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1
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Kiessling P, Kim G, Meister K, Balakrishnan K, Din TF. Pediatric tracheal mucoepidermoid carcinoma treated with cricotracheal resection: A rare case and review of literature. OTOLARYNGOLOGY CASE REPORTS 2025; 34:100654. [PMID: 40190414 PMCID: PMC11970917 DOI: 10.1016/j.xocr.2025.100654] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/09/2025] Open
Abstract
Mucoepidermoid carcinoma (MEC) is a rare pathology in children, and even less commonly seen in the pediatric trachea. Here we describe an adolescent patient who presented with exercise intolerance following COVID-19 infection and was found to have a near-totally obstructive subglottic mass. This mass was debulked and biopsied, demonstrating MEC. Cricotracheal resection with cervical tracheoplasty was performed. A comprehensive review of the published cases of pediatric tracheal MEC was then conducted. This review of 19 patients comprises the largest collection of pediatric tracheal MEC cases analyzed to date, revealing important collective trends and insights for this rare diagnosis.
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Affiliation(s)
- Patrick Kiessling
- Department of Otolaryngology – Head & Neck Surgery, Stanford Health Care, Stanford, CA, USA
| | - Grace Kim
- Department of Otolaryngology – Head & Neck Surgery, Stanford Health Care, Stanford, CA, USA
- Division of Pediatric Otolaryngology – Head & Neck Surgery, Lucille Packard Children’s Hospital, Stanford Children’s Health, Stanford, CA, USA
| | - Kara Meister
- Department of Otolaryngology – Head & Neck Surgery, Stanford Health Care, Stanford, CA, USA
- Division of Pediatric Otolaryngology – Head & Neck Surgery, Lucille Packard Children’s Hospital, Stanford Children’s Health, Stanford, CA, USA
| | - Karthik Balakrishnan
- Department of Otolaryngology – Head & Neck Surgery, Stanford Health Care, Stanford, CA, USA
- Division of Pediatric Otolaryngology – Head & Neck Surgery, Lucille Packard Children’s Hospital, Stanford Children’s Health, Stanford, CA, USA
| | - Taseer Feroze Din
- Division of Pediatric Otolaryngology – Head & Neck Surgery, Lucille Packard Children’s Hospital, Stanford Children’s Health, Stanford, CA, USA
- Division of Pediatric Otolaryngology, Sidra Medicine, Ar-Rayyan, Doha, Qatar
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2
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Abulhamail A, Okuducu Y, Sagar P, Kinane B, Onge IS, Coates A, Nelson BA. Recurrent pneumonia in an otherwise healthy child. Pediatr Pulmonol 2024; 59:3706-3711. [PMID: 39194356 DOI: 10.1002/ppul.27226] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/17/2024] [Revised: 08/10/2024] [Accepted: 08/12/2024] [Indexed: 08/29/2024]
Affiliation(s)
- Adila Abulhamail
- Division of Pediatric Pulmonology, MassGeneral for Children, Harvard Medical School, Boston, Massachusetts, USA
| | - Yanki Okuducu
- Division of Pediatric Pulmonology, MassGeneral for Children, Harvard Medical School, Boston, Massachusetts, USA
| | - Pallavi Sagar
- Department of Pediatric Radiology, MassGeneral for Children, Harvard Medical School, Boston, Massachusetts, USA
| | - Bernard Kinane
- Division of Pediatric Pulmonology, MassGeneral for Children, Harvard Medical School, Boston, Massachusetts, USA
| | - Ina St Onge
- Department of Pediatrics, Tufts University School of Medicine at Maine Medical Center, Portland, Maine, USA
| | - Anne Coates
- Department of Pediatrics, Tufts University School of Medicine at Maine Medical Center, Portland, Maine, USA
| | - Benjamin A Nelson
- Division of Pediatric Pulmonology, MassGeneral for Children, Harvard Medical School, Boston, Massachusetts, USA
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3
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Figueroa-Hurtado E, Peña MJ, Cortes-Telles A. Asthma Symptoms Mimicking Myofibroblastic Tracheal Tumor in Pediatric Diagnosis. Cureus 2024; 16:e74097. [PMID: 39712775 PMCID: PMC11661893 DOI: 10.7759/cureus.74097] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 11/19/2024] [Indexed: 12/24/2024] Open
Abstract
Tracheal tumors in pediatric patients are rare, accounting for 2% of all airway abnormalities and 0.2% of all pediatric tumors. Diagnosis is often delayed due to the heterogeneity of presenting symptoms, such as stridor and wheezing, which are frequently misattributed to other conditions. We report the case of a previously healthy nine-year-old male who was diagnosed with an inflammatory myofibroblastic tumor (IMT) following five months of persistent airway symptoms, including cough, biphasic stridor, wheezing, and dyspnea. Despite evaluation by multiple physicians and treatment for presumed asthma, his symptoms did not fully resolve. Imaging studies ultimately confirmed the diagnosis, and surgical resection of the tracheal tumor was performed. In the late postoperative period (12 weeks), the patient continued to experience cough and dyspnea. Given a family history of asthma (father with asthma), spirometry with a bronchodilator was conducted, confirming a diagnosis of asthma alongside IMT. The patient is currently alive and undergoing treatment in Step 2 of the Global Initiative for Asthma (GINA) guidelines. This case highlights the importance of a thorough evaluation in children with persistent stridor and wheezing to rule out underlying tracheobronchial pathologies.
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Affiliation(s)
- Esperanza Figueroa-Hurtado
- Respiratory Diseases Clinic, Regional Hospital of High Specialty of the Yucatan Peninsula, Instituto Mexicano del Seguro Social-Bienestar, Merida, MEX
| | - Mario J Peña
- Thoracic Surgery, Hospital Infantil de México Federico Gomez, Mexico City, MEX
| | - Arturo Cortes-Telles
- Respiratory Diseases Clinic, Regional Hospital of High Specialty of the Yucatan Peninsula, Instituto Mexicano del Seguro Social-Bienestar, Merida, MEX
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4
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Promsawasdi K, Phewplung T. Pediatric endobronchial tumors with a mimicker: A case series. Pediatr Pulmonol 2024; 59:2669-2678. [PMID: 38837689 DOI: 10.1002/ppul.27106] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/07/2024] [Accepted: 05/23/2024] [Indexed: 06/07/2024]
Affiliation(s)
- Kulwiwat Promsawasdi
- Department of Radiology, Faculty of Medicine, Chulalongkorn University and King Chulalongkorn Memorial Hospital, Bangkok, Thailand
| | - Teerasak Phewplung
- Department of Radiology, Faculty of Medicine, Chulalongkorn University and King Chulalongkorn Memorial Hospital, Bangkok, Thailand
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5
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Liu D, Liu X, Li X, Liu Y, Yu J. Primary endobronchial multifocal Ewing's sarcoma: a rare case report. Front Oncol 2024; 14:1431950. [PMID: 39281384 PMCID: PMC11392889 DOI: 10.3389/fonc.2024.1431950] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/13/2024] [Accepted: 06/25/2024] [Indexed: 09/18/2024] Open
Abstract
Extraskeletal Ewing's sarcoma (ES) has been reported to originate from various sites. Primary endobronchial ES is an extremely rare bronchial tumor, especially multifocal lesions. This report describes a rare presentation of primary bronchial ES in a 31-year-old female who was referred to the emergency department of our hospital due to suspicion of a foreign body in the bronchus. Computed tomography and bronchoscopy revealed multiple polypoid nodules in the middle bronchus of her right lung, thus excluding the initial diagnosis. Infection-related laboratory tests and serum tumor markers were normal. The bronchial sleeve resection was performed to remove the tumor completely and the patient's clinical symptoms obviously improved. Subsequent imaging, histopathological, immunohistochemical and genetic analyses made a conclusive diagnosis of primary endobronchial ES. To our knowledge, this is the eighth case of primary bronchial ES reported in medical literature.
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Affiliation(s)
- Dan Liu
- Department of Radiology, Sichuan Provincial People's Hospital, University of Electronic Science and Technology of China, Chengdu, China
| | - Xiaoge Liu
- Department of Ultrasound, Ya'an People's Hospital, Ya'an, China
| | - Xin Li
- Department of Radiology, Sichuan Provincial People's Hospital, University of Electronic Science and Technology of China, Chengdu, China
| | - Yisha Liu
- Department of Pathology, Sichuan Provincial People's Hospital, University of Electronic Science and Technology of China, Chengdu, China
| | - Junlun Yu
- Department of Radiology, Sichuan Provincial People's Hospital, University of Electronic Science and Technology of China, Chengdu, China
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6
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Shchomak Z, Oliveira L, Saianda A, Bandeira T. Endobronchial lesion in a premature neonate. BMJ Case Rep 2024; 17:e259839. [PMID: 38508601 PMCID: PMC10952865 DOI: 10.1136/bcr-2024-259839] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 03/22/2024] Open
Abstract
Endobronchial and endotracheal tumours are rare in the paediatric population, especially in neonates. The common presentation is respiratory distress with persistent lung collapse or hyperinflation. Treatment usually involves endoscopic or surgical lobar resection. This case presents a preterm neonate who developed acute respiratory distress with persistent right upper lobe atelectasis. A flexible bronchoscopy showed a pediculate violaceous endobronchial lesion in the right main bronchus. Endoscopic resection was not possible due to the patient's low weight and the decision was made to support the patient with continuous positive airway pressure (CPAP) at home, while monitoring her closely. Remarkably, the lesion spontaneously resolved, and CPAP support was discontinued successfully. The case emphasises the importance of early bronchoscopy, continuous monitoring and the possibility of using CPAP support until conditions are reunited for an eventual resection. This unique case also demonstrates the potential for spontaneous resolution in some cases.
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Affiliation(s)
- Zakhar Shchomak
- Pediatrics Department, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal
| | - Lia Oliveira
- Pediatrics Department, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal
| | - Ana Saianda
- Pediatrics Department, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal
| | - Teresa Bandeira
- Pediatrics Department, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal
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7
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Boyle MA, Rotimi O, Palmares A, Alvarez Gallesio J, Alshammari A, Semple T, Buderi S, Jordan S. Surgical Management in a Paediatric Case of Endobronchial Mucoepidermoid Carcinoma Involving the Carina. Cureus 2023; 15:e48680. [PMID: 37965236 PMCID: PMC10642615 DOI: 10.7759/cureus.48680] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 11/12/2023] [Indexed: 11/16/2023] Open
Abstract
Although mucoepidermoid carcinoma (MEC) is the most diagnosed malignancy of the salivary gland, it rarely localises to the bronchus, accounting for only 0.1-0.2% of all primary lung malignancies. Of those pulmonary MECs, most are found in segmental or lobar bronchi, and they are rarely found in mainstem bronchi, highlighting the novelty of this presentation for thoracic specialists. We present a case report of a seven-year-old female who underwent a carinal resection and a right upper lobectomy for the management of an endobronchial MEC causing right middle lobe (RML) obstruction. Intraoperatively, an exophytic mass originating from the junction of the right main bronchus and bronchus intermedius was identified, causing a partial obstruction of the RML bronchus. Frozen sections demonstrated clear margins and follow-up bronchoscopies have been unremarkable. Given their rarity, endobronchial MECs can be diagnostically difficult and cause uncertainty with respect to their management. Low-grade tumours have a much more favourable prognosis than their high-grade counterparts, with surgical resection being the gold standard of care. Therefore, the index of suspicion, time to diagnosis, and definitive treatment are critical to the outcome.
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Affiliation(s)
- Mark A Boyle
- Department of Surgery and Cancer, Imperial College London, London, GBR
- Department of Thoracic Surgery, Royal Brompton Hospital, London, GBR
| | - Oloruntobi Rotimi
- Department of General Surgery, Medway NHS Foundation Trust, Gillingham, GBR
| | - Abigail Palmares
- Department of Thoracic Surgery, Royal Brompton Hospital, London, GBR
| | | | | | - Thomas Semple
- Department of Radiology, Royal Brompton Hospital, London, GBR
| | - Silviu Buderi
- Department of Thoracic Surgery, Royal Brompton Hospital, London, GBR
| | - Simon Jordan
- Department of Thoracic Surgery, Royal Brompton Hospital, London, GBR
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8
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Dumitru CȘ, Balica NC. Subglottotracheal Adenoid Cystic Carcinoma in a 16-Year-Old Female-A Case Report. MEDICINA (KAUNAS, LITHUANIA) 2023; 59:1140. [PMID: 37374345 DOI: 10.3390/medicina59061140] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/09/2023] [Revised: 06/02/2023] [Accepted: 06/12/2023] [Indexed: 06/29/2023]
Abstract
Cystic adenoid carcinoma (ACC) is a rare malignant epithelial tumor arising from exocrine glands and accounts for only 1% of head and neck cancers. ACCs are common in the fifth and sixth decades of life, predominantly in women, and characterized by slow progression, local aggression, recurrence, and high metastasis. Subglottotracheal ACC is a rare tumor in the pediatric population, with only a few cases reported in the literature. We present a case of a 16-year-old female who was diagnosed with ACC in the subglottic and tracheal region. The patient presented with respiratory failure but without a history of dysphonia, dyspnea, stridor, or dysphagia. The diagnosis was confirmed by a biopsy, and subsequent imaging studies showed a large tumor involving the subglottic and tracheal region. The therapeutic management of this patient has been challenging due to the rarity of this tumor in the pediatric population and the potential long-term complications associated with tumor recurrence and psychological impact. This case highlights the diagnostic and therapeutic challenges in the management of subglottotracheal ACC in children and the importance of a multidisciplinary approach to optimize patient outcomes.
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Affiliation(s)
- Cristina Ștefania Dumitru
- Department of Microscopic Morphology/Histology, Angiogenesis Research Center, "Victor Babes" University of Medicine and Pharmacy, Sq. EftimieMurgu No. 2, 300041 Timisoara, Romania
| | - Nicolae Constantin Balica
- Department of Ear-Nose-Throat, "Victor Babes" University of Medicine and Pharmacy, Eftimie Murgu Square 2, 300041 Timisoara, Romania
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9
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Muacevic A, Adler JR, Rhazari M, Thouil A, Marouf R, Kouismi H. Tracheal Hamartoma: A Case Report. Cureus 2022; 14:e32128. [PMID: 36601160 PMCID: PMC9805699 DOI: 10.7759/cureus.32128] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 12/02/2022] [Indexed: 12/03/2022] Open
Abstract
Hamartoma is the most frequently observed benign lung tumor, but its tracheal form is still exceptionally encountered. Cough, dyspnea, hemoptysis, and chest pain are all possible symptoms of tracheal hamartoma. The non-specific symptoms may also lead to a delayed diagnosis, and while the choice of treatment varies depending on the size and location of the lesion, conservative treatments remain strongly recommended. This report presents the case of a 57-year-old male who presented to our department with inspiratory dyspnea. Clinico-radiological data and bronchoscopy revealed a benign tracheal tumor of the lipomatous hamartoma type. The patient underwent a tumor resection by rigid bronchoscopy with satisfactory clinical results.
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10
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A 19-Year-Old Man with a Cavitating Lung Lesion. Ann Am Thorac Soc 2022; 19:1920-1924. [PMID: 36318076 DOI: 10.1513/annalsats.202202-129cc] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/05/2022] Open
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11
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Zhao Z, Kim RC, Tavernier F, Choksi R, Van Brunt T, Davis JE, Kevill K, Hsieh H. A Young Child With Recurrent Pneumonia and Hemoptysis During the COVID-19 Pandemic. Chest 2022; 162:e77-e80. [PMID: 35940666 PMCID: PMC9353104 DOI: 10.1016/j.chest.2022.03.053] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/17/2022] [Accepted: 03/11/2022] [Indexed: 11/23/2022] Open
Abstract
In July 2020, a previously healthy 6-year-old boy was evaluated in a pulmonary clinic in New York after two episodes of pneumonia in the previous 3 months. For each episode, the patient presented with cough, fever, and hemoptysis, all of which resolved with antibiotic therapy and supportive care. The patient never experienced dyspnea during these episodes of pneumonia. He was asymptomatic at the current visit. The patient had no history of travel, sick contacts, asthma, or bleeding disorders.
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12
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Sultan M, Beza L, Debebe F, Hassen GW, Duvvi A, Tilahun S, Nasser N, Bekele S. Distal Tracheal Obstructive Mass Leading to Bilateral Pneumothorax and Respiratory Failure. Open Access Emerg Med 2022; 14:217-222. [PMID: 35651588 PMCID: PMC9148919 DOI: 10.2147/oaem.s363020] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/01/2022] [Accepted: 05/13/2022] [Indexed: 11/23/2022] Open
Abstract
Tracheal masses are rare in occurrence, but could lead to complications depending on the speed of growth, duration and degree of obstruction. Some of the complications are recurrent pneumonia and air trapping resulting in increased intrathoracic pressure. The latter phenomenon can result in obstruction of the venous return and pneumothorax. We are reporting a rare presentation of bilateral pneumothorax (presumed tensioned) in a young patient with a distal obstructive tracheal tumor. In the emergency department (ED) the patient was in respiratory distress and was found to have extensive subcutaneous emphysema of the neck, chest, and abdominal wall with hypotension. Respiratory failure from bilateral tension pneumothorax was suspected and the patient was intubated with simultaneous bilateral thoracostomy. These measures did not improve the patient's ventilation and oxygenation status. Further fiberoptic investigation revealed a distal tracheal obstructive mass. An emergency surgical intervention was required to remove the tumor. We recommend considering alternative pathologies, such as an obstructive tracheal tumor, in a patient with respiratory distress. They should especially be considered when oxygenation and ventilation are difficult, particularly when endotracheal intubation and/or tube thoracostomy fail to improve the symptoms. A high index of suspicion and a timely multidisciplinary team approach are essential when managing the life-threatening presentation of a patient with a distal tracheal tumor.
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Affiliation(s)
- Menbeu Sultan
- Department of Emergency Medicine and Critical Care, St. Paul's hospital millennium medical College, Addis Ababa, Ethiopia
| | - Lemlem Beza
- Department of Emergency Medicine, Addis Ababa University, Addis Ababa, Ethiopia
| | - Finot Debebe
- Department of Emergency Medicine, Addis Ababa University, Addis Ababa, Ethiopia
| | - Getaw Worku Hassen
- Department of Emergency Medicine, New York Metropolitan Hospital, New York, NY, USA
| | - Anisha Duvvi
- Department of Emergency Medicine, New York Metropolitan Hospital, New York, NY, USA
| | - Selamawit Tilahun
- Department of Internal Medicine, St. Paul's Hospital Millennium Medical College, Addis Ababa, Ethiopia
| | - Nura Nasser
- Department of Anesthesiology, St Paul's Hospital Millennium Medical College, Addis Ababa, Ethiopia
| | - Sisay Bekele
- Department of Surgery, St. Paul's Hospital Millennium Medical College, Addis Ababa, Ethiopia
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13
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Scolnik M, Glozman L, Bar-Yoseph R, Gur M, Toukan Y, Bentur L, Ilivitzki A. Atypical pulmonary metastases in children: the spectrum of radiologic findings. Pediatr Radiol 2021; 51:1907-1916. [PMID: 33847786 DOI: 10.1007/s00247-021-05035-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/25/2020] [Revised: 10/01/2020] [Accepted: 02/22/2021] [Indexed: 11/27/2022]
Abstract
Pulmonary nodules present a diagnostic challenge when they appear as atypical metastases in pediatric oncology patients. Chest computed tomography (CT) is the primary imaging modality for assessing lung nodules. In pediatric populations, Wilms tumor and osteosarcoma are the cancers most likely to produce pulmonary metastasis, both typical and atypical. This pictorial essay provides a thorough description of the specific radiologic features of atypical pediatric pulmonary metastases, and their pathogenesis and differential diagnosis. We also address diagnostic approaches to incidental lung nodules in healthy children found in the literature. Our aim is to help radiologists identify atypical lung metastases on CT, ensuring that children receive prompt, and potentially lifesaving, treatment.
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Affiliation(s)
- Michal Scolnik
- Pediatric Radiology Unit, Ruth Rappaport Children's Hospital, 8 HaAliya HaShniya Street, 3109601, Haifa, Israel
| | - Luda Glozman
- Pediatric Radiology Unit, Ruth Rappaport Children's Hospital, 8 HaAliya HaShniya Street, 3109601, Haifa, Israel
- Department of Radiology, Rambam Health Care Campus, Haifa, Israel
| | - Ronen Bar-Yoseph
- Pediatric Pulmonary Institute, Ruth Rappaport Children's Hospital, Rambam Health Care Campus, Haifa, Israel
- The Bruce Rappaport Faculty of Medicine, Technion Israel Institute of Technology, Haifa, Israel
| | - Michal Gur
- Pediatric Pulmonary Institute, Ruth Rappaport Children's Hospital, Rambam Health Care Campus, Haifa, Israel
- The Bruce Rappaport Faculty of Medicine, Technion Israel Institute of Technology, Haifa, Israel
| | - Yazeed Toukan
- Pediatric Pulmonary Institute, Ruth Rappaport Children's Hospital, Rambam Health Care Campus, Haifa, Israel
- The Bruce Rappaport Faculty of Medicine, Technion Israel Institute of Technology, Haifa, Israel
| | - Lea Bentur
- The Bruce Rappaport Faculty of Medicine, Technion Israel Institute of Technology, Haifa, Israel
| | - Anat Ilivitzki
- Pediatric Radiology Unit, Ruth Rappaport Children's Hospital, 8 HaAliya HaShniya Street, 3109601, Haifa, Israel.
- Department of Radiology, Rambam Health Care Campus, Haifa, Israel.
- The Bruce Rappaport Faculty of Medicine, Technion Israel Institute of Technology, Haifa, Israel.
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14
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Ferraro VA, Baraldi E, Stabinger D, Zamunaro A, Zanconato S, Carraro S. Pediatric flexible bronchoscopy: A single-center report. Pediatr Pulmonol 2021; 56:2634-2641. [PMID: 33969642 PMCID: PMC8360175 DOI: 10.1002/ppul.25458] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/27/2020] [Revised: 04/16/2021] [Accepted: 05/02/2021] [Indexed: 12/27/2022]
Abstract
INTRODUCTION Pediatric flexible laryngotracheal bronchoscopy (FB) is an integral part of diagnostics and treatment at tertiary pediatric respiratory centers. AIM FBs performed between 2013 and 2018 at our Pediatric Allergy and Respiratory Medicine Unit of the Department of Women's and Children's Health at Padua University were examined in terms of the indications, findings, and adverse events. MATERIALS AND METHODS The electronic medical records of pediatric patients who underwent FB at least once between 1 January 2013 and 31 December 2018 were considered. Patients' clinical data, indications for FB, anatomical findings, information derived from bronchoalveolar lavage (BAL) and bronchial brushing, and possible adverse events were analyzed. RESULTS There were 447 pediatric FBs performed in 428 patients (aged from 1 month to 18 years) for diagnostic purposes (92.4%), to clear secretions (3.6%), or to monitor a known condition (4.0%). The main indications were recurrent lower respiratory tract infections (LRTI, 32.2%) and chronic wet cough (9.4%). Lower airway malacia was the most common abnormal finding in these two groups (36.1% and 28.6%, respectively). BAL bacterial culture was positive in 55 children (39.6%) with recurrent LRTI and in 25 (59.5%) with chronic wet cough, being Haemophilus influenzae, Streptococcus pneumoniae, and Moraxella catarrhalis the microorganisms most commonly isolated. FB proved a safe procedure and was well tolerated. CONCLUSIONS Pediatric FB is an essential tool at our tertiary pediatric respiratory center. It helps establish the anatomical conditions underlying several chronic respiratory conditions and any correlated microbiological findings, with a significant impact on further patient management.
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Affiliation(s)
| | - Eugenio Baraldi
- Department of Women's and Children's Health, University of Padova, Padua, Italy
| | - Diana Stabinger
- Department of Women's and Children's Health, University of Padova, Padua, Italy
| | - Andrea Zamunaro
- Department of Women's and Children's Health, University of Padova, Padua, Italy
| | - Stefania Zanconato
- Department of Women's and Children's Health, University of Padova, Padua, Italy
| | - Silvia Carraro
- Department of Women's and Children's Health, University of Padova, Padua, Italy
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15
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MacInnes R, Warris A. Paediatric Histoplasmosis 2000-2019: A Review of 83 Cases. J Fungi (Basel) 2021; 7:jof7060448. [PMID: 34199970 PMCID: PMC8229079 DOI: 10.3390/jof7060448] [Citation(s) in RCA: 12] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/14/2021] [Revised: 05/31/2021] [Accepted: 06/01/2021] [Indexed: 12/22/2022] Open
Abstract
Histoplasmosis is an endemic fungal infection that is confined to specific geographical regions. Histoplasma spp. are primary pathogens that cause disease in both immunocompetent and immunocompromised patients, ranging from a single-organ (mostly affecting the lungs) infection to life-threatening disseminated disease. Knowledge about the clinical epidemiology relies on data from adult populations; little is known about the patient and disease characteristics in the paediatric population. Therefore, a structured review of published cases of paediatric histoplasmosis between 2000 and 2019 was performed. A literature search of PubMed was conducted and the epidemiological and clinical data from 83 cases were analysed. The mean age at presentation was 9.5 ± 5.5 years, and 51% were girls. Two-thirds of the children were immunocompromised. The majority of children presented with disseminated disease. The most frequently observed clinical symptoms were respiratory symptoms, alongside non-specific systemic features, including fever, myalgia, fatigue and weight loss. The mortality rate was 11%. Histoplasmosis affects children of any age. Being immunocompromised is a risk factor for severe and disseminated disease. The lack of specific presenting features leads to underreporting and delay in diagnosis. To improve the recognition and outcome of histoplasmosis in childhood, increased awareness and surveillance systems are warranted.
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Affiliation(s)
- Rebecca MacInnes
- Aberdeen Fungal Group, Institute of Medical Sciences, University of Aberdeen, Aberdeen AB25 2ZD, UK;
| | - Adilia Warris
- Aberdeen Fungal Group, Institute of Medical Sciences, University of Aberdeen, Aberdeen AB25 2ZD, UK;
- MRC Centre for Medical Mycology, University of Exeter, Geoffrey Pope Building, Stocker Road, Exeter 4EX 4QD, UK
- Correspondence: ; Tel.: +44-1392-727593
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16
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Virgone C, Ferrari A, Chiaravalli S, De Pasquale MD, Inserra A, D'Angelo P, Ogunleye MF, Crocoli A, Vallero S, Cesaro S, Alaggio R, Bisogno G, Dall'Igna P. Extra-appendicular neuroendocrine tumors: A report from the TREP project (2000-2020). Pediatr Blood Cancer 2021; 68:e28880. [PMID: 33522705 DOI: 10.1002/pbc.28880] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/08/2020] [Revised: 12/15/2020] [Accepted: 12/16/2020] [Indexed: 11/06/2022]
Abstract
BACKGROUND Extra-appendicular neuroendocrine tumors (NETs) are very rare tumors. While diagnostic and therapeutic guidelines are well established for adults, data on children and adolescents are lacking. PATIENTS AND METHODS Patients with a diagnosis of extra-appendicular NET registered on the Tumori Rari in Età Pediatrica - Rare Tumors in Pediatric Age (TREP) from 2000 to 2020 were analyzed. Clinical characteristics including patients' presentation, tumor features, treatment, and outcome were reviewed. RESULTS Twenty-seven patients with extra-appendicular NET registered on TREP with a median age of 173 months. The primary site was the pancreas (12) or bronchi (10) in the majority of cases. Other primary sites included the thymus, Meckel's diverticulum, and liver. Thirteen (48%) of tumors extended beyond the organ of origin: four invaded neighboring organs and/or regional nodes and nine involved distant metastases. The 3-year event-free survival (EFS) for those with localized disease was superior to those with metastatic disease (66.6% 95% CI 5-95% vs 33% 95% CI 5-68%, respectively; P = .005). A complete resection was feasible in 17 patients. The 3-year EFS in these patients was superior to those with no or incomplete resection (R0 vs R1/R2, respectively; P = .007). Overall, 16 children had no evidence of disease at follow-up, and one is alive with disease; five died, and five were lost to follow-up. CONCLUSIONS Data from our experience demonstrated a wide heterogeneity of presentation and outcome of these tumors. Localized disease and complete surgical resection were the main prognostic factors of good outcome. Other therapies may have a role in prolonging survival in metastatic disease.
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Affiliation(s)
- Calogero Virgone
- Pediatric Surgery, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy
| | - Andrea Ferrari
- Hematology Oncology, IRCCS Istituto Nazionale dei Tumori, Milan, Italy
| | | | | | - Alessandro Inserra
- Department of Pediatric Surgery, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Paolo D'Angelo
- Hematology/Oncology Unit, A.R.N.A.S. Civico Di Cristina e Benfratelli, Palermo, Italy
| | - Martina Funmilayo Ogunleye
- Pediatric Surgery, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy
| | - Alessandro Crocoli
- Department of Pediatric Surgery, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Stefano Vallero
- Department of Pediatric Onco-Hematology, Regina Margherita Children's Hospital, Turin, Italy
| | - Simone Cesaro
- Pediatric Hematology-Oncology, Azienda Ospedaliera Universitaria Integrata, Verona, Italy
| | - Rita Alaggio
- Department of Pathology, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Gianni Bisogno
- Hematology Oncology, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy
| | - Patrizia Dall'Igna
- Pediatric Surgery, Department of Emergencies and Organ Transplantation, Azienda Ospedaliero-Universitaria Consorziale Ospedale Pediatrico Giovanni XXIII, Bari, Italy
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17
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Paraskakis E, Froudarakis M, Tsalkidou EA, Deftereos S, Sarris G, Tsalkidis A. An eight-year-old girl with tracheal mass treated as a difficult asthma case. J Asthma 2020; 58:1689-1693. [PMID: 32991249 DOI: 10.1080/02770903.2020.1825730] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/23/2022]
Abstract
INTRODUCTION Endobronchial masses such as mucoepidermoid carcinomas or carcinoid tumors are extremely rare in children and they usually originate from large bronchi. These lesions may cause wheezing and dyspnea with poor response to bronchodilators and mimic the airway obstruction caused by asthma. CASE STUDY We present the case of an 8-year-old girl with tracheal mucoepidermoid carcinoma who was treated as a difficult asthma case with high dose of inhaled corticosteroids. RESULTS The characteristic stridor, the lack of response to bronchodilators and to inhaled corticosteroid treatment, combined with the characteristic flow loop in spirometry and the hyperinflation seen on the chest radiograph, all raised the clinical suspicion of a tracheal lesion and indicated the need for flexible bronchoscopy. The bronchoscopy revealed a large lesion obstructing totally the trachea lumen. The latter finding was confirmed by chest high resolution CT. The mass was completely excised via sternotomy under cardiopulmonary bypass, and the pathologic examination showed a low-grade mucoepidermoid carcinoma of the trachea. One month after the surgery she was free of symptoms and her spirometry was normal. CONCLUSION Tracheal lesions mimic the symptoms of airway obstruction caused by asthma and should be always be part of the differential diagnosis in young patients with no response to asthma treatment.
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Affiliation(s)
- Emmanouil Paraskakis
- Department of Paediatrics, Democritus University of Thrace, Alexandroupolis, Greece
| | - Marios Froudarakis
- Department of Pulmonology, Democritus University of Thrace, Alexandroupolis, Greece
| | - Evanthia A Tsalkidou
- Department of Paediatrics, Democritus University of Thrace, Alexandroupolis, Greece
| | - Savvas Deftereos
- Department of Radiology, Democritus University of Thrace, Komotini, Greece
| | - George Sarris
- Department of Pediatric Cardiac Surgery, Athens Heart Surgery Institute, Athens, Greece
| | - Aggelos Tsalkidis
- Department of Paediatrics, Democritus University of Thrace, Alexandroupolis, Greece
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18
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Rizk A, Salam MA, Elnahas E, Fekry H, Salem M, Magdy M, Zidan M, Nosseir A, Eldowik Y, Kotb A, Mahmoud MI, Elmallawany H. Bronchoscopic management of endobronchial carcinoid presenting as asthma Mimic. Respir Med Case Rep 2020; 29:101020. [PMID: 32140402 PMCID: PMC7044744 DOI: 10.1016/j.rmcr.2020.101020] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/02/2019] [Revised: 02/03/2020] [Accepted: 02/04/2020] [Indexed: 11/29/2022] Open
Abstract
We report a 25-year-old woman with persistent dyspnea and wheezes that had been unsuccessfully treated with inhaled beta 2-agonists and steroids for about one year. Spirometry demonstrated a restrictive pattern. Chest CT demonstrated polypoidal lesion in left main bronchus. The lesion was excised via rigid bronchoscopy. Pathology showed a picture of typical bronchial carcinoid. In this patient, due to the lack of awareness, diagnosis of carcinoid was delayed for one year.
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Affiliation(s)
- Ayman Rizk
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Mohamed A Salam
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Engy Elnahas
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Hasnaa Fekry
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Mohamed Salem
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Marwa Magdy
- Interventional Pulmonology Unit of Alexandria Police Hospital, Egypt
| | - Mohamed Zidan
- Chest Department of University Hospital of Alexandria, Egypt
| | - Ayman Nosseir
- Thoracic Surgery Department University Hospital of Alexandria, Egypt
| | - Yasser Eldowik
- Pathology Department, Faculty of Medicine, Al-Azhar University, Egypt
| | - Adel Kotb
- Chest Department of University Hospital of Alexandria, Egypt
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19
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Pio L, Varela P, Eliott MJ, Couloigner V, Guillén Burrieza G, Paraboschi I, Virgone C, Maunsell R, Rachkov V, Rutter MJ, Boglione M, Penchyna Grub J, Bellía Munzón G, Sarnacki S, Irtan S, Schweiger C, Larroquet M, Khen Dunlop N, Ramaswamy M, Pistorio A, Cecchetto G, Ferrari A, Bisogno G, Torre M. Pediatric airway tumors: A report from the International Network of Pediatric Airway Teams (INPAT). Laryngoscope 2019; 130:E243-E251. [PMID: 31090942 DOI: 10.1002/lary.28062] [Citation(s) in RCA: 13] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/15/2018] [Revised: 04/11/2019] [Accepted: 04/22/2019] [Indexed: 12/26/2022]
Abstract
OBJECTIVE Primary tracheobronchial tumors (PTTs) are rare heterogeneous lesions arising from any part of the tracheobronchial tree. Nonspecific symptoms may lead to delayed diagnosis that requires more aggressive surgical treatment. An analysis of cases collected by the International Network of Pediatric Airway Team was undertaken to ensure proper insight into the behavior and management of PTTs. METHODS Patients <18 years of age with a histological confirmation of PTT diagnosed from 2000 to 2015 were included in this multicenter international retrospective study. Medical records, treatment modalities, and outcomes were analyzed. The patient presentation, tumor management, and clinical course were compared between malignant and benign histotypes. Clinical and surgical variables that might influence event-free survival were considered. RESULTS Among the 78 children identified, PTTs were more likely to be malignant than benign; bronchial carcinoid tumor (n = 31; 40%) was the most common histological subtype, followed by inflammatory myofibroblastic tumor (n = 19; 25%) and mucoepidermoid carcinoma (n = 15; 19%). Regarding symptoms at presentation, wheezing (P = 0.001) and dyspnea (P = 0.03) were more often associated with benign growth, whereas hemoptysis was more frequently associated with malignancy (P = 0.042). Factors that significantly worsened event-free survival were age at diagnosis earlier than 112 months (P = 0.0035) and duration of symptoms lasting more than 2 months (P = 0.0029). CONCLUSION The results of this international study provide important information regarding the clinical presentation, diagnostic workup, and treatment of PTTs in children, casting new light on the biological behavior of PTTs to ensure appropriate treatments. LEVEL OF EVIDENCE NA Laryngoscope, 130:E243-E251, 2020.
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Affiliation(s)
- Luca Pio
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cité, Paris, France
| | - Patricio Varela
- Department of Pediatric Surgery, Clinica Las Condes Medical Center, Hospital de Niños Calvo Mackenna, University of Chile, Santiago, Chile
| | - Martin J Eliott
- Tracheal Team, Great Ormond Street Children's Hospital, London, United Kingdom
| | - Vincent Couloigner
- Pediatric Ear, Nose, and Throat Department, Necker Hospital for Sick Children, Public Assistance-Hospitals of Paris, Paris, France
| | | | | | - Calogero Virgone
- Pediatric Surgery, Department of Women's and Children's Health, University of Padua, Padua, Italy
| | - Rebecca Maunsell
- Department of Otorhinolaryngology, Head and Neck Surgery, University of Campinas, Campinas, São Paulo, Brazil
| | - Victor Rachkov
- Chair of Pediatric Surgery of N.I. Pirogov Russian Research Medical University, Dmitry Rogachev Federal Research Center of Pediatric Hematology, Oncology and Immunology; ZAO European Medical Center, Moscow, Russia
| | - Michael J Rutter
- Division of Pediatric Otolaryngology-Head and Neck Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, U.S.A
| | - Mariano Boglione
- Department of General Surgery, Hospital de Pediatría "Prof. Dr. Juan P. Garrahan", Pichincha, 1850, Ciudad De Buenos Aires
| | - Jaime Penchyna Grub
- Department of Thoracic Surgery and Endoscopy, Hospital Infantil de México, Federico Gómez, Mexico
| | - Gastón Bellía Munzón
- Department of Surgery, Fundación Hospitalaria, Private Children's Hospital, Ciudad Autónoma de Buenos Aires, Buenos Aires, Argentina
| | - Sabine Sarnacki
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cité, Paris, France
| | - Sabine Irtan
- Department of Surgery, Hôpital Trousseau-Hôpitaux Universitaires de l'Est Parisien (AP-HP), Université Pierre et Marie Curie, Paris, France
| | - Claudia Schweiger
- Division of Pediatric Otolaryngology-Head and Neck Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, U.S.A
| | - Michèle Larroquet
- Department of Surgery, Hôpital Trousseau-Hôpitaux Universitaires de l'Est Parisien (AP-HP), Université Pierre et Marie Curie, Paris, France
| | - Naziha Khen Dunlop
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cité, Paris, France
| | - Madhavan Ramaswamy
- Tracheal Team, Great Ormond Street Children's Hospital, London, United Kingdom
| | | | - Giovanni Cecchetto
- Pediatric Surgery, Department of Women's and Children's Health, University of Padua, Padua, Italy
| | - Andrea Ferrari
- Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale Tumori, Milan, Italy
| | - Gianni Bisogno
- Pediatric Hematology and Oncology Division, University of Padua, Padua, Italy
| | - Michele Torre
- Airway Unit, IRCCS Istituto Giannina Gaslini, Genoa, Italy
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20
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Abstract
Bronchial carcinoid tumor, while rare, remains the most common primary malignant lung tumor in children. We present a retrospective analysis of 7 patients with typical bronchial carcinoid tumors diagnosed at 2 pediatric tertiary care referral centers between 1990 and 2014. The most common presenting symptom was pneumonia, followed by respiratory distress. Somatostatin scans were performed in selected patients. All patients had negative resection margin following surgery and were alive without disease at last follow-up. Typical carcinoid tumors have a good prognosis following definitive surgical resection. A review of published literature on pediatric bronchial carcinoid tumors is provided.
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21
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Szymanski LJ, Molas-Torreblanca K, Bawab R, Kim E, Don D, Mascarenhas L, Stanley P, Zhou S, Shillingford N. Bronchial Mucoepidermoid Carcinoma With the Classic MAML2 Gene Rearrangement in a 2-year-old Boy. Pediatr Dev Pathol 2018; 21:480-485. [PMID: 28492094 DOI: 10.1177/1093526617707855] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
Pulmonary mucoepidermoid carcinoma (PMEC) is rare. To date, primary PMEC has not been reported in a child younger than 3 years of age. We report a case of a 2-year-old boy who presented with 3 episodes of wheezing, cough, and fever over a period of 1 month. Radiologic findings were consistent with foreign body aspiration with consequent bronchial obstruction. Bronchoscopy was performed and attempts to retrieve the foreign body resulted in a biopsy of a fleshy lesion. By histology, the lesion was an epithelial neoplasm comprising cells arranged in a nested pattern. The neoplastic cells were round with round nuclei and amphophilic, vacuolated cytoplasm. Our diagnosis was low-grade salivary gland-type carcinoma of the bronchus. The pneumonectomy specimen showed a well-circumscribed, polypoid intrabronchial mass measuring 2.1 cm in greatest dimension. Histologic examination of the tumor showed an admixture of intermediate cells which were predominant, a small number of mucus cells and rare foci of squamous cells. The final diagnosis rendered was a low-grade mucoepidermoid carcinoma of the bronchus. Accurate diagnosis of PMEC can be challenging on limited biopsy material as seen in the case reported here. The use of molecular studies such as MAML2 gene rearrangement may facilitate diagnosis in difficult cases. Increased awareness of this entity and further molecular studies are needed for a better understanding of the pathogenesis of PMEC. To date, the reported age range for primary bronchial mucoepidermoid carcinoma is between 3 years and 78 years. This case represents the youngest patient reported in the English literature.
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Affiliation(s)
- Linda J Szymanski
- 1 Department of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Los Angeles, California
| | - Kira Molas-Torreblanca
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
- 3 Department of Hospital Medicine, Children's Hospital Los Angeles, Los Angeles, California
| | - Ramzi Bawab
- 1 Department of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Los Angeles, California
| | - Eugene Kim
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
- 4 General Pediatric Surgery, Children's Hospital Los Angeles, Los Angeles, California
| | - Debra Don
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
- 5 Division of Otolaryngology, Department of Surgery, Children's Hospital Los Angeles, Los Angeles, California
| | - Leo Mascarenhas
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
- 6 Division of Hematology-Oncology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, California
| | - Phillip Stanley
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
- 7 Department of Radiology, Children's Hospital Los Angeles, Los Angeles, California
| | - Shengmei Zhou
- 1 Department of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Los Angeles, California
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
| | - Nick Shillingford
- 1 Department of Pathology and Laboratory Medicine, Children's Hospital Los Angeles, Los Angeles, California
- 2 Keck School of Medicine, University of Southern California, Los Angeles, California
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22
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Lim YC, Tay SY. Case 4: Hemoptysis in a 17-year-old Chinese Boy. Pediatr Rev 2017; 38:570-571. [PMID: 29196515 DOI: 10.1542/pir.2016-0161] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/24/2022]
Affiliation(s)
- Yang-Chern Lim
- Department of Paediatric Medicine, National University Health System, Singapore
| | - Sok-Yan Tay
- Department of Paediatric Medicine, National University Health System, Singapore
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23
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Cho HJ, Yoon J, Lee E, Lee YS, Kim SY, Roh JL, Kim DK, Choi SH, Park SI, Kim YH, Koh KN, Im HJ, Seo JJ, Hong SJ, Yu J. The Different Clinical Aspects of Pediatric Primary Airway Tumors in the Larynx, Trachea, and Bronchi. J Korean Med Sci 2017; 32:1304-1311. [PMID: 28665067 PMCID: PMC5494330 DOI: 10.3346/jkms.2017.32.8.1304] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/28/2017] [Accepted: 05/18/2017] [Indexed: 01/31/2023] Open
Abstract
Primary airway tumors are rare in children and no literature reviewed their characteristics each location. We evaluate the clinical characteristics and outcomes of Korean children with primary airway tumors, from the larynx to bronchi. A retrospective chart review of children with primary tumors of the larynx, trachea, and bronchi at Asan Medical Center from January 2000 to July 2016 was conducted. Nineteen children were diagnosed with primary airway tumors of the larynx (47.4%), trachea (10.5%), and bronchi (42.1%). Median follow-up duration was 2.8 years and there were recurrences in 21.1%. Laryngeal tumors were associated with a younger median age at onset (2 months) and diagnosis (4 months), and most were relatively small (median size = 5.3 mm) and symptomatic. Tracheal and bronchial tumors were found in older children (age at onset and diagnosis > 11 years) and large (> 15.0 mm). Most (75%) patients with bronchial tumors were asymptomatic and all the patients with tracheal tumors were symptomatic. This study suggests that we should consider different the locations in primary airway tumor based on the age at onset and diagnosis, initial symptoms or signs, and size of tumor.
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Affiliation(s)
- Hyun Ju Cho
- Division of Pediatric Allergy and Respiratory, Department of Pediatrics, Childhood Asthma Atopy Center, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Jisun Yoon
- Division of Pediatric Allergy and Respiratory, Department of Pediatrics, Childhood Asthma Atopy Center, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Eun Lee
- Department of Pediatrics, Chonnam National University Hospital, Gwangju, Korea
| | - Yoon Se Lee
- Department of Otolaryngology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Sang Yoon Kim
- Department of Otolaryngology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Jong Lyel Roh
- Department of Otolaryngology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Dong Kwan Kim
- Department of Thoracic and Cardiovascular Surgery, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Se Hoon Choi
- Department of Thoracic and Cardiovascular Surgery, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Seung Il Park
- Department of Thoracic and Cardiovascular Surgery, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Yong Hee Kim
- Department of Thoracic and Cardiovascular Surgery, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Kyung Nam Koh
- Division of Pediatric Hematology and Oncology, Department of Pediatrics, University of Ulsan College of Medicine, Asan Medical Center Children's Hospital, Seoul, Korea
| | - Ho Joon Im
- Division of Pediatric Hematology and Oncology, Department of Pediatrics, University of Ulsan College of Medicine, Asan Medical Center Children's Hospital, Seoul, Korea
| | - Jong Jin Seo
- Division of Pediatric Hematology and Oncology, Department of Pediatrics, University of Ulsan College of Medicine, Asan Medical Center Children's Hospital, Seoul, Korea
| | - Soo Jong Hong
- Division of Pediatric Allergy and Respiratory, Department of Pediatrics, Childhood Asthma Atopy Center, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea
| | - Jinho Yu
- Division of Pediatric Allergy and Respiratory, Department of Pediatrics, Childhood Asthma Atopy Center, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.
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24
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25
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Abstract
Primary pulmonary tumors are rare in pediatrics. When they are encountered, they are usually carcinoid tumors or mucoepidermoid carcinomas. We present a patient who presented to both his primary care physician and the pediatric emergency department with recurrent bouts of wheezing and pneumonia, none of which ever completely resolved despite appropriate treatment. The patient had multiple chest films, which demonstrated the persistence of what appeared to be a right-sided infiltrate/atelectasis. Ultimately, the patient underwent a diagnostic workup that included a computed tomography scan and bronchoscopy. These studies revealed the presence of a bronchial mucoepidermoid carcinoma. The patient was successfully treated with photoablation of the lesion through the involvement of multiple subspecialists, including pediatric pulmonology, pediatric surgery, pediatric otolaryngology, and pediatric oncology. We discuss the incidence and epidemiology of pediatric bronchial tumors in general and mucoepidermoid carcinoma in particular as well as diagnosis, treatment options, and prognosis. Emergency physicians must maintain a high index of suspicion for alternate diagnoses in patients whose disease fails to respond to traditionally accepted therapy.
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26
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Liu L, Kong X, Lu X, Cao D. Pediatric Endobronchial Inflammatory Myofibroblastic Tumor: A Case Report and Review of the Literature. Clin Pract 2016; 6:853. [PMID: 28174621 PMCID: PMC5294928 DOI: 10.4081/cp.2016.853] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/17/2016] [Revised: 11/13/2016] [Accepted: 11/17/2016] [Indexed: 01/25/2023] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) belongs to the group of soft tissue tumor and could occur at any anatomical site from the central nervous system to gastrointestinal tract. The lung and abdomen are commonly affected sites, however, pulmonary IMT is predominantly located within the parenchyma rather than presenting as endobronchial lesion. IMTs may occur in any age group, but they are observed most commonly in children and adolescents. Here, we present a case of IMT arising from the left main stem bronchus in a 10-year-old girl.
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Affiliation(s)
- Leilei Liu
- Department of Radiology, The First Hospital of Jilin University , Changchun, China
| | - Xianglei Kong
- Department of Radiology, The First Hospital of Jilin University , Changchun, China
| | - Xiaoqian Lu
- Department of Radiology, The First Hospital of Jilin University , Changchun, China
| | - Dianbo Cao
- Department of Radiology, The First Hospital of Jilin University , Changchun, China
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27
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Varela P, Pio L, Brandigi E, Paraboschi I, Khen-Dunlop N, Hervieux E, Muller C, Mattioli G, Sarnacki S, Torre M. Tracheal and bronchial tumors. J Thorac Dis 2016; 8:3781-3786. [PMID: 28149577 PMCID: PMC5227203 DOI: 10.21037/jtd.2016.12.67] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/30/2016] [Accepted: 12/05/2016] [Indexed: 11/06/2022]
Abstract
Although primary tracheobronchial tumors are extremely rare in children, recurrent respiratory symptoms resistant to conventional therapy require further investigations to exclude possible malignant obstructive causes. As the matter of fact, early diagnosis may allow minimally invasive surgeries, improving the standard of living and the globally survival rate. The aim of this article is to provide an overview of diagnosis and management of tracheobronchial tumors in the early age, since only few reports are reported in the worldwide literature.
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Affiliation(s)
- Patricio Varela
- Department of Pediatric Surgery, Clinica Las Condes Medical Center, Hospital de Niños Calvo Mackenna, University of Chile, Santiago, Chile
| | - Luca Pio
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cite, Paris, France
| | - Elisa Brandigi
- Department of Medical Sciences, Surgery and Neurosciences, Clinic Pediatric Surgery, University of Siena, Siena, Italy
| | - Irene Paraboschi
- DINOGMI University of Genoa, Genoa, Italy
- Pediatric Surgery Unit, Istituto Giannina Gaslini, Genoa, Italy
| | - Nazhia Khen-Dunlop
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cite, Paris, France
| | - Erik Hervieux
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cite, Paris, France
| | - Cecile Muller
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cite, Paris, France
| | - Girolamo Mattioli
- DINOGMI University of Genoa, Genoa, Italy
- Pediatric Surgery Unit, Istituto Giannina Gaslini, Genoa, Italy
| | - Sabine Sarnacki
- Department of Pediatric Surgery, Necker Enfants-Malades Hospital-Université Paris Descartes, Sorbonne Paris Cite, Paris, France
| | - Michele Torre
- Pediatric Surgery Unit, Istituto Giannina Gaslini, Genoa, Italy
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28
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Abstract
Tumors of trachea and bronchi are uncommon and can occur in the form of benign or low- and high-grade malignant tumors. Although tracheobronchial tumors (TBTs) represent only 0.6% of all pulmonary tumors, they are clinically significant. Delays in diagnosis of these tumors commonly occur because the signs and symptoms caused by these tumors are nonspecific and chest radiographs are often considered unremarkable. Therefore, novel radiological techniques and better access to flexible bronchoscopy enable detection of larger number of TBT. The purpose of this article is to provide a review of tracheal and bronchial tumors and discuss significant aspects of the different TBT with focus on clinical manifestations and diagnostic procedures.
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Affiliation(s)
- Ruza Stevic
- Faculty of Medicine, University of Belgrade, Belgrade, Serbia;; Center for Radiology and MRI, Clinical Center of Serbia, Belgrade, Serbia
| | - Branislava Milenkovic
- Faculty of Medicine, University of Belgrade, Belgrade, Serbia;; Clinic for Pulmonology, Clinical Center of Serbia, Belgrade, Serbia
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29
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Pulmonary Mucoepidermoid Carcinoma and Solid Pseudopapillary Neoplasm of the Pancreas: A Coincidence of 2 Rare Tumors in an 8-Year-Old Boy. J Pediatr Hematol Oncol 2016; 38:649-652. [PMID: 27164522 DOI: 10.1097/mph.0000000000000572] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Primary pulmonary mucoepidermoid carcinoma (MEC) is extremely rare in children and is characterized by a mucus-secreting and squamous cell combination. Solid pseudopapillary neoplasm of the pancreas is a rare tumor of the pancreas, which is mostly seen in young women. We present the case of an 8-year-old boy previously diagnosed and treated by lung lobectomy for MEC. He was admitted to our hospital with an incidentally recognized mass at the pancreas during postoperative workout studies. Distal pancreatectomy was performed. Surgery was curative for both tumors. To the best of our knowledge, this is the first reported case of pseudopapillary neoplasm concomitant with MEC in the literature.
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Vageriya NL, Shah RS, Prabhu S, Naphade D, Athawale HR. Intra bronchial mucoepidermoid carcinoma in an 8 year old girl: A case report of rare tumor with review of literature. JOURNAL OF PEDIATRIC SURGERY CASE REPORTS 2016. [DOI: 10.1016/j.epsc.2016.07.002] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/01/2022] Open
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Irodi A, Leena RV, Prabhu SM, Gibikote S. Role of Computed Tomography in Pediatric Chest Conditions. Indian J Pediatr 2016; 83:675-90. [PMID: 26916888 DOI: 10.1007/s12098-015-1955-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/01/2015] [Accepted: 11/04/2015] [Indexed: 12/15/2022]
Abstract
CT is the preferred cross-sectional imaging modality for detailed evaluation of anatomy and pathology of the lung and tracheobronchial tree, and plays a complimentary role in the evaluation of certain chest wall, mediastinal, and cardiac abnormalities. The article provides an overview of indications and different types of CT chest, findings in common clinical conditions, and briefly touches upon the role of each team member in optimizing and thus reducing radiation dose.
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Affiliation(s)
- Aparna Irodi
- Department of Radiology, Christian Medical College, Vellore, 632004, Tamil Nadu, India
| | - R V Leena
- Department of Radiology, Christian Medical College, Vellore, 632004, Tamil Nadu, India
| | - Shailesh M Prabhu
- Department of Radiology, SSM Superspeciality Hospital, Hassan, Karnataka, India
| | - Sridhar Gibikote
- Department of Radiology, Christian Medical College, Vellore, 632004, Tamil Nadu, India.
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Abstract
Primary tracheobronchial tumors are rare lesions that can be benign or malignant, with different location along the airway tree. Symptoms may include wheezing, chronic pneumonia, asthma, chest pain, recurrent cough, atelectasis, haemoptysis, and weight loss. Due to the heterogeneity of symptoms, diagnosis can be difficult and the airway involvement can lead progressively to a bronchial or tracheal obstruction. Due to the rarity of primary tracheobronchial tumors in children, there are not any oncological guidelines on pre-operative work-up, treatment, and follow-up. Only few reports and multicentric studies are reported. In most cases, surgical resection seems to be the treatment of choice. Brachytherapy, endoscopic treatment, and chemotherapy are rarely described. In this article we present an overview on these rare tumors, including pathological aspects, clinical presentation, imaging assessment, and endoscopic or open surgical treatments. We discuss different surgical approaches, according with tumor location.
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Affiliation(s)
- Patricio Varela
- Department of Pediatric Surgery, Clinica Las Condes Medical Center, Hospital de Niños Calvo Mackenna, University of Chile, Camino el tucuquere 4080, Santiago, Chile.
| | - Luca Pio
- DINOGMI, University of Genoa, Genova, Italy; Airway Team and Pediatric Surgery Unit, Istituto Giannina Gaslini, Genova, Italy
| | - Michele Torre
- Airway Team and Pediatric Surgery Unit, Istituto Giannina Gaslini, Genova, Italy
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du Toit-Prinsloo L, Bunn BK. Massive hemoptysis due to primary mucoepidermoid carcinoma of the lung in a 12-year-old. Forensic Sci Med Pathol 2016; 12:380-3. [PMID: 27216748 DOI: 10.1007/s12024-016-9782-0] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/25/2016] [Indexed: 11/30/2022]
Affiliation(s)
- Lorraine du Toit-Prinsloo
- Department of Forensic Medicine, University of Pretoria, Private Bag X323, Arcadia, 0007, South Africa.
| | - Belinda K Bunn
- Department of Oral Pathology and Oral Biology, School of Dentistry, University of Pretoria, Private Bag X323, Arcadia, 0007, South Africa
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Jaramillo S, Rojas Y, Slater BJ, Baker ML, Hicks MJ, Muscal JA, Vece TJ, Wesson DE, Nuchtern JG. Childhood and adolescent tracheobronchial mucoepidermoid carcinoma (MEC): a case-series and review of the literature. Pediatr Surg Int 2016; 32:417-24. [PMID: 26790674 DOI: 10.1007/s00383-015-3849-y] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 01/20/2014] [Indexed: 01/30/2023]
Abstract
Tracheobronchial mucoepidermoid carcinomas (MEC) are rare in the pediatric population with literature limited primarily to case reports. Here we present our institutional experience treating MEC in three patients and review the literature of 142 pediatric cases previously published from 1968 to 2013. Although rare, tracheobronchial MEC should be included in the differential diagnosis in a child with recurrent respiratory symptoms. Conservative surgical management is often sufficient to achieve complete resection and good outcomes.
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Affiliation(s)
- Sergio Jaramillo
- Department of Radiation Oncology, Baylor College of Medicine, Houston, TX, USA
| | - Yesenia Rojas
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, 6701 Fannin, Suite 1210, Houston, 77030, TX, USA
| | - Bethany J Slater
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, 6701 Fannin, Suite 1210, Houston, 77030, TX, USA
| | - Michael L Baker
- Department of Pathology and Immunology, Baylor College of Medicine, Houston, TX, USA
| | - M John Hicks
- Department of Pathology and Immunology, Baylor College of Medicine, Houston, TX, USA
| | - Jodi A Muscal
- Division of Hematology-Oncology, Department of Pediatrics, Baylor College of Medicine, Houston, TX, USA
| | - Timothy J Vece
- Division of Pulmonary Medicine, Department of Pediatrics, Baylor College of Medicine, Houston, TX, USA
| | - David E Wesson
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, 6701 Fannin, Suite 1210, Houston, 77030, TX, USA
| | - Jed G Nuchtern
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, 6701 Fannin, Suite 1210, Houston, 77030, TX, USA.
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Inflammatory myofibroblastic tumor of the trachea in the pediatric age group: case report and systematic review of the literature. J Bronchology Interv Pulmonol 2015; 22:58-65. [PMID: 25590486 DOI: 10.1097/lbr.0000000000000105] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Inflammatory myofibroblastic tumors are uncommon tumors, which present as solitary masses in the pulmonary parenchyma. Tracheal involvement by these tumors is extremely rare and can be misdiagnosed as asthma. The closest histologic differential diagnoses are IgG4-related sclerosing pseudotumors, which are differentiated by IgG4 positivity. Fifty percent of inflammatory myofibroblastic tumors are positive for anaplastic lymphoma kinase gene rearrangements. The treatment modality of choice is surgical resection with therapeutic bronchoscopy reserved for patients presenting with acute airway obstruction or in those unwilling for surgery. New and upcoming treatments include anaplastic lymphoma kinase inhibitors like crizotinib.
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Rojas Y, Shi YX, Zhang W, Beierle EA, Doski JJ, Goldfarb M, Goldin AB, Gow KW, Langer M, Vasudevan SA, Nuchtern JG. Primary malignant pulmonary tumors in children: a review of the national cancer data base. J Pediatr Surg 2015; 50:1004-8. [PMID: 25812444 DOI: 10.1016/j.jpedsurg.2015.03.032] [Citation(s) in RCA: 44] [Impact Index Per Article: 4.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/26/2015] [Accepted: 03/10/2015] [Indexed: 11/18/2022]
Abstract
BACKGROUND The purpose is to delineate the clinical and pathological characteristics of rare primary malignant pulmonary tumors in children. METHODS Utilizing the National Cancer Data Base (NCDB), we analyzed all children (≤ 18 years) with a primary malignant pulmonary tumor from 1998 to 2011 to identify factors associated with better survival. RESULTS Of 211 children identified, the most common histology was carcinoid tumor (n=133, 63%) followed by mucoepidermoid carcinoma (MEC) (n=37, 18%), squamous cell carcinoma (SCC) (n=19, 9%), adenocarcinoma (n=16, 8%), bronchoalveolar carcinoma (BAC) (n=4, 2%), and small cell carcinoma (SCLC) (n=2, <1%). Factors that significantly affected survival include histology, race, tumor size, lymph node status, and extent of surgery. Patients with MEC and carcinoid tumors had a better overall survival compared to patients with other histologies (p<0.0001). The 5-year overall survival for MEC and carcinoid tumors was 100% and 95% (95% CI 87-98), respectively, versus 50% (95%CI 1-91) for BAC, 28% (95%CI 9-52) for SCC, and 26% (95%CI 5-55) for adenocarcinoma. CONCLUSION The majority of pediatric patients with a primary malignant pulmonary tumor present with carcinoid tumor or MEC and have an excellent prognosis. Lung cancers which are common in adults, but rare in children, have a worse prognosis.
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Affiliation(s)
- Yesenia Rojas
- Michael E. DeBakey Department of Surgery, Baylor College of Medicine, and Department of Surgery, Texas Children's Hospital, Houston, TX
| | - Yan X Shi
- Michael E. DeBakey Department of Surgery, Baylor College of Medicine, and Department of Surgery, Texas Children's Hospital, Houston, TX
| | - Wei Zhang
- Outcomes and Impact Service, Department of Surgery, Texas Children's Hospital, Houston, TX
| | | | - John J Doski
- Department of Surgery, Methodist Children's Hospital of South Texas, University of Texas Health Science Center - San Antonio, San Antonio, TX
| | - Melanie Goldfarb
- Department of Surgery, John Wayne Cancer Institute/Providence St. Johns Medical Center, Santa Monica, CA
| | - Adam B Goldin
- Department of Surgery, Seattle Children's Hospital, University of Washington, Seattle, WA
| | - Kenneth W Gow
- Department of Surgery, Seattle Children's Hospital, University of Washington, Seattle, WA
| | - Monica Langer
- Department of Surgery, Maine Children's Cancer Program, Tufts University, Portland, ME
| | - Sanjeev A Vasudevan
- Michael E. DeBakey Department of Surgery, Baylor College of Medicine, and Department of Surgery, Texas Children's Hospital, Houston, TX
| | - Jed G Nuchtern
- Michael E. DeBakey Department of Surgery, Baylor College of Medicine, and Department of Surgery, Texas Children's Hospital, Houston, TX.
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Madafferi S, Catania VD, Accinni A, Boldrini R, Inserra A. Endobronchial tumor in children: Unusual finding in recurrent pneumonia, report of three cases. World J Clin Pediatr 2015; 4:30-34. [PMID: 26015878 PMCID: PMC4438439 DOI: 10.5409/wjcp.v4.i2.30] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/25/2014] [Revised: 01/27/2015] [Accepted: 04/02/2015] [Indexed: 02/05/2023] Open
Abstract
We are reporting 3 cases of pediatric endobronchial tumors presented with recurrent pneumonia. The median age of patients, at time of presentation, was 10.6 years. All patients presented with recurrent pneumonia with a mean time to occurrence, after onset of symptoms, of 14 mo. Bronchoscopy was early performed as part of diagnostic work-up and it revealed an endobronchial mass in every case. Complete surgical resection was performed in all cases, with lung preservation in two of them. Neither post-operative chemotherapy nor radiotherapy was required. The mean duration of follow-up was 7 years and all patients are still alive and disease-free. Recurrent pneumonia, in pediatrics, should raise the suspicion of an obstructing lesion, congenital malformation or systemic disease. A systematic approach is useful for organize the clinicians initial workup. Prompt diagnosis allows parenchymal-sparing surgery, which offers the best chance of cure and reduces clinical and functional complications in these patients.
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A Case of Chest Pain and Hemoptysis in a Young Child. J Bronchology Interv Pulmonol 2015; 22:152-7. [DOI: 10.1097/lbr.0000000000000130] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/23/2022]
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Malkan AD, Sandoval JA. Controversial tumors in pediatric surgical oncology. Curr Probl Surg 2014; 51:478-520. [PMID: 25524425 DOI: 10.1067/j.cpsurg.2014.11.004] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/03/2014] [Accepted: 11/17/2014] [Indexed: 12/13/2022]
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Fujino H, Park YD, Uemura S, Tanaka S, Kawabe M, Maeda S, Kato I, Watanabe KI, Umeda K, Hiramatsu H, Adachi S, Sato T, Date H, Haga H, Sumimoto S. An endobronchial inflammatory myofibroblastic tumor in a 10-yr-old child after allogeneic hematopoietic cell transplantation. Pediatr Transplant 2014; 18:E165-8. [PMID: 24814936 DOI: 10.1111/petr.12275] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 03/07/2014] [Indexed: 12/24/2022]
Abstract
IMTs belong to the group of soft tissue tumor and could occur at any anatomical site; however, the causes and growth feature remain unclear. This case report documents a 10-yr-old male suffering from slowly developing dyspnea on exertion and cough around seven months post-HCT. He was diagnosed with an endobronchial tumor based on imaging, and histology confirmed ALK-positive submucosal spindle-shaped cells with infiltrative cells, compatible with IMT. We should be aware that IMT is a potential complication of pediatric allogeneic HCT and can cause sudden airway obstruction.
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Affiliation(s)
- Hisanori Fujino
- Department of Pediatrics, Osaka Red Cross Hospital, Osaka, Japan
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Eyssartier E, Ang P, Bonnemaison E, Gibertini I, Diot P, Carpentier E, Chantepie A, Leclair MD, Brouard J, Boutard P, Deneuville E, Marie-Cardine A, Lardy H. Characteristics of endobronchial primitive tumors in children. Pediatr Pulmonol 2014; 49:E121-5. [PMID: 24532419 DOI: 10.1002/ppul.22987] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/26/2013] [Accepted: 11/30/2013] [Indexed: 11/05/2022]
Abstract
Primary endobronchial tumors are rare in children and they include a broad spectrum of lesions. The aim of this study was to determine the characteristic features, treatments and outcomes of these tumors. We report a retrospective analysis of all patients treated for endobronchial tumor in nine French hospitals between 1990 and 2010 and a comparison of the results with those reported in the medical literature. Twelve tumors were reported: five low grade muco epidermoid carcinomas, two inflammatory myofibroblastic tumors, two hemangiomas, one anaplastic large cell lymphoma, one carcinoid tumor, and one juvenile xanthogranuloma. The mean age of the patients was 7.5 ± 3.5 years. The most common sign revealing the disease was persistent atelectasis or recurrent pneumonia (eight cases). The other revealing signs were a persistent bronchospasm (three cases) and hemoptysis (one case). The clinical presentation, biology, serum tumor markers, and chest X-ray abnormalities were not specific to a particular histological diagnosis. Chest CT scan revealed the presence of an endobronchial tumor in 11 cases. Nine tumors could be diagnosed from a biopsy obtained by video endoscopy. Complete surgical resection was performed in seven patients. Bronchoscopic removal was performed in five cases and was successful in three. There were no deaths. Endobronchial tumors are rare in childhood and their histology is diverse. Chest CT scan and per-endoscopic endobronchial biopsies are required for diagnosis, when possible. Surgical or endoscopic treatment should be discussed by a multidisciplinary team. Despite the multiple etiologies, the prognosis of these tumors is good if diagnosis is early and if resection is complete. Long-term recurrences have been described, so long-term follow-up of these children is recommended.
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Affiliation(s)
- E Eyssartier
- Service de Chirurgie Pédiatrique Viscérale, Urologique et Plastique, Brûlés, Hôpital Gatien de Clocheville, CHRU de Tours, Tours, France
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Abstract
Isolated endobronchial inflammatory myofibroblastic tumor is an unusual diagnosis among endobronchial masses in childhood. The presenting signs and symptoms may mimic asthma. Rigid bronchoscopy is effective for the diagnosis and treatment. Follow-up is mandatory to check for recurrent disease. Here in, the authors report on a 9-year-old girl with endobronchial inflammatory myofibroblastic tumor to emphasize the possibility of endobronchial lesion in children with longstanding obstructive symptoms.
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Khawaja A, Maheshwari PK, Haque A, Zubairi ABS. A rare aetiology of respiratory failure in a 10-year-old boy: inflammatory myofibroblastic tumour. BMJ Case Rep 2013; 2013:bcr-2013-201102. [PMID: 24248314 DOI: 10.1136/bcr-2013-201102] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Primary neoplasms of the respiratory tract are rarely encountered in the paediatric population. Inflammatory myofibroblastic tumour (IMT) is a rare soft tissue mesenchymal tumour but a distinct disease entity accounting for less than 1% of all primary lung tumours. We report a case of a 10-year-old boy who presented with respiratory failure and left lung collapse. On flexible fiberoptic bronchoscopy, a pedunculated mass in the lower part of the trachea originating from the left main stem bronchus was identified. The patient subsequently underwent a left-sided pneumonectomy with complete resection of the mass. The histopathological analysis was consistent with IMT. Two years of follow-up and the patient remains well.
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Affiliation(s)
- Ali Khawaja
- Medical College, The Aga Khan University Hospital, Karachi, Sindh, Pakistan
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Amini B, Huang SY, Tsai J, Benveniste MF, Robledo HH, Lee EY. Primary lung and large airway neoplasms in children: current imaging evaluation with multidetector computed tomography. Radiol Clin North Am 2013; 51:637-57. [PMID: 23830790 DOI: 10.1016/j.rcl.2013.04.005] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/09/2023]
Abstract
Multidetector computed tomography (MDCT) offers an important noninvasive imaging modality for confirmation and further characterization of primary lung and large airway neoplasms encountered in pediatric patients. Children represent a unique challenge in imaging, not only because of unique patient factors (eg, inability to follow instructions, motion, need for sedation) but because of the technical factors that must be optimized to reduce radiation dose. This article reviews an MDCT imaging algorithm, up-to-date imaging techniques, and clinical applications of MDCT for evaluating benign and malignant primary neoplasms of lung and large airway in infants and children.
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Affiliation(s)
- Behrang Amini
- Department of Diagnostic Radiology, The University of Texas MD Anderson Cancer Center, 1515 Holcombe Blvd, Houston, TX 77030, USA
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Yu YF, Ling HY, Xiao GS, Sun P, Li MY, Wu WY. Misdiagnosed rare subglottic lesions with bronchial asthma as the initial symptom. Am J Emerg Med 2013; 31:1295.e1-4. [PMID: 23702064 DOI: 10.1016/j.ajem.2013.04.023] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2013] [Accepted: 04/20/2013] [Indexed: 10/26/2022] Open
Affiliation(s)
- Ya-Feng Yu
- Department of Otolaryngology, the First Affiliated Hospital of Soochow University, Suzhou, China
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Kobayashi T, Hyodo M, Honda N. Primary endobronchial Burkitt's lymphoma in a child: a case report. Int J Pediatr Otorhinolaryngol 2013; 77:875-8. [PMID: 23548894 DOI: 10.1016/j.ijporl.2013.03.012] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/13/2012] [Revised: 03/01/2013] [Accepted: 03/07/2013] [Indexed: 10/27/2022]
Abstract
Primary endobronchial tumors are rare in children and often are misdiagnosed, resulting in a delay of appropriate treatment. Here, we present a case of 4-year-old girl with primary endobronchial lymphoma who presented with persistent pneumonia despite medical treatment. Her chest radiograph showed left atelectasis, and a bronchial foreign body was initially suspected. Bronchoscopic examination demonstrated a granulomatous tumor occupying the left main bronchus. Localized Burkitt's lymphoma in the left trachea was diagnosed histopathologically. This case emphasizes the consideration of endobronchial tumor and bronchoscopy in children who show persistent atelectasis or recurrent pneumonia.
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Affiliation(s)
- Taisuke Kobayashi
- Department of Otolaryngology, Kochi Medical School, Nankoku, Kochi, Japan.
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Hayakawa A, Hirase S, Matsunoshita N, Yamamoto N, Kubokawa I, Mori T, Yanai T, Maniwa Y, Iijima K. Primary pediatric endobronchial Ewing sarcoma family of tumors. AMERICAN JOURNAL OF CASE REPORTS 2013; 14:67-9. [PMID: 23569566 PMCID: PMC3619045 DOI: 10.12659/ajcr.883821] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/04/2012] [Accepted: 02/25/2013] [Indexed: 12/01/2022]
Abstract
Background: Ewing sarcoma family of tumors is the second most common primary bone tumor of childhood. Extraosseous Ewing sarcoma family of tumors is rare. We present a pediatric case of primary endobronchial Ewing sarcoma family of tumors. Case Report: A 12-year-old boy presented with dyspnea and chest radiography showed right pulmonary atelectasis. Chest computed tomography demonstrated tumor in the right main bronchus. Histopathological examination of the resected tumor demonstrated Ewing sarcoma family of tumors. No other lesions were detected throughout the body and the right main bronchus was thought to be the primary site. As of 1 year and 6 months after further resection of residual tumor followed by chemotherapy and radiotherapy, the patient remains disease-free. Conclusions: Extraosseous Ewing sarcoma family of tumors arises in soft tissues of the trunk or extremities, but primary endobronchial Ewing sarcoma family of tumors has rarely been reported. Although quite rare, Ewing sarcoma family of tumors should be considered among the differential diagnoses for pediatric bronchial tumor.
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Affiliation(s)
- Akira Hayakawa
- Department of Pediatrics, Kobe University Graduate School of Medicine, Kobe, Japan
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48
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A case of tracheal hamartoma resected with loop electrocautery. Case Rep Pulmonol 2013; 2013:568590. [PMID: 23401830 PMCID: PMC3557623 DOI: 10.1155/2013/568590] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/12/2012] [Accepted: 12/19/2012] [Indexed: 11/17/2022] Open
Abstract
The authors report on the case of a 67-year-old man with longstanding breathlessness, which was eventually attributed to a fixed mass in the upper third of the trachea causing upper airway obstruction. The lesion was amenable to loop electrocautery resection via flexible bronchoscopy that led to prompt resolution of patient symptoms. Biopsy was consistent with tracheal hamartoma, an exceedingly rare benign tracheal tumor. All the cases of tracheal hamartomas in the literature to date, the application of electrocautery and other methods of interventional bronchoscopy for resection of selected tracheal tumors are discussed.
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49
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Denne C, von Lüttichau I, Steinborn M, Schenk D, Wawer A, Sell R, Schießl J, Grübl A, Burdach S. Differenzialdiagnose therapierefraktäres Asthma. Monatsschr Kinderheilkd 2012. [DOI: 10.1007/s00112-012-2772-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/27/2022]
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