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

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Dr Mohan Z Mani

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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

Case Series
Year : 2023 | Month : November | Volume : 17 | Issue : 11 | Page : OR05 - OR09 Full Version

Bilateral Adrenal Masses with Different Aetiologies Presenting as Addisonian Crisis with Acute Kidney Injury: A Case Series


Published: November 1, 2023 | DOI: https://doi.org/10.7860/JCDR/2023/64509.18672
Mohini, Rohit Parsad, Shaveta Dahiya, Diksha Nayak, Manoj Malik

1. Professor, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India. 2. Postgraduate, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India. 3. Assistant Professor, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India. 4. Postgraduate Student, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India. 5. Postgraduate Student, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India.

Correspondence Address :
Rohit Parsad,
Postgraduate, Department of General Medicine, Pt. B.D. Sharma PGIMS, Rohtak-124001, Haryana, India.
E-mail: rohitparsad77@gmail.com

Abstract

An Addisonian Crisis (AC) is an endocrinological emergency associated with high mortality, resulting from an acute deficit of hormones from the adrenal glands. It can occur either in a patient with known Adrenal Insufficiency (AI) or as the initial presentation of the disease. Primary Adrenal Insufficiency (PAI), which can be caused by autoimmune adrenalitis or tubercular adrenalitis, is the most common cause worldwide and particularly prevalent in developing countries like India. Other causes of PAI include infiltrative pathology, drugs, haemorrhage, or, rarely, malignancy. Secondary Adrenal Insufficiency (SAI) often occurs due to abrupt steroid withdrawal or defects at the level of the pituitary or hypothalamus. The present case series highlights three cases (three male patients) in which patients presented with adrenal crisis despite no prior history of AI. Subsequently, they were diagnosed with bilateral adrenal masses of different aetiologies, including primary adrenal malignancy and granulomatous infiltration of the adrenals due to Tuberculosis (TB). One case involved bilateral primary adrenal malignancy with left perirenal and pararenal space infiltration, while the other two patients were diagnosed with disseminated TB with adrenal gland infiltration. The presentation of all three cases was characterised by non specific symptoms. Initial management for all three patients involved mineralocorticoid and glucocorticoid replacement, and they were discharged with corticosteroid supplements. However, the patient with bilateral adrenal malignancy was referred to the surgery and oncology department for further evaluation and management, whereas the remaining two patients were started on Antitubercular (ATT) drugs for definite treatment.

Keywords

Adrenalitis, Adrenal crisis, Glucocorticoid, Malignancy, Mineralocorticoid

The adrenal or suprarenal glands play a pivotal role in maintaining the physiological stress response, as well as Blood Pressure (BP) and electrolyte homeostasis. They mainly secrete three corticosteroids: glucocorticoid (cortisol) and mineralocorticoid (aldosterone) from the cortex, and adrenal androgen precursors and catecholamines (epinephrine and norepinephrine) from the inner medulla. Glucocorticoids and sex steroids are under the feedback control of the Hypothalamo-Pituitary-Adrenal (HPA) axis, while mineralocorticoid levels are maintained by the Renin-Angiotensin-Aldosterone System (RAAS). AI can be caused by PAI or defects at the level of the pituitary or hypothalamus SAI. PAI can result from autoimmune adrenalitis, either isolated or as part of Autoimmune Polyglandular Syndromes (APS). It can also be genetic, caused by blocks in adrenal steroidogenesis like Congenital Adrenal Hyperplasia (CAH), or by adrenal gland destruction due to infections, haemorrhage, infiltration, or rarely, metastasis or malignancy. Secondary or central AI is caused by defects at the level of the hypothalamus or pituitary, which can be inherited structural defects or hormone mutations, or acquired causes like trauma, radiation damage, infiltrations, infections, vascular issues, neoplasia, or metastasis.

Addisonian Crisis occurs due to an acute insufficiency of adrenal hormones, either because of abrupt withdrawal of steroids or failure to increase their dose in response to any form of stress like surgery, infection, or trauma. The disease rapidly evolves from initial symptoms of fatigue, weakness, anorexia, nausea, asthenia, abdominal pain, and diarrhoea, eventually leading to acute abdomen, circulatory collapse, syncope, hypoglycaemia, mental obtundation, encephalopathy, or death. Risk factors include a previous episode of adrenal crisis, increasing age group, bacterial infections (most commonly gastroenteritis, followed by respiratory and urinary tract infections), and concomitant diabetes and bronchial asthma (1).

Case 1

A 56-year-old male, who was not known to have any chronic illnesses, experienced mild, dull aching abdominal pain for one month, along with headaches and fatigue. However, he did not seek medical advice during this time. After one month of mild symptoms, he suddenly developed severe and diffuse abdominal pain. This was followed by an episode of syncope and loss of consciousness for approximately 5 to 10 minutes. As a result, the patient was brought to the hospital. During the general physical examination, the patient appeared drowsy and afebrile. His BP was 74/42 mmHg in the supine position, and his Pulse Rate (PR) was 120 beats per minute. His respiratory rate was 22 breaths per minute. Both the general physical and systemic examinations yielded normal results. Initially, the patient received an intravenous bolus of normal saline (0.9% crystalline) and later underwent Central Venous Pressure (CVP)-guided fluid administration while his vital signs were closely monitored. Routine blood investigations were conducted to rule out surgical emergencies such as visceral perforation.

The routine investigations revealed normocytic normochromic anaemia {Haemoglobin (Hb): 9.8 g/dL} and leucocytosis {Total Leukocyte Count (TLC): 14,200/mm3 with 80% Polymorphonuclear Leukocytes (PMN)}. The patient’s Renal Function Test (RFT) showed abnormal results, with blood urea of 116 mg/dL and serum creatinine of 2.9 mg/dL. Serum calcium was 8.9 g/dL, phosphorus was 3.6 mg/dL, uric acid was 6.3 mg/dL, and urine Complete Examination (CE) was normal. The patient’s Liver Function Test (LFT) showed normal results, except for low serum albumin of 2.4 mg/dL.

At the time of presentation, Arterial Blood Gas (ABG) analysis indicated uncompensated metabolic acidosis, with a pH of 7.21, bicarbonate (HCO3-) of 13.1 mmol/L, sodium (Na+) of 125 mmol/L, and potassium (K+) of 7.24 mmol/L. Random Blood Sugar (RBS) measurements were on the lower side of the normal range, with values of 68 mg/dL and 65 mg/dL. Electrocardiogram (ECG) results were normal, without any findings suggestive of hyperkalaemia. X-ray imaging of the chest and abdomen did not reveal any pathology.

Based on the investigations conducted so far, there were indications of acute stress, as evidenced by the decreased serum albumin levels. Initially, sepsis leading to septic shock was considered as a possible cause of hypotension, hypoglycaemia, leukocytosis, deranged renal parameters, and electrolyte imbalance. Blood and urine cultures were sent for testing, but they later came back as sterile. However, the presence of anaemia and one month of abdominal pain suggested an underlying chronic pathology with a strong possibility of an autoimmune condition.

On the second day of admission, the cortisol level was measured at 8 am and was found to be in the intermediate range of 13.8 ug/dL, which was alarming as it should have increased in response to stress. The adrenocorticotrophin stimulation t-test was performed the next day, revealing a basal serum cortisol level of 11.94 μg/dL. After 30 minutes and 60 minutes of synthetic Adrenocorticotropic Hormone (ACTH) stimulation, the cortisol levels were 13.77 μg/dL and 14.04 μg/dL, respectively. An abdominal Ultrasonography (USG) showed bilateral adrenal masses. The serum aldosterone level was 5.52 ng/dL, which was at the lower limits of the reference range, and the serum renin level was significantly elevated at 98 μIU/mL.

The serum Dehydroepiandrosterone Sulphate (DHEAS) level was subnormal at 10.4 μg/dL. Plasma metanephrines (45 pg/mL) and normetanephrines (151 pg/mL) were within the normal range. A Contrast Enhanced Computed Tomography (CECT) of the whole abdomen revealed well-defined lobulated heterogeneously enhancing mass lesions in the suprarenal region (Table/Fig 1),(Table/Fig 2). The right mass measured 57×40 mm and caused compression of the inferior vena cava, while the left mass measured 98×64 mm and extended into the left perinephric and left anterior pararenal space. Enlarged lymph nodes were observed in the paraaortic, paracaval, and peripancreatic regions, suggestive of a malignant aetiology. There was no evidence of primary malignancy in the chest or abdomen.

Initially, the differential diagnosis included sepsis with septic shock, adrenal crisis, and chronic duodenal ulcer with perforation peritonitis. However, all the investigations supported the diagnosis of PAI secondary to bilateral adrenal malignancy, which presented with adrenal crisis and Acute Kidney Injury (AKI). The patient received fluid resuscitation with 1.5 L of 0.9% normal saline and 1.5 L of 5% dextrose over the initial 12 to 15 hours, successfully correcting their hypotension. CVP-guided fluid administration was followed. Antihyperkalemic measures were implemented, including injection calcium gluconate, injection dextrose 25% W/V with 12.5 units of regular insulin, and nebulisation with salbutamol. Analgesics were administered for pain management. Definitive management began on the second day with an intravenous bolus of 100 mg hydrocortisone, followed by 200 mg over four divided doses. Subsequent days involved doses of 200 mg, 100 mg, and 50 mg, gradually transitioning to oral hydrocortisone (20 mg in the morning and 10 mg in the evening) on the 6th day of admission. The patient’s abdominal pain and fatigue completely resolved, and they experienced a dramatic improvement with no residual sequelae. Upon discharge, the patient was prescribed Tablet (Tab.) hydrocortisone 10 mg at 8 am, 5 mg at 1 pm, and 5 mg at 5 pm, Tab. fludrocortisone 100 mg OD, and Tab. Dehydroepiandrosterone 25 mg OD. They were referred to a specialised Endosurgery Department in a higher centre for adrenal surgery and further management.

Case 2

A 53-year-old male, known diabetic for five years on Oral Hypoglycaemic Agents (OHAs), was brought to the medicine emergency with chief complaints of dizziness associated with tremor, sweating, irrelevant talk, and palpitation for five hours. On examination, the patient was hypotensive with a BP of 80/60 mmHg, heart rate of 110/minute, and oxygen saturation of 98% at room air. The patient’s RBS was 45 mg/dL, for which he was immediately transfused with injection dextrose 25% weight/volume along with fluid therapy using 0.9% Normal saline initially as an intravenous bolus, then central venous guided. The patient regained consciousness within a few hours, his vitals became stable, and all his systemic examinations were normal. Initially, this seemed to be an episode of hypoglycaemia secondary to OHA. However, later the patient revealed that he had been experiencing mild diffuse, dull aching abdominal pain for the past 20 days and had been self-medicating for symptomatic relief. His abdominal ultrasound revealed hypoechoic lesions measuring 6.3×2.4 cm over the upper lobe of the right kidney and 7.6×3.4 cm over the left kidney, suggestive of adrenal pathology. His ABG analysis on presentation showed uncompensated metabolic acidosis with a pH of 7.19 and HCO3- of 12.0 mmol/L, hyponatraemia (Na+126 mmol/L), and hyperkalaemia (K+7.15 mmol/L), indicating AI. Other investigations, such as a complete blood count, LFT, and urine examination, were normal, but the patient had deranged renal function tests with blood urea of 110 mg/dL and serum creatinine of 2.2 mg/dL. Signs of hyperkalaemia were evident on the ECG with tall T waves and a prolonged PR segment. The patient was started on antihyperkalemic measures along with monitoring of vital signs and RBS levels. The measurement of 8 am cortisol was 1.8 μg/dL, confirming AI. Meanwhile, his sensorium deteriorated again due to hyponatraemia (Na+112 mmol/L) with recurrent episodes of hypoglycaemia, and the patient was started on injection hydrocortisone 100 mg as an intravenous bolus and 200 mg over 4 divided doses. This was followed by 200 mg, 100 mg, and 50 mg on subsequent days, and he was switched to oral hydrocortisone 20 mg in the morning and 10 mg in the evening. A CECT of the abdomen was performed when his serum creatinine improved to 1.3 mg/dL, confirming ill-defined mildly enhancing hypodense lesions in the bilateral adrenal glands, likely adrenal deposits with the possibility of granulomatous or tubercular aetiology (Table/Fig 3),(Table/Fig 4). The Mantoux test came out positive with a raised Erythrocyte Sedimentation Rate (ESR) of 60 mm in the first hour, and mycobacterium TB was detected in the sputum Cartridge Based Nucleic Acid Amplification Test (CBNAAT). Initially, the differential diagnoses considered were OHA-induced hypoglycaemia, sepsis with septic shock, adrenal crisis secondary to tubercular infiltration of adrenal glands, and myocardial infarction. However, the final diagnosis was pulmonary tuberculosis with PAI due to tubercular adrenalitis with bilateral adrenal masses presenting with adrenal crisis and AKI. The patient improved dramatically and 7was discharged on ATT, Tab. hydrocortisone 10 mg, 5 mg, and 5 mg at 8 am, 1 pm, and 5 pm respectively, and Tab. fludrocortisone 100 μg once daily. Now the patient is on Outpatient Department (OPD) follow-up, continuation phase of ATT, and has improved symptomatically.

Case 3

A 55-year-old male chronic smoker with 40 pack years, who is not known to have any other chronic illness, presented to the medicine casualty with complaints of fever. The patient had experienced eight episodes of loose stools one day ago, followed by a two hour period of altered sensorium accompanied by dizziness and irrelevant talk. The patient’s attendant reported decreased appetite, weight loss, and diffuse, dull, aching abdominal pain of insidious onset that had been present for the past two years but was non progressive. On examination, the patient was found to be hypotensive with a blood pressure of 60/44 mmHg, a pulse rate of 108/minute, a saturation level of 97% on room air, and a blood sugar level of 34 mg/dL. General physical examination revealed pallor and bilateral enlarged inguinal lymph nodes, while the rest of the systemic examinations were within normal limits. The patient’s sensorium improved after receiving a transfusion of one unit of injection dextrose 25% W/V. Simultaneously, an i.v. bolus of 0.9% normal saline was initiated, followed by an infusion of injection noradrenaline due to persistent hypotension. Routine examination revealed anaemia (Hb 9.0 g/dL), leukopenia (TLC 2600/mm3), and deranged RFT (blood urea 81 mg/dL and serum creatinine 2.5 mg/dL). LFT, lipid profile, urine CE, X-ray chest, and ECG were all normal. An Arterial Blood Gas (ABG) analysis revealed uncompensated metabolic acidosis with a pH of 7.05 and HCO3-10.8 mmol/L, hyponatraemia (Na+120 mmol/L), and hyperkalaemia (K+5.9 mmol/L) for which anti-hyperkalemic measures were administered. An abdominal USG showed normal findings except for mild fatty changes in the liver and bulky adrenal glands. The patient’s sensorium again deteriorated, and he remained in a state of refractory hypotension that did not respond to fluids and ionotropes. Considering the strong possibility of AI leading to a crisis, a serum cortisol test was performed at 8 am, which confirmed the diagnosis with a value of 2.5 μg/dL. The patient was immediately started on injection hydrocortisone 100 mg i.v., followed by 200 mg in four divided doses. With adequate fluid resuscitation, his creatinine levels decreased to 1.2 mg/dL, and a CECT scan of the abdomen revealed bilateral enlarged adrenal glands with smooth borders, peripheral calcification, minimal peripheral enhancement, and central hypoattenuating areas, indicative of adrenal TB with a few lymph nodes in the periportal and peripancreatic regions (Table/Fig 5),(Table/Fig 6). Initially, the differentials considered were sepsis with septic shock, Addison’s crisis, and malignancy (haematological or solid).

Later, his Mantoux test came out positive, with an elevated ESR of 40 mm in the first hour. Fine Needle Aspiration Cytology (FNAC) from the aspirate of the right inguinal lymph node revealed a large number of macrophages, lymphocytes, histiocytes, and a few degenerated neutrophils amidst necrotic material. Staining for Acid Fast Bacilli (AFB) yielded highly positive results, suggesting tubercular inflammation. His sensorium and hypotension improved dramatically with steroids. Thus, the final diagnosis was extrapulmonary TB with PAI, accompanied by Addison’s crisis due to TB adrenalitis, which presented as an adrenal crisis likely triggered by acute gastroenteritis with AKI. Like the previous patient, he was discharged on Tab. hydrocortisone 10 mg, 5 mg, and 5 mg at 8 am, 1 pm, and 5 pm respectively, Tab. fludrocortisone 100 μg, and antitubercular drugs. However, after leaving the hospital, the patient did not take his medications and was subsequently lost to follow-up.

Discussion

The AI can be primary or secondary due to acquired or inherited causes. The condition may remain undetected in a compensated state until an acute illness or any form of stress precipitates it into a state of adrenal or AC (1). The prevalence of adrenal crisis ranges from 5.2 to 8.3 per 100 patient-years (2),(3). TB was the most common cause (70%) during the 1930s. Currently, autoimmune adrenalitis (Addison’s disease) is the most common cause of PAI in developed countries, while TB remains the leading cause of AI in developing countries (4). However, in many cases, the cause may remain unidentified. The clinical presentations start with vague symptoms like fatigue, abdominal pain, and anorexia, progressing into syncope, confusion, encephalopathy, etc. Upon evaluation, these patients usually have normocytic anaemia, eosinophilia, lymphocytosis, hypotension, hypoglycaemia, AKI, hyperkalaemia, hyponatraemia, and metabolic acidosis. The definitive diagnosis of AI requires the demonstration of low serum cortisol. High serum ACTH with no rise in cortisol levels on ACTH stimulation suggests PAI, while low ACTH levels with normal mineralocorticoid levels are seen in SAI. The next step is to establish if the cause is treatable and manage it.

Mineralocorticoid deficiency can be established with low or normal serum aldosterone and increased serum renin. A prospective study involving 364 patients over two years concluded that AC-associated mortality is around 6% of the total patients who had a crisis and can occur even in educated patients (3). Thus, in acutely ill patients, treatment should be started without delay, and meanwhile, blood samples could be sent or kept for diagnostics.

An adrenal incidentaloma refers to a mass lesion with a diameter greater than 1 cm, typically unilateral, benign, and non functioning.

However, it can also be bilateral, malignant, and secretory. Bilateral adrenal masses can be associated with cortical adenomas, lymphoma, Adrenocortical Carcinomas (ACC), metastasis, infections (such as TB or fungal), haemorrhage, amyloidosis, or infiltrative diseases. Primary adrenal malignancy is much less common than secondary metastasis to the adrenals, with an estimated incidence of 7.4 cases per million population, most of which are ACC. In rare cases, adrenal malignancy can result in adrenocortical hypofunction instead of hormone excess (5). This occurs when more than 90% of the adrenal glandular tissue is destroyed, leaving the remaining tissues unable to compensate through hypertrophy or hyperplasia (6). AI can also rarely occur due to pituitary metastasis with hormone deficits, adrenalitis, immunotherapies for certain malignancies like melanoma, or as an iatrogenic effect of adrenolytic agents such as mitotane used in ACC treatment (7),(8).

In a retrospective study by Lubomski et al., which analysed 15,376 patients with adrenal malignancies from 2006 to 2017, 182 cases of AI were reported, with 21 (11%) being primary and the majority 161 (88.5%) being secondary to malignancy. Similarly, out of a total of 24 AC, 5 (20.8%) were primary and 19 (79.2%) were associated with secondary malignancy (9). Patient with bilateral adrenal malignancy is a scarce finding and those presenting with AC is equally unique. CT, MRI, Positron Emission Tomography (PET) scan, fine needle aspiration biopsy are the investigation modalities used to define the type, nature (benign or malignant) and extent of the disease (10).

In all three of our cases, adrenal masses were initially detected using USG and later confirmed to be bilateral adrenal malignancy or bilateral tubercular deposits through CECT scans of the abdomen. Nevertheless, the occurrence of bilateral adrenal malignancy or bilateral adrenal masses secondary to tubercular adrenalitis presenting with acute adrenal crisis and AKI is extremely rare in Rohtak, Haryana region and possibly the first of its kind.

Conclusion

The AC is a medical emergency that requires early identification of the condition and immediate management. Many patients may lack prior knowledge of their disease and present for the first time, so a high degree of vigilance is required to avoid missing them. Early supportive management should be followed by treatment with corticosteroid replacement. After stabilisation, the cause of AI (Addison’s disease) - primary or secondary - can be evaluated, which might lead to the discovery of rare conditions like bilateral adrenal masses due to malignancy or TB.

Addison’s crisis is a common disease that requires a high level of suspicion, early diagnosis, and treatment to prevent mortality. Patient education is necessary to explain the need for compliance with medications and increasing their dose during acute illness to prevent future crises. In conclusion, AC may be the first presenting manifestation of bilateral adrenal masses attributed to various causes, including primary bilateral malignancy, granulomatous lesions like TB and sarcoidosis, which can be devastating for a patient if not diagnosed and treated in a timely manner.

References

1.
Rushworth RL, Torpy DJ. A descriptive study of adrenal crises in adults with adrenal insufficiency: Increased risk with age and in those with bacterial infections. BMC Endocrinol Disord. 2014;14:79. Doi: 10.1186/1472-6823-14-79. [crossref][PubMed]
2.
Smans LC, Van der Valk ES, Hermus AR, Zelissen PM. Incidence of adrenal crisis in patients with adrenal insufficiency. Clin Endocrinol (Oxf). 2016;84(1):17-22. [crossref][PubMed]
3.
Hahner S, Spinnler C, Fassnacht M, Burger-Stritt S, Lang K, Milovanovic D, et al. High incidence of adrenal crisis in educated patients with chronic adrenal insufficiency: A prospective study. J Clin Endocrinol Metab. 2015;100(2):407-16. [crossref][PubMed]
4.
Omori K, Nomura K, Shimizu S, Omori N, Takano K. Risk factors for adrenal crisis in patients with adrenal insufficiency. Endocr J. 2003;50(6):745-52. [crossref][PubMed]
5.
Chandrasekar T, Goldberg H, Klaassen Z, Wallis CJD, Woon DTS, Herrera-Caceres JO, et al. The who, when, and why of primary adrenal malignancies: Insights into the epidemiology of a rare clinical entity. Canc. 2019;125(7):1050-59.[crossref][PubMed]
6.
Patrova J, Jarocka I, Wahrenberg H, Falhammar H. Clinical outcomes in adrenal incidentaloma: Experience from one center. Endocr Pract. 2015;21(8):870-77. [crossref][PubMed]
7.
Salinas C, Renner A, Rojas C, Samtani S, Burotto M. Primary adrenal insufficiency during immune checkpoint inhibitor treatment: Case reports and review of the literature. Case Rep in Oncol. 2020;13(2):621-26. [crossref][PubMed]
8.
Lin CH, Chen KH, Chen KY, Shih SR, Lu JY. Immune checkpoint inhibitor therapy-induced hypophysitis~a case series of Taiwanese patients. J of the Formos Med Assoc. 2019;118(1):524-29. [crossref][PubMed]
9.
Lubomski A, Falhammar H, Torpy DJ, Rushworth RL. The epidemiology of primary and secondary adrenal malignancies and associated adrenal insufficiency in hospitalised patients: An analysis of hospital admission data, NSW, Australia. BMC Endocrinol Disord. 2021;21(1):141. Doi: 10.1186/s12902-021-00787-6. [crossref][PubMed]
10.
Fassnacht M, Dekkers OM, Else T, Baudin E, Berruti A, De Krijger RR, et al. European Society of Endocrinology Clinical Practice Guidelines on the management of adrenocortical carcinoma in adults, in collaboration with the European Network for the Study of Adrenal Tumors. Eur J of Endocrinol. 2018;179(4):G1-G46.[crossref][PubMed]

DOI and Others

DOI: 10.7860/JCDR/2023/64509.18672

Date of Submission: Apr 06, 2023
Date of Peer Review: Jun 09, 2023
Date of Acceptance: Jul 31, 2023
Date of Publishing: Nov 01, 2023

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Apr 08, 2023
• Manual Googling: Jun 17, 2023
• iThenticate Software: Jul 28, 2023 (5%)

ETYMOLOGY: Author Origin

EMENDATIONS: 6

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