Journal of Clinical and Diagnostic Research, ISSN - 0973 - 709X

Users Online : 32588

AbstractMaterial and MethodsCase ReportResultsDiscussionConclusionAcknowledgementReferencesDOI and Others
Article in PDF How to Cite Citation Manager Readers' Comments (0) Audio Visual Article Statistics Link to PUBMED Print this Article Send to a Friend
Advertisers Access Statistics Resources

Dr Mohan Z Mani

"Thank you very much for having published my article in record time.I would like to compliment you and your entire staff for your promptness, courtesy, and willingness to be customer friendly, which is quite unusual.I was given your reference by a colleague in pathology,and was able to directly phone your editorial office for clarifications.I would particularly like to thank the publication managers and the Assistant Editor who were following up my article. I would also like to thank you for adjusting the money I paid initially into payment for my modified article,and refunding the balance.
I wish all success to your journal and look forward to sending you any suitable similar article in future"



Dr Mohan Z Mani,
Professor & Head,
Department of Dermatolgy,
Believers Church Medical College,
Thiruvalla, Kerala
On Sep 2018




Prof. Somashekhar Nimbalkar

"Over the last few years, we have published our research regularly in Journal of Clinical and Diagnostic Research. Having published in more than 20 high impact journals over the last five years including several high impact ones and reviewing articles for even more journals across my fields of interest, we value our published work in JCDR for their high standards in publishing scientific articles. The ease of submission, the rapid reviews in under a month, the high quality of their reviewers and keen attention to the final process of proofs and publication, ensure that there are no mistakes in the final article. We have been asked clarifications on several occasions and have been happy to provide them and it exemplifies the commitment to quality of the team at JCDR."



Prof. Somashekhar Nimbalkar
Head, Department of Pediatrics, Pramukhswami Medical College, Karamsad
Chairman, Research Group, Charutar Arogya Mandal, Karamsad
National Joint Coordinator - Advanced IAP NNF NRP Program
Ex-Member, Governing Body, National Neonatology Forum, New Delhi
Ex-President - National Neonatology Forum Gujarat State Chapter
Department of Pediatrics, Pramukhswami Medical College, Karamsad, Anand, Gujarat.
On Sep 2018




Dr. Kalyani R

"Journal of Clinical and Diagnostic Research is at present a well-known Indian originated scientific journal which started with a humble beginning. I have been associated with this journal since many years. I appreciate the Editor, Dr. Hemant Jain, for his constant effort in bringing up this journal to the present status right from the scratch. The journal is multidisciplinary. It encourages in publishing the scientific articles from postgraduates and also the beginners who start their career. At the same time the journal also caters for the high quality articles from specialty and super-specialty researchers. Hence it provides a platform for the scientist and researchers to publish. The other aspect of it is, the readers get the information regarding the most recent developments in science which can be used for teaching, research, treating patients and to some extent take preventive measures against certain diseases. The journal is contributing immensely to the society at national and international level."



Dr Kalyani R
Professor and Head
Department of Pathology
Sri Devaraj Urs Medical College
Sri Devaraj Urs Academy of Higher Education and Research , Kolar, Karnataka
On Sep 2018




Dr. Saumya Navit

"As a peer-reviewed journal, the Journal of Clinical and Diagnostic Research provides an opportunity to researchers, scientists and budding professionals to explore the developments in the field of medicine and dentistry and their varied specialities, thus extending our view on biological diversities of living species in relation to medicine.
‘Knowledge is treasure of a wise man.’ The free access of this journal provides an immense scope of learning for the both the old and the young in field of medicine and dentistry as well. The multidisciplinary nature of the journal makes it a better platform to absorb all that is being researched and developed. The publication process is systematic and professional. Online submission, publication and peer reviewing makes it a user-friendly journal.
As an experienced dentist and an academician, I proudly recommend this journal to the dental fraternity as a good quality open access platform for rapid communication of their cutting-edge research progress and discovery.
I wish JCDR a great success and I hope that journal will soar higher with the passing time."



Dr Saumya Navit
Professor and Head
Department of Pediatric Dentistry
Saraswati Dental College
Lucknow
On Sep 2018




Dr. Arunava Biswas

"My sincere attachment with JCDR as an author as well as reviewer is a learning experience . Their systematic approach in publication of article in various categories is really praiseworthy.
Their prompt and timely response to review's query and the manner in which they have set the reviewing process helps in extracting the best possible scientific writings for publication.
It's a honour and pride to be a part of the JCDR team. My very best wishes to JCDR and hope it will sparkle up above the sky as a high indexed journal in near future."



Dr. Arunava Biswas
MD, DM (Clinical Pharmacology)
Assistant Professor
Department of Pharmacology
Calcutta National Medical College & Hospital , Kolkata




Dr. C.S. Ramesh Babu
" Journal of Clinical and Diagnostic Research (JCDR) is a multi-specialty medical and dental journal publishing high quality research articles in almost all branches of medicine. The quality of printing of figures and tables is excellent and comparable to any International journal. An added advantage is nominal publication charges and monthly issue of the journal and more chances of an article being accepted for publication. Moreover being a multi-specialty journal an article concerning a particular specialty has a wider reach of readers of other related specialties also. As an author and reviewer for several years I find this Journal most suitable and highly recommend this Journal."
Best regards,
C.S. Ramesh Babu,
Associate Professor of Anatomy,
Muzaffarnagar Medical College,
Muzaffarnagar.
On Aug 2018




Dr. Arundhathi. S
"Journal of Clinical and Diagnostic Research (JCDR) is a reputed peer reviewed journal and is constantly involved in publishing high quality research articles related to medicine. Its been a great pleasure to be associated with this esteemed journal as a reviewer and as an author for a couple of years. The editorial board consists of many dedicated and reputed experts as its members and they are doing an appreciable work in guiding budding researchers. JCDR is doing a commendable job in scientific research by promoting excellent quality research & review articles and case reports & series. The reviewers provide appropriate suggestions that improve the quality of articles. I strongly recommend my fraternity to encourage JCDR by contributing their valuable research work in this widely accepted, user friendly journal. I hope my collaboration with JCDR will continue for a long time".



Dr. Arundhathi. S
MBBS, MD (Pathology),
Sanjay Gandhi institute of trauma and orthopedics,
Bengaluru.
On Aug 2018




Dr. Mamta Gupta,
"It gives me great pleasure to be associated with JCDR, since last 2-3 years. Since then I have authored, co-authored and reviewed about 25 articles in JCDR. I thank JCDR for giving me an opportunity to improve my own skills as an author and a reviewer.
It 's a multispecialty journal, publishing high quality articles. It gives a platform to the authors to publish their research work which can be available for everyone across the globe to read. The best thing about JCDR is that the full articles of all medical specialties are available as pdf/html for reading free of cost or without institutional subscription, which is not there for other journals. For those who have problem in writing manuscript or do statistical work, JCDR comes for their rescue.
The journal has a monthly publication and the articles are published quite fast. In time compared to other journals. The on-line first publication is also a great advantage and facility to review one's own articles before going to print. The response to any query and permission if required, is quite fast; this is quite commendable. I have a very good experience about seeking quick permission for quoting a photograph (Fig.) from a JCDR article for my chapter authored in an E book. I never thought it would be so easy. No hassles.
Reviewing articles is no less a pain staking process and requires in depth perception, knowledge about the topic for review. It requires time and concentration, yet I enjoy doing it. The JCDR website especially for the reviewers is quite user friendly. My suggestions for improving the journal is, more strict review process, so that only high quality articles are published. I find a a good number of articles in Obst. Gynae, hence, a new journal for this specialty titled JCDR-OG can be started. May be a bimonthly or quarterly publication to begin with. Only selected articles should find a place in it.
An yearly reward for the best article authored can also incentivize the authors. Though the process of finding the best article will be not be very easy. I do not know how reviewing process can be improved. If an article is being reviewed by two reviewers, then opinion of one can be communicated to the other or the final opinion of the editor can be communicated to the reviewer if requested for. This will help one’s reviewing skills.
My best wishes to Dr. Hemant Jain and all the editorial staff of JCDR for their untiring efforts to bring out this journal. I strongly recommend medical fraternity to publish their valuable research work in this esteemed journal, JCDR".



Dr. Mamta Gupta
Consultant
(Ex HOD Obs &Gynae, Hindu Rao Hospital and associated NDMC Medical College, Delhi)
Aug 2018




Dr. Rajendra Kumar Ghritlaharey

"I wish to thank Dr. Hemant Jain, Editor-in-Chief Journal of Clinical and Diagnostic Research (JCDR), for asking me to write up few words.
Writing is the representation of language in a textual medium i e; into the words and sentences on paper. Quality medical manuscript writing in particular, demands not only a high-quality research, but also requires accurate and concise communication of findings and conclusions, with adherence to particular journal guidelines. In medical field whether working in teaching, private, or in corporate institution, everyone wants to excel in his / her own field and get recognised by making manuscripts publication.


Authors are the souls of any journal, and deserve much respect. To publish a journal manuscripts are needed from authors. Authors have a great responsibility for producing facts of their work in terms of number and results truthfully and an individual honesty is expected from authors in this regards. Both ways its true "No authors-No manuscripts-No journals" and "No journals–No manuscripts–No authors". Reviewing a manuscript is also a very responsible and important task of any peer-reviewed journal and to be taken seriously. It needs knowledge on the subject, sincerity, honesty and determination. Although the process of reviewing a manuscript is a time consuming task butit is expected to give one's best remarks within the time frame of the journal.
Salient features of the JCDR: It is a biomedical, multidisciplinary (including all medical and dental specialities), e-journal, with wide scope and extensive author support. At the same time, a free text of manuscript is available in HTML and PDF format. There is fast growing authorship and readership with JCDR as this can be judged by the number of articles published in it i e; in Feb 2007 of its first issue, it contained 5 articles only, and now in its recent volume published in April 2011, it contained 67 manuscripts. This e-journal is fulfilling the commitments and objectives sincerely, (as stated by Editor-in-chief in his preface to first edition) i e; to encourage physicians through the internet, especially from the developing countries who witness a spectrum of disease and acquire a wealth of knowledge to publish their experiences to benefit the medical community in patients care. I also feel that many of us have work of substance, newer ideas, adequate clinical materials but poor in medical writing and hesitation to submit the work and need help. JCDR provides authors help in this regards.
Timely publication of journal: Publication of manuscripts and bringing out the issue in time is one of the positive aspects of JCDR and is possible with strong support team in terms of peer reviewers, proof reading, language check, computer operators, etc. This is one of the great reasons for authors to submit their work with JCDR. Another best part of JCDR is "Online first Publications" facilities available for the authors. This facility not only provides the prompt publications of the manuscripts but at the same time also early availability of the manuscripts for the readers.
Indexation and online availability: Indexation transforms the journal in some sense from its local ownership to the worldwide professional community and to the public.JCDR is indexed with Embase & EMbiology, Google Scholar, Index Copernicus, Chemical Abstracts Service, Journal seek Database, Indian Science Abstracts, to name few of them. Manuscriptspublished in JCDR are available on major search engines ie; google, yahoo, msn.
In the era of fast growing newer technologies, and in computer and internet friendly environment the manuscripts preparation, submission, review, revision, etc and all can be done and checked with a click from all corer of the world, at any time. Of course there is always a scope for improvement in every field and none is perfect. To progress, one needs to identify the areas of one's weakness and to strengthen them.
It is well said that "happy beginning is half done" and it fits perfectly with JCDR. It has grown considerably and I feel it has already grown up from its infancy to adolescence, achieving the status of standard online e-journal form Indian continent since its inception in Feb 2007. This had been made possible due to the efforts and the hard work put in it. The way the JCDR is improving with every new volume, with good quality original manuscripts, makes it a quality journal for readers. I must thank and congratulate Dr Hemant Jain, Editor-in-Chief JCDR and his team for their sincere efforts, dedication, and determination for making JCDR a fast growing journal.
Every one of us: authors, reviewers, editors, and publisher are responsible for enhancing the stature of the journal. I wish for a great success for JCDR."



Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
Department of Paediatric Surgery, Gandhi Medical College & Associated
Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

"On looking back through my Gmail archives after being requested by the journal to write a short editorial about my experiences of publishing with the Journal of Clinical and Diagnostic Research (JCDR), I came across an e-mail from Dr. Hemant Jain, Editor, in March 2007, which introduced the new electronic journal. The main features of the journal which were outlined in the e-mail were extensive author support, cash rewards, the peer review process, and other salient features of the journal.
Over a span of over four years, we (I and my colleagues) have published around 25 articles in the journal. In this editorial, I plan to briefly discuss my experiences of publishing with JCDR and the strengths of the journal and to finally address the areas for improvement.
My experiences of publishing with JCDR: Overall, my experiences of publishing withJCDR have been positive. The best point about the journal is that it responds to queries from the author. This may seem to be simple and not too much to ask for, but unfortunately, many journals in the subcontinent and from many developing countries do not respond or they respond with a long delay to the queries from the authors 1. The reasons could be many, including lack of optimal secretarial and other support. Another problem with many journals is the slowness of the review process. Editorial processing and peer review can take anywhere between a year to two years with some journals. Also, some journals do not keep the contributors informed about the progress of the review process. Due to the long review process, the articles can lose their relevance and topicality. A major benefit with JCDR is the timeliness and promptness of its response. In Dr Jain's e-mail which was sent to me in 2007, before the introduction of the Pre-publishing system, he had stated that he had received my submission and that he would get back to me within seven days and he did!
Most of the manuscripts are published within 3 to 4 months of their submission if they are found to be suitable after the review process. JCDR is published bimonthly and the accepted articles were usually published in the next issue. Recently, due to the increased volume of the submissions, the review process has become slower and it ?? Section can take from 4 to 6 months for the articles to be reviewed. The journal has an extensive author support system and it has recently introduced a paid expedited review process. The journal also mentions the average time for processing the manuscript under different submission systems - regular submission and expedited review.
Strengths of the journal: The journal has an online first facility in which the accepted manuscripts may be published on the website before being included in a regular issue of the journal. This cuts down the time between their acceptance and the publication. The journal is indexed in many databases, though not in PubMed. The editorial board should now take steps to index the journal in PubMed. The journal has a system of notifying readers through e-mail when a new issue is released. Also, the articles are available in both the HTML and the PDF formats. I especially like the new and colorful page format of the journal. Also, the access statistics of the articles are available. The prepublication and the manuscript tracking system are also helpful for the authors.
Areas for improvement: In certain cases, I felt that the peer review process of the manuscripts was not up to international standards and that it should be strengthened. Also, the number of manuscripts in an issue is high and it may be difficult for readers to go through all of them. The journal can consider tightening of the peer review process and increasing the quality standards for the acceptance of the manuscripts. I faced occasional problems with the online manuscript submission (Pre-publishing) system, which have to be addressed.
Overall, the publishing process with JCDR has been smooth, quick and relatively hassle free and I can recommend other authors to consider the journal as an outlet for their work."



Dr. P. Ravi Shankar
KIST Medical College, P.O. Box 14142, Kathmandu, Nepal.
E-mail: ravi.dr.shankar@gmail.com
On April 2011
Anuradha

Dear team JCDR, I would like to thank you for the very professional and polite service provided by everyone at JCDR. While i have been in the field of writing and editing for sometime, this has been my first attempt in publishing a scientific paper.Thank you for hand-holding me through the process.


Dr. Anuradha
E-mail: anuradha2nittur@gmail.com
On Jan 2020

Important Notice

Reviews
Year : 2022 | Month : June | Volume : 16 | Issue : 6 | Page : ME01 - ME08 Full Version

Pyogenic Granuloma of the Lower Airway- A Systematic Review


Published: June 1, 2022 | DOI: https://doi.org/10.7860/JCDR/2022/53755.16496
VM Lakshana Deve, R Sithananda Kumar @Venkatesan, TC Vikram Raj Mohanam, RG Arthy, Jayapriya Djamboulingam, Mary Kurien

1. Assistant Professor, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India. 2. Associate Professor, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India. 3. Associate Professor, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India. 4. Postgraduate Resident, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India. 5. Senior Resident, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India. 6. Professor and Head, Department of ENT, Pondicherry Institute of Medical Sciences, Puducherry, India.

Correspondence Address :
Vikram Raj Mohanam,
Associate Professor, Department of ENT, Pondicherry Institute of Medical Sciences, Ganapathichettikulam, Kalapet-605014, Puducherry, India.
E-mail: drtcvikram@gmail.com

Abstract

Introduction: Pyogenic granulomas (PGs) are benign lesions that are very common in the upper aerodigestive tract. These lesions occur due to hormonal imbalance or due to a predisposing traumatic factor. They are relatively rare in the lower respiratory tract. There are few published papers of Pyogenic granulomas (PG) involving the lower airway in the past 30 years.

Aim: A case of pyogenic granuloma of larynx mimicking malignancy has been presented along with the systematic review on methods of diagnosis, successful management, and prevention of pyogenic granuloma recurrence in the lower respiratory tract.

Materials and Methods: This systematic review of literature included reports from 1981 till date, where complete details of the records were available. These reports were collected from the search engines “PubMed” and “Google Scholar” using the MeSH terms “pyogenic granuloma” OR “lobular capillary hemangioma” AND “lower respiratory tract”. The results were reviewed by three different authors independently with a main focus on methods of diagnosis and successful management and prevention of recurrence.

Results: A 59-year-old male patient presented to ENT Department with hoarseness of voice for the past six months. On examination with video laryngoscope, a pale pinkish polypoidal mass originating from the anterior commissure and extending into the subglottic wedge with normal vocal cord mobility was noted. Neck examination showed splaying of thyroid cartilage with tenderness and there were no palpable lymph nodes. CT examination suggested a neoplastic etiology of thyroid cartilage erosion. The patient was managed by microlaryngeal excision of the mass and histopathological analysis revealed pyogenic granuloma with no evidence of malignancy. From the 25 papers reviewed, a predisposing trauma like a history of intubation/lower airway procedures like bronchoscopy or laryngoscopy is not a prerequisite for the occurrence of pyogenic granuloma of the lower airway. The lesions can be excised via microlaryngoscopy or using the bronchoscope depending on the site of lesion. The various surgical modalities used for excision are cold steel dissection, laser excision and cryotherapy.

Conclusion: Meticulous dissection and removal of the lesion with post operative measures to prevent additional trauma like antireflux measures and appropriate antibiotic therapy seems to be helpful in preventing recurrence. Role of steroids in the management of pyogenic granuloma has not been supported by adequate literature. Further studies are required to comment on the adequacy of duration of follow-up.

Keywords

Larynx, Lobular capillary hemangioma, Tracheobronchial tree

Pyogenic granulomas or Lobular Capillary Haemangiomas (LCH) are common benign vascular lesions with rapid growth pattern. They are usually seen in the skin and upper aerodigestive tract. Pyogenic granulomas have a multifactorial etiology which include viral infections, chronic irritation, sudden hormonal variations, pre-existing arteriovenous malformations, and trauma (1),(2). Pyogenic granuloma of the larynx and the tracheobronchial tree is an exceedingly rare occurrence (3),(4). They are usually bound to recur and have been effectively managed by surgical excision, intralesional corticosteroids and laser therapy (5),(6),(7). This article focuses on one such case and a systematic review of the current literature on the varying manifestations, diagnostic and treatment modalities, focussing on the incidence of recurrence and the role of steroids to prevent it.

Material and Methods

A systematic review of literature was conducted according to PRISMA guidelines 2020 (8), with all published records available till date. Specific MeSh terms were used to search case reports/series those involving the lower airway from the larynx till the tracheobronchial tree. The search engines “PubMed” and “Google Scholar” were searched to retrieve the published reports.

Search strategy: To search for relevant articles, MeSH terms with a combination of Boolean operators were used. The terms used were “pyogenic granuloma” OR “lobular capillary hemangioma” AND “lower respiratory tract”.

Selection process: All articles published from the year 1980 till date were retrieved. Articles in English language with full text available were included. Those which were published in other language or those where full text was unavailable were excluded. Total 25 studies/case reports were included (Table/Fig 6).

Data collection process: The authors screened each of the retrieved record independently. All data available from each of these records were tabulated and analysed.

Data items: Any data that went missing or any unclear information were noted as such. The data from the relevant articles like the varying manifestations of symptoms, details on the exact site of presentation of the lesion, treatment modalities, duration of follow-up, incidence of recurrence and morbidity were tabulated. The following calculations were performed with the available data: average and range of age of the patients, geographic distribution, relative frequency of the most common sites of occurrence of the lesion, presenting features, management options employed, complications, rate of recurrence.

Study risk of bias or certainty assessment: Two reviewers independently screened the complete published records of each article. Disagreements were resolved by consensus or by a third reviewer.

The tools proposed by Murad MH et al., Munn Z et al., and the JBI tool was used for quality assessment (9),(8),(10),(11). The JBI tool domains and their leading explanatory questions are:

Selection: 1. Does the patient(s) represent(s) the whole experience of the investigator (centre) or is the selection method unclear to the extent that other patients with similar presentation may not have been reported?

Ascertainment: 2. Was the exposure adequately ascertained?

3. Was the outcome adequately ascertained?

Causality: 4. Were other alternative causes that may explain the observation ruled out?

5. Was there a challenge/rechallenge phenomenon?
6. Was there a dose-response effect?
7. Was follow-up long enough for outcomes to occur?

Reporting: 8. Is the case(s) described with sufficient details to allow other investigators to replicate the research or to allow practitioners make inferences related to their own practice?

Questions 4, 5 and 6 are mostly relevant to cases of adverse drug events)

Effect measures/Synthesis methods: These are not applicable as most of the records were case reports. Hence no sensitivity analysis was conducted.

Ethical concerns: An informed written consent was obtained from the patient to publish his details. There are no other ethical concerns involved in this paper.

Case Report

A 59-year-old male patient, farmer from Tamil Nadu, presented to ENT Department with persistent progressive hoarseness of voice for the past six months. There was no pain or difficulty in breathing or swallowing. He had no history of diabetes, hypertension, tuberculosis, or bronchial asthma, COPD. He did not give any history of consumption of alcohol or tobacco in any form. He denied any history of previous illness requiring hospital admission. A detailed ENT examination including video laryngoscopy (Table/Fig 1) showed a pale pinkish fleshy polypoidal mass arising from the anterior commissure and extending into the anterior subglottic wedge with normal vocal cord mobility. Airway was adequate posterior to the lesion and other areas of larynx appeared normal. Neck examination revealed splaying of thyroid cartilage with tenderness although there were no palpable lymph nodes. With the above history and clinical presentation, a provisional diagnosis of malignancy of the larynx was made and hence a computed tomography (Table/Fig 2) was requested; which was also suggestive of a neoplastic aetiology with radiologic evidence of thyroid cartilage erosion. Hence, the patient underwent for a microlaryngeal excision of the mass and biopsy (Table/Fig 3) under general anaesthesia. The histopathological analysis of the specimen (Table/Fig 4)a-c showed polypoidal lesion with ulceration of overlying epithelium. The angiomatous tissue had numerous proliferations of thin-walled blood vessels arranged in lobules. There was a background of stroma with fibroblastic proliferation and mixed inflammatory infiltrates. This was reported to be polypoidal capillary haemangioma (pyogenic granuloma) with no evidence of malignancy.

Patient was started on antibiotics (amoxicillin and potassium clavulanate 625mg, twice a day), anti reflux measures and was kept on close follow-up. In the 6th post operative week (Table/Fig 5), he again began to have hoarseness of voice and was found to have a recurrence at the same site. He was taken up for microlaryngeal excision and biopsy under general anaesthesia, once again. Histopathological evaluation revealed features of pyogenic granuloma. Adjuvant measures like adequate voice rest, speech therapy and breathing exercises along with the anti-reflux measures were started. He was also given a short course of oral steroids at a dose of 1 mg/kg body weight, under antibiotic cover, which was then gradually tapered and stopped. He was found to be asymptomatic till six months post operatively. Hence, a complete management of laryngeal pyogenic granuloma with meticulous excision, along with antireflux measures and oral steroid therapy under antibiotic- amoxicillin and potassium clavulanate 625 mg, twice a day cover helped in preventing recurrence of the lesion in this patient.

Results

There were 25 studies with complete data from the year 1981.There were three case studies/reports, one with four neonates by Walner DL et al., and the other being a retrospective study by Fechner RE et al., comprising of the records of 46 patients (3),(4). There was also a retrospective study on receptors presented in these lesions with 22 patients (12). The rest were all case reports. Most of the reports were from the USA followed by Turkey (Table/Fig 7) (1),(2),(3),(4),(5),(6),(7),(12),(13),(14),(15),(16),(17),(18),(19),(20),(21),(22),(23),(24),(25),(26),(27),(28),(29). There was a total of 94 cases reported from all the available articles other than the present case. Although PGs are more common in pregnancy due to hormonal imbalance (9), these studies showed males and females to have been equally affected. They appear to present in varied age groups (4). History of airway manipulation like an intubation were present in 75% of the study population. Tobacco and alcohol did not seem to have an additional influence according to this literature (5),(13),(15). Hoarseness of voice, cough, and haemoptysis were the predominant presenting complaints. Patients with laryngeal PG manifested with voice change whereas those with tracheal or bronchial lesions manifested with cough and haemoptysis. Although laryngeal PG were more than the tracheal PG (10/94 cases), lesions in the trachea bronchial tree were more difficult as it was difficult to secure the airway prior to the surgery or for administration of anesthesia (14). All these lesions were surgically removed using microlaryngoscopy with cold steel dissection or laser. Cryotherapy and brachytherapy were used in a few cases in an attempt to prevent recurrence (1),(27). There was one report (19), where botulinum toxin was given for a successful management and to prevent recurrence. All these lesions were confirmed histopathologically as PG. According to the data available, steroids where used in a total of 5 cases- both in adults as well as in children (3),(5). Topical application was done intraoperatively in neonates and multiple intralesional steroid injections were given in a young adolescent boy for recurrence (5). Out of 94 patients that were reported, 11 cases showed recurrence. Presence of a laryngeal trauma like an intubation is not a primary entity determining the occurrence of pyogenic granulomas. Meticulous dissection and removal along with antireflux measures seem to be the important factors determining recurrence in these cases, however there are reports in which the cause of recurrence is unknown (20),(21),(22). Administration of steroids may be helpful but is not proved to be the modality of choice to prevent recurrence.

Quality appraisal of the case reports and case series reviewed: The original articles which were 3 in number were methodologically analysed using the format used by Munn Z et al., and Murad MH et al., (20),(21). Each article was appraised for quality as tabulated in (Table/Fig 8). The validity was the average of subjective opinion of the three authors.

Discussion

Pyogenic granulomas are most often seen in children and young adults. About 70% of PGs are seen in the head and neck region. They occur predominantly in the nasal cavity, oral cavity and oropharynx (1),(2),(3). These benign tumors are known for recurrence as they have a rapid regrowth period of 18 months to 3 years. LCH or PG are pinkish friable masses that can be pedunculated or sessile, often arising from the upper aerodigestive tract (4),(5). The exact cause for PG is unknown but are often thought to be predisposed by prior insult to the local tissue in the form of trauma or instrumentation as they are often surrounded by inflammatory changes. This inference is supported by the few reports with a positive history of laryngeal manipulation prior to the occurrence of this lesion in present literature review (12),(15). Other factors seem to be hormonal shifts such as in pregnancy, effect of certain drugs, cytogenetic clonal deletion, production of angiogenetic caused by local irritation etc, VEGF, decorin, transcription factors pATF2 and pSTAT3, signal transduction pathways MPAK are factors overexpressed in PG. These are stimulated by local trauma. Their exact mechanism and roles in LCH or PGs remain unclear. In the lower airway, laryngopharyngeal reflux is an additional traumatic factor that can predispose to PGs (1),(6).

According to Fechner RE et al., there are no pyogenic granulomas in the larynx or trachea (4), but according to current review of literature, these rare laryngo- tracheo-bronchial lesions generally present with hoarseness of voice, aphonia, decrease in voice quality, dyspnoea, wheezing, sensitivity in the throat, dry cough, haemoptysis, stridor, and dysphagia. Most patients do not complain of pain. In a laryngeal PG, the subglottis is the most common subsite to be involved. (7),(12),(14). Tracheal and bronchial lesions are fewer in number and are diagnosed with fibreoptic bronchoscopy and high-resolution computer tomography (24),(25),(26),(27),(28),(29).

Pyogenic granulomas were earlier used synonymous with granulomas that occur due to a pre-existing traumatic experience like intubation granuloma that occur in the arytenoids. According to Fechner RE et al., LCH have diagnostic lobular arrangement of capillaries that clearly distinguish them from granulomas (4). They also claim that, LCH occurred spontaneously whereas granulomas occurred due to pre-existing trauma and that, they should not be used as pathological misnomers for each other (4),(5),(6),(7).

The histopathology is the gold standard of diagnosis. Microscopically, although they are similar to granulation tissue in early stages, there is a background of mixed inflammatory infiltrates with oedematous stroma. The prominent finding is supposed to be numerous capillaries and venules arranged in a radial lobular pattern. As the lesion matures, the stroma becomes more fibromyxoid and there is less inflammatory infiltrate. There are surroundings of acute and chronic inflammatory cells forming granulomas. Surface erosion or ulceration which may occur initially may reepithelialize. The differential diagnosis for these lesions includes granulation tissue, lipoma, papilloma, angiofibroma, histiocytoma, hemangiopericytoma, angioendothelioma, angiosarcoma, tuften haemangioma, intravascular angiomatosis granulomatous infections, hyperplasia, and varicosities (11),(12),(13),(14),(15).

Treatment options include surgical excision using cold steel dissection, laser photocoagulation, electrocautery snare, liquid nitrogen freezing, micro irradiation, brachytherapy, intralesional injection of ethanol or corticosteroids and sodium tetradecyl sulphate sclerotherapy. All modalities of treatment seem to give satisfying results although, recurrences have been observed in a few cases (19),(20),(21),(22),(23),(24),(25),(26),(27),(28). Although there is one report with usage of botulinum toxin to prevent recurrence, there is no strong evidence to support this finding (19). There is dearth of studies in this literature comparing the treatment modalities to prove the superiority of any single technique and chances of reducing recurrences. Adequate long term follow-up for atleast three years is required according to the current data. Many studies have been reported without following-up the patient and hence the status of recurrence cannot be commented upon. There are number of studies that had less than three months or no follow-up of the patients (2),(6),(12),(16),(22). Whilst there are reports where the lesion was successfully managed without recurrence with a follow-up of at least one year (1),(13),(20). There were two studies in which patients had multiple recurrence which was treated using multiple injections of steroids and brachytherapy respectively (5), (28). An additional therapy with anti reflux measures is proven to be beneficial. Proton pump inhibitors and H2 blockers given for a period of 12 weeks post operatively reduces laryngopharyngeal reflux and aids in preventing recurrence as seen in the present case (6).

Limitation(s)

There are several limitations in this study. Since most studies are case reports, a publication bias is possible. The results in this study depend on the quality of literature search. Completeness of the search was maximised by having two reviewers perform this task independently. Some articles were not able to be retrieved in full text and in English language and thus were not included in the study.

Conclusion

Pyogenic granulomas or lobular capillary haemangiomas occur very rarely in the lower airway. Prior trauma is not a prerequisite for these lesions. These lesions present with progressive voice change and breathing difficulty. They can be successfully managed by thorough surgical excision, intralesional or post operative steroid therapy along with long term anti reflux measures to prevent recurrence. Although there is not adequate literature to optimise the duration for follow-up in these cases, a period of 1 to 3 years following the procedure seem to be the usual teething period for recurrence, based on this systematic review. Hence, a close follow-up till 1 year and a yearly visit till 3 years postoperatively is suggested.

Acknowledgement

The authors would like to acknowledge Dr. Anand. M and the entire Department of Pathology of our institution for providing us with the histopathological images and for participating actively in ruling out other probable differentials.

References

1.
Qiu X, Dong Z, Zhang J, Yu J. Lobular capillary hemangioma of the tracheobronchial tree: A case report and literature review. Medicine. 2016;95(48):e5499. [crossref] [PubMed]
2.
Chawla M, Stone C, Simoff MJ. Lobular capillary hemangioma of the trachea: The second case. J Bronchology Interv Pulmonol. 2010;17(3):238-40. [crossref] [PubMed]
3.
Walner DL, Parker NP, Kim OS, Angeles RM, Stich DD. Lobular capillary hemangioma of the neonatal larynx. Archives of Otolaryngology-Head & Neck Surgery. 2008;134(3):272-77. [crossref] [PubMed]
4.
Fechner RE, Cooper PH, Mills SE. Pyogenic granuloma of the larynx and trachea: A causal and pathologic misnomer for granulation tissue. Archives of Otolaryngology. 1981;107(1):30-32. [crossref] [PubMed]
5.
Galos D, Chowdhury FR, Gupta R, Heman-Ackah YD, Sataloff RT. Recurrent pyogenic granuloma in a noncompliant patient. Ear, Nose & Throat Journal. 2014;93(1):E32-33. [crossref]
6.
Ozturk B, Gunduz FK, Altun E. Lobular capillary hemangioma of vocal cord in an adult. Clin Surg. 2018;3:1966.
7.
Hanick AL, Meleca JB, Billings SD, Bryson PC. Pyogenic granuloma of the larynx: A rare cause of hemoptysis. American Journal of Otolaryngology. 2019;40(2):331-33. [crossref] [PubMed]
8.
Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: An updated guideline for reporting systematic reviews. International Journal of Surgery. 2021;372:n71.
9.
Murad MH, Sultan S, Haffar S, Bazerbachi F. Methodological quality and synthesis of case series and case reports. BMJ Evid Based Med. 2018;23(2):60-63. [crossref] [PubMed]
10.
Munn Z, Barker TH, Moola S, Tufanaru C, Stern C, McArthur A et al. Methodological quality of case series studies: An introduction to the JBI critical appraisal tool. JBI evidence synthesis. 2020;18(10):2127-33. [crossref] [PubMed]
11.
Moola SZ, Munn Z, Tufanaru C, Aromataris E, Sears K, Sfetcu R, et al. Chapter 7: Systematic reviews of etiology and risk. Joanna briggs institute reviewer’s manual. The Joanna Briggs Institute. 2017.
12.
Marrinan MS, Myssiorek D, Fuchs A, Wasserman P. Laryngeal pyogenic granulomas do not express oestrogen or progesterone receptors. J Laryngol Otol. 2001;115(10):798-801. [crossref] [PubMed]
13.
Garrett MM, Lee WT. Obstructing pyogenic granuloma as a result of blunt laryngeal trauma. Otolaryngol Head Neck Surg. 2007;136(3):489-90. [crossref] [PubMed]
14.
Liew YT, Ting KN, Zulkiflee AB, Prepageran N. Different Perspective in Managing Airway Obstruction Secondary to Giant Tracheal Pyogenic Granuloma. Int J Respir Pulm Med. 2018;5(1):01-04. [crossref]
15.
Egilmez O, Uzun L, Ozkanli S, Kalcioglu M, Tekin M. A troublesome lesion of the larynx: Lobular capillary hemangioma. Scripta Scientifica Medica. 2015;47(4):56-60. [crossref]
16.
Kalanjeri S, Kumar A, Mukhopadhyay S, Mehta AC. Lobular capillary hemangioma (“pyogenic granuloma”) of the trachea. Am J Respir Crit Care Med. 2016;193(12):1429-30. [crossref] [PubMed]
17.
Acharya MN, Kotidis K, Loubani M. Tracheal lobular capillary haemangioma: A rare benign cause of recurrent haemoptysis. Case Reports in Surgery. 2016;2016:01-04. [crossref] [PubMed]
18.
Özgül MA, Tanr??verdi E, Gül S¸????, Asuk ZY, Acat M, Abbasl?? K, Fener NA, Çetinkaya E. A Rare Cause of Hemoptysis in Childhood: Tracheal Capillary Hemangioma. Turkish Thoracic Journal. 2017;18(4):131. [crossref] [PubMed]
19.
Sataloff RT, Hawkshaw MJ. Multiple bilateral vocal fold cysts and recurrent pyogenic ‘granuloma’. Ear, Nose & Throat Journal. 2001;80(2):72. [crossref]
20.
Drosnes DL, Zwillenberg DA. Laryngeal granulomatous polyp after short-term intubation of a child. Annals of Otology, Rhinology & Laryngology. 1990;99(3):183-86. [crossref]
21.
Tedla M, Bežová M, Biró C, Tedlová E, Eng CY, Zeleník K. Intravascular papillary endothelial hyperplasia of larynx: Case report and literature review of all head and neck cases. Otolaryngologia Polska. 2014;68(4):200-03. [crossref] [PubMed]
22.
Güvenç MG, Dereköylü L, Korkut N, Öz F, Öz B. Intravascular papillary endothelial hyperplasia (Masson lesion) of the hypopharynx and larynx. Ear, Nose & Throat Journal. 2008;87(12):700-04. [crossref]
23.
Irani S, Brack T, Pfaltz M, Russi EW. Tracheal lobular capillary hemangioma: A rare cause of recurrent hemoptysis. Chest. 2003;123(6):2148-49. [crossref] [PubMed]
24.
Madhumita K, Sreekumar KP, Malini H, Indudharan R. Tracheal haemangioma: case report. J Laryngol Otol. 2004;118(8):655-58. [crossref] [PubMed]
25.
Porfyridis I, Zisis C, Glinos K, Stavrakaki K, Rontogianni D, Zakynthinos S, et al. Recurrent cough and hemoptysis associated with tracheal capillary hemangioma in an adolescent boy: A case report. J Thorac Cardiovasc Surg. 2007;134(5):1366-67. [crossref] [PubMed]
26.
Amy FT, Enrique DG. Lobular capillary hemangioma in the posterior trachea: A rare cause of hemoptysis. Case Rep Pulmonol. 2012;2012:592524. [crossref] [PubMed]
27.
Udoji TN, Bechara RI. Pyogenic granuloma of the distal trachea: A case report. J Bronchology Interv Pulmonol. 2011;18(3):281-84. [crossref] [PubMed]
28.
Jie S, Hong-Rui L, Fu-Quan Z. Brachytherapy for tracheal lobular capillary haemangioma (LCH). Journal of Thoracic Oncology. 2012 1;7(5):939-40. [crossref] [PubMed]
29.
Prakash S, Bihari S, Wiersema U. A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy. BMC Pulmonary Medicine. 2014;14(1):01-04. [crossref] [PubMed]

DOI and Others

DOI: 10.7860/JCDR/2022/53755.16496

Date of Submission: Jan 06, 2022
Date of Peer Review: Feb 07, 2022
Date of Acceptance: May 20, 2022
Date of Publishing: Jun 01, 2022

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was Ethics Committee Approval obtained for this study? No
• Was informed consent obtained from the subjects involved in the study? No
• For any images presented appropriate consent has been obtained from the subjects. NA

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Jan 07, 2022
• Manual Googling: Feb 04, 2022
• iThenticate Software: Feb 11, 2022 (10%)

ETYMOLOGY: Author Origin

JCDR is now Monthly and more widely Indexed .
  • Emerging Sources Citation Index (Web of Science, thomsonreuters)
  • Index Copernicus ICV 2017: 134.54
  • Academic Search Complete Database
  • Directory of Open Access Journals (DOAJ)
  • Embase
  • EBSCOhost
  • Google Scholar
  • HINARI Access to Research in Health Programme
  • Indian Science Abstracts (ISA)
  • Journal seek Database
  • Google
  • Popline (reproductive health literature)
  • www.omnimedicalsearch.com