinfection and co - infection together with other clinical and historical data.

The hyper-immunoglobulin E (IgE) syndromes (HIES) are primary immunodeficiencies characterized by the recurrent staphylococcal abscesses, recurrent pneumonia and highly elevated serum IgE levels. There are two forms of HIES: a dominant form (AD-HIES) and a recessive form (AR-HIES). AD form of HIES is caused by mutations in STAT3 and the AR form is caused by mutations in DOCK8 and TYK2. These syndromes have different clinical presentations and outcomes. AD-HIES is a multisystem disorder that includes abnormalities of the skin, lungs, musculo-skeletal system and dental system. In contrast, these symptoms in patients with AR-HIES are missing. AR-HIES patients have severe viral infections and may develop neurological complications. This review article discusses the clinical presentation and laboratory findings in both forms of HIES, as well as the establishment of diagnose, inheritance, molecular genetics and immunological abnormalities of HIES.

Background Consensus on treatment of chronic rhinosinusitis (CRS) in children with primary ciliary dyskinesia (PCD) is limited. We sought to synthesize the best available evidence pertaining to treatment and outcomes of CRS in children with PCD. Methods This work is a systematic review of PubMed and EMBASE for studies pertaining to treatment and outcomes of CRS in children with PCD in a 20-year period. Two reviewers independently extracted information on study design, setting, medical or surgical therapy for CRS, and treatment outcomes. Results Twelve studies from 8 countries were included for analysis: (3 retrospective cohorts, 2 prospective cohorts, 2 cross-sectional, 1 case series, and 4 case reports). The number of patients with PCD ranged from 1 to 78. Medical treatment included oral antibiotics, intranasal steroids, and oral steroids. Surgical treatment was mentioned in 8 studies, with endoscopic sinus surgery (ESS) described in 3 of 8. Outcome measures were rarely defined but included the 20-item Sino-Nasal Outcome Test (SNOT-20) questionnaire (1/12), symptom questionnaire (1/12), decreased recurrent pneumonia (1/12), and subjective report of CRS symptoms. No randomized, controlled, or long-term prospective cohort studies were identified, and no study employed a disease-specific, pediatric-validated instrument to measure outcomes of therapy. Conclusion There is a paucity of evidence pertaining to CRS treatment or outcomes in children with PCD. Given the rare prevalence of PCD and the insufficient evidence pertaining to treatment effectiveness for CRS, we propose an international, multicenter database to prospectively track data pertaining to diagnosis, treatment, and outcomes of CRS for children with PCD. This effort would encourage implementation of validated and standardized outcome measures.

Recurrent pneumonia (RP), i.e., at least two episodes of pneumonia in one year or three episodes ever with intercritical radiographic clearing of densities, occurs in 7.7%-9% of children with community-acquired pneumonia. In RP, the challenge is to discriminate between children with self-limiting or minor problems, that do not require a diagnostic work-up, and those with an underlying disease. The aim of the current review is to discuss a reasoned diagnostic approach to RP in childhood. Particular emphasis has been placed on which children should undergo a diagnostic work-up and which tests should be performed. A pediatric case series is also presented, in order to document a single centre experience of RP. A management algorithm for the approach to children with RP, based on the evidence from a literature review, is proposed. Like all algorithms, it is not meant to replace clinical judgment, but it should drive physicians to adopt a systematic approach to pediatric RP and provide a useful guide to the clinician.

Background: Recurrent pneumonia is defined as more than two episodes of pneumonia in one year or three or more episodes anytime in life. Common clinical scenarios leading to recurrent pneumonia include anatomical abnormalities of respiratory tract, immunodeficiency, congenital heart diseases, primary ciliary dyskinesia, etc. Case report: A school-aged girl suffered from 1-2 episodes of pneumonia each year after trachea connection and lung repair operation resulted from an accident of car crash. Bronchoscopy revealed the sutures twisted with granulation in the left main bronchus and the patient's symptoms relieved after removal of the sutures. Here we report for the first time that surgical suture was the cause of recurrent pneumonia. Conclusions: This case indicates that children with late and recurrent onset of pneumonia should undergo detailed evaluation including bronchoscopy.

Background Ring chromosome 14 syndrome is a rare chromosomal disorder characterized by early onset refractory epilepsy, intellectual disability, autism spectrum disorder and a number of diverse health issues. Results The aim of this work is to provide recommendations for the diagnosis and management of persons affected by ring chromosome 14 syndrome based on evidence from literature and experience of health professionals from different medical backgrounds who have followed for several years subjects affected by ring chromosome 14 syndrome. The literature search was performed in 2016. Original papers, meta-analyses, reviews, books and guidelines were reviewed and final recommendations were reached by consensus. Conclusion Conventional cytogenetics is the primary tool to identify a ring chromosome. Children with a terminal deletion of chromosome 14q ascertained by molecular karyotyping (CGH/SNP array) should be tested secondarily by conventional cytogenetics for the presence of a ring chromosome. Early diagnosis should be pursued in order to provide medical and social assistance by a multidisciplinary team. Clinical investigations, including neurophysiology for epilepsy, should be performed at the diagnosis and within the follow-up. Following the diagnosis, patients and relatives/caregivers should receive regular care for health and social issues. Epilepsy should be treated from the onset with anticonvulsive therapy. Likewise, feeding difficulties should be treated according to need. Nutritional assessment is recommended for all patients and nutritional support for malnourishment can include gastrostomy feeding in selected cases. Presence of autistic traits should be carefully evaluated. Many patients with ring chromosome 14 syndrome are nonverbal and thus maintaining their ability to communicate is always essential; every effort should be made to preserve their autonomy.

Cystic Fibrosis (CF) is an autosomal recessive disease that affects mucus and sweat producing cells involving multiple organs. CF is usually diagnosed in childhood; however, a considered number of adults are diagnosed every year. Atypical CF can be a milder form of the CF disorder, and individuals with atypical CF can remain undiagnosed for many years. Physicians should suspect of CF in adult individuals when these present recurrent pneumonia or bronchiectasis. Therefore, since early diagnosis of CF can avoid morbidities and unnecessary hospitalizations, the recognition of CF symptoms by clinicians is necessary to avoid late diagnosis.

Background In Japan and other societies with rapidly aging populations, recurrent pneumonia (RP) is a major clinical problem yet only limited information exists regarding the burden of this disease. Methods A prospective study of adult pneumonia was conducted to investigate the incidence of RP and potential risk factors. From February 1, 2012 to January 31, 2013, patients aged ≥ 15 years who were diagnosed with pneumonia were prospectively enrolled in a representative community hospital located in central Japan. Patients were followed for one-year to evaluate the recurrence of pneumonia and characteristics associated with RP. Cox proportional hazards models were constructed to compute adjusted hazard ratios (aHR) and ascertain risk factors significantly associated with RP. Results In total, 841 patients with a median age of 73 years (range 15–101 years) were enrolled totaling 1,048 person-years of observation with a median follow-up time of 475 days. A total of 137 patients had at least one recurrent episode with an incidence rate of 13.1 per 100 person-years (95% confidence interval: 11.1–15.5). In multivariate analysis, a past history of pneumonia (aHR 1.95, 95% CI: 1.35–2.8), chronic pulmonary disease (aHR 1.86, 1.24–2.78) and inhaled corticosteroid usage (aHR 1.78, 1.12–2.84) and hypnotic/sedative medication usage (aHR 2.06, 1.28–3.31) were identified as independent risk factors for recurrent pneumonia, whereas angiotensin converting enzyme-inhibitors usage was associated with a reduction of the risk of RP (aHR 0.22, 0.05–0.91). The detection of P. aeruginosa was significantly associated with RP even after adjusting for chronic pulmonary diseases (aHR = 2.37). Conclusions Recurrent pneumonia constitutes a considerable proportion of the pneumonia burden in Japan. A past history of pneumonia, chronic pulmonary disease, inhaled corticosteroid and hypnotic/sedative medication usage and detection of P. aeruginosa were identified as independent risk factors for recurrent pneumonia and special attention regarding the use of medications in this vulnerable population is needed to reduce the impact of this disease in aging populations. Electronic supplementary material The online version of this article (doi:10.1186/s12890-016-0359-1) contains supplementary material, which is available to authorized users.

Bronchial carcinoid tumor comprises 1 to 3% of lung neoplasms. The common age of onset is mainly post-puberty although atypical carcinoid tumors occur at ages 44 to 55. Carcinoid tumors cause two groups of symptoms in patients: symptoms due to obstruction and symptoms due to the production and release of active neuropeptides. Histologically, carcinoid tumors are categorized into two groups of typical and atypical while in terms of location of lesion, they are grouped into central and peripheral types. Differentiation between malignant and benign carcinoid tumors is based on presence or absence of metastasis. Bronchoscopy and endobronchial biopsy are the best diagnostic measures in these patients. Serologic evaluation and assessment of active metabolites in case of liver metastasis also help the diagnosis. Surgical resection is the treatment of choice for this condition. This report discusses a patient with carcinoid tumor who was receiving bronchodilator treatment for a couple of months because of chronic cough with possible diagnosis of asthma and had received several courses of antibiotic therapy with possible diagnosis of lung infection until he eventually developed bronchiectasis in the right lower lobe. In patients with chronic pulmonary symptoms especially with localized bronchiectasis, diagnostic bronchoscopy must be included in the diagnostic work-up to rule out intrabronchial lesions.

Tracheobronchial pediatric tumors are very rare and procedures like pneumonectomy are seldomly indicated due to the associated morbidity. If a surgical approach is considered, the ideal oncological technique would be the minimally invasive sleeve resection, allowing preservation of lung parenchyma (very important in pediatric patients). Here we present the first report of a thoracoscopic right upper tracheo-bronchial sleeve lobectomy in a pediatric patient. A 10-year-old female patient, who received multiple antibiotic treatments for recurrent pneumonia without improvement, was diagnosed with a right upper lobe (RUL) carcinoid tumor. The patient was proposed for uniportal thoracoscopic surgery. The patient was placed in a lateral decubitus position and a single 3 cm incision was performed at the anterior level of 4th intercostal space. A right upper lobectomy with a tracheo-bronchial sleeve resection using the uniportal technique was successfully performed. The postoperative course was uneventful and the patient was discharged home on the 7th postoperative day. The bronchoscopic control showed excellent caliber of the anastomosis with no complications. The uniportal video-assisted thoracoscopic surgery (VATS) approach is an excellent option for endobronchial tumor management in pediatric patients, offering a quick recovery and low morbidity. The performance of a thoracoscopic sleeve anastomosis in young patients is crucial and should only be performed by very experienced thoracoscopic surgeons.

Congenital Lobar Emphysema (CLE) is a medical condition which is quiet rarely seen especially in neonates. The disease is characterized by over inflation of lungs which is produced due to inhibition of escape of inspired air from lungs. This condition usually occurs in infants due to partial obstruction of the bronchus and can be reversed with timely and planned anaesthetic management. Here in, we are presenting a case of an infant who was merely four months of age. CLE was affecting his left upper lobe since birth which was being treated as lobar pneumonia outside at some peripheral health center. The case presented to hospital with the symptoms acute respiratory distress and was taken up for urgent surgical management. Left upper lobectomy was performed which relived his symptoms. Case was discharged from hospital after 10 days of uneventful hospital stay. The case was challenging, as it involved careful and planned anaesthetic management of lung separation as well as prevention of hyperventilation of the un-involved lung.