Bilateral Adrenal Hemorrhage Research Articles

Waterhouse-Friderichsen syndrome, septic adrenal apoplexy.

Waterhouse-Friderichsen syndrome is a rare but potentially fatal disorder of the adrenal gland characterized by bilateral adrenal hemorrhage. It is classically a result of meningococcal sepsis and presents acutely with features of shock, petechial rashes, abdominal pain, and non-specific symptoms such as headache, fatigue, and vomiting. Treatment consists of fluid resuscitation, corticosteroid replacement, and possibly surgery. The prognosis is poor despite treatment. This chapter will review the etiology, pathogenesis, clinical features, and management of the disease.

HIT（heparin–induced thrombocytopenia）により両側副腎出血を来し副腎機能不全を呈した墜落外傷の1例(A case of fall trauma with bilateral adrenal hemorrhage and adrenal insufficiency due to HIT (heparin–induced thrombocytopenia))

要旨 HIT（heparin–induced thrombocytopenia）により両側副腎出血，副腎機能不全を呈した報告はわが国では1例のみである。今回HITを発症し両側副腎出血により副腎機能不全を呈した外傷の1例を経験したので報告する。症例は50歳代の女性。墜落により胸腰椎，骨盤，下肢の骨折を受傷，動脈塞栓術と創外固定術を行い入院した。入院後血小板数は減少したが数日で増加傾向にあった。第7病日に胸腰椎，骨盤に対して内固定術を行い，第10病日より血栓形成の予防目的にヘパリンを持続で開始した。第12病日に血小板数が50%以上減少したためHITを疑い，入院時から使用していた動脈圧ライン内に添加したヘパリンとヘパリンの持続投与を中止した。その後血小板数は増加し，HIT抗体が陽性でありHITと診断した。第13病日に施行した造影CTで両側副腎出血と右外腸骨静脈血栓を認めた。副腎出血は経過観察とし，外腸骨静脈血栓に対しては下大静脈フィルターを留置した。第15病日，迅速ACTH（adrenocorticotropin hormone）負荷試験により副腎機能不全と診断しステロイドを開始し，反応は良好であった。その後副腎出血の増大はなく血栓も縮小傾向となり第42病日，他院に転院した。重症外傷症例では，HITの発症を考慮する必要があり，さらに両側副腎出血，副腎機能不全の合併も念頭におくべきである。

A case of multiple trauma complicated with bilateral adrenal hemorrhage causing refractory hypotension

A case of multiple trauma complicated with bilateral adrenal hemorrhage causing refractory hypotension

A Case of Laryngeal Cancer with Bilateral Adrenal Hemorrhage Associated with Sepsis due to Radiation-Induced Mucositis That Showed a Rapidly Deteriorating Course

A Case of Laryngeal Cancer with Bilateral Adrenal Hemorrhage Associated with Sepsis due to Radiation-Induced Mucositis That Showed a Rapidly Deteriorating Course

Covid-19 Patient with Combined Bilateral Adrenal Hemorrhage

Covid-19 Patient with Combined Bilateral Adrenal Hemorrhage

A COVID-19 PATIENT WITH COMBINED BILATERAL ADRENAL HEMORRHAGE.

A COVID-19 PATIENT WITH COMBINED BILATERAL ADRENAL HEMORRHAGE.

Hémorragie bilatérale des surrénales sous apixaban dans le syndrome primaire des antiphospholipides

Adrenal hemorrhage is a classical but rare complication of antiphospholipid syndrome, revealing diagnosis in one third of the cases. Anti-vitamin K therapy is the standard treatment but direct oral anticoagulants are discussed as an alternative. In the latest recommendations, it is advised not to use direct oral anticoagulants in the setting of antiphospholipid syndrome.We present a case of bilateral adrenal hemorrhage revealing primary antiphospholipid syndrome with triple positive antibody profile, in 53-year-old women treated by apixaban for previous venous thromboembolism.

Approach to the Patient With Adrenal Hemorrhage.

Adrenal hemorrhage is an uncommon, underrecognized condition that can be encountered in several clinical contexts. Diagnosing adrenal hemorrhage is challenging due to its nonspecific clinical features. Therefore, it remains a diagnosis that is made serendipitously on imaging of acutely unwell patients rather than with prospective clinical suspicion. Adrenal hemorrhage can follow abdominal trauma or appear on a background of predisposing conditions such as adrenal tumors, sepsis, or coagulopathy. Adrenal hemorrhage is also increasingly reported in patients with COVID-19 infection and in the context of vaccine-induced immune thrombocytopenia and thrombosis. Unexplained abdominal pain with hemodynamic instability in a patient with a predisposing condition should alert the physician to the possibility of adrenal hemorrhage. Bilateral adrenal hemorrhage can lead to adrenal insufficiency and potentially fatal adrenal crisis without timely recognition and treatment. In this article, we highlight the clinical circumstances that are associated with higher risk of adrenal hemorrhage, encouraging clinicians to prospectively consider the diagnosis, and we share a diagnostic and management strategy.

PSUN43 Anti-phospholipid syndrome presenting with bilateral adrenal hemorrhage in a young female

Abstract Patient is a 29-year-old female with PCOS and morbid obesity and recent admission for abdominal pain is found to have bilateral adrenal hemorrhage and prolonged PTT. Patient was found to be anemic with hemoglobin of 6.1 a week prior to presentation, attributed to dysfunctional uterine bleeding and was treated with subsequent transfusion. She presents a week after her initial presentation with recurrent constant sharp pulsating abdominal pain, and reported pain was worse in mid-epigastric area radiating into her back, with a temperature of 38.2 and heart rate of 135, troponin level 2.64, with a negative CT for PE, but incidental bilateral adrenal hemorrhage absent on CTA from last week, with a CBC showing WBC 21.7 RBC 3.5 Hgb 8.6 MCV 76.9 MCH 24.7 RDW 17.6, with normal folate and B12. Patient was monitored closely and was started on IV hydrocortisone 50 mg every six hours, her PTT was found to be 88 and PT of 17.5, so antiphospholipid syndrome was suspected and confirmed with positive lupus anti-coagulant (LAC) and anticardiolipin IgG ab, and B2 glycoprotein. Patient had no evidence of adrenal infract that could have converted to hemorrhage. Objectives Antiphospholipid syndrome (APS) is an autoimmune systemic disorder characterized by persistent antiphospholipid antibodies including lupus anticoagulant, anticardiolipin or anti B2- glycoprotein I antibodies, presenting with arterial venous or small vessel thrombosis or recurrent early pregnancy loss. Other manifestation including livedo reticularis, cutaneous ulceration, thrombocytopenia, and hemolytic anemia, valvular heart disease and nephropathy. In our case, the patient APS was confirmed by the presence of the markers, but no evidence of adrenal infracts what could have converted to hemorrhage was found. Conclusions Although APS leads to a hypercoagulable state which can lead to adrenal vein thrombosis, leading to adrenal hemorrhage which can be fatal. Screening for antiphospholipid antibodies should be considered in all patients with unknown etiology of adrenal hemorrhage to promote diagnosis and timely management. Presentation: Sunday, June 12, 2022 12:30 p.m. - 2:30 p.m.

PSAT040 Bilateral Adrenal Infarcts: Initial Presentation in Concurrent Myelodysplastic Syndrome and Anti-Phospholipid Syndrome

Abstract Introduction Bilateral adrenal hemorrhage (BAH) has been noted in several conditions. Bilateral adrenal infarcts (BAI) are rarer but have been seen in anti-phospholipid syndrome (APS). Adrenal insults are not commonly associated with myelodysplastic syndrome (MDS). We present a patient who developed BAI as initial presentation of APS in the setting of MDS and discuss management considerations. Case A 58-year-old female with MDS diagnosed one month prior presented with abdominal pain, present for one year but acutely worse in the past week. CT A/P showed nodularity of bilateral adrenal glands. She was treated conservatively but continued to experience nausea, vomiting, and abdominal pain – repeat CT A/P one month later was suspicious for bilateral peri-adrenal hemorrhages. MRI with adrenal protocol demonstrated bilateral non-hemorrhagic adrenal infarcts. VS were negative for hypothermia or hypotension. Labs showed Na 136 (135-145 mmol/L), K 3.5 (3.4-5.1 mmol/L), BG 108 (mg/dL), DHEA-S 9.3 (8.0-391 mcg/dL), renin 6.7 (0.5-4.0 ng/mL/hr), aldosterone 6.2 (<31.0 ng/dL), 60-minute cortisol after ACTH stimulation test 34.3 (5.2-22.5 mcg/dL), plasma metanephrines <0.10 (0.00-0.49 nmol/L), plasma normetanephrines 0.34 (0.00-0.89 nmol/L), Hgb 11.8 (12.0-15.5 g/dL), and plts 129K (140-450 K/mcL). No history of anticoagulant use, trauma, or VTE. COVID-19 PCR was negative on two separate tests. Hypercoagulability testing noted positive lupus anticoagulant. Anticoagulation was deferred due to underlying bone marrow abnormalities and thrombocytopenia; aspirin was recommended instead by hematology. Outpatient testing revealed 12pm cortisol of 12.6 (5.2-22.5 mcg/dL), ACTH 108.3 (7.2-63.0 pg/mL), and DHEA-S 6.2 (8.0-391 mcg/dL). Patient was empirically started on hydrocortisone for glucocorticoid replacement therapy. Conclusion BAI is not typically associated with MDS but can occur in the setting of a concurrent hypercoagulable process such as APS. The adrenal glands are the most commonly involved glands in APS. Patients often present in adrenal crisis but can present in stable condition as in this case. The adrenal glands are particularly vulnerable due to the "vascular dam" near the zona reticularis. Imaging and laboratory investigation can clarify the nature of the adrenal insult and guide management. Treatment strategy may include anticoagulation to prevent further VTE events, but bleeding risk must be considered. Both glucocorticoid and mineralocorticoid replacement should be considered until residual adrenal reserve can be fully elucidated, which may take several months from initial diagnosis. References Rao RH. Bilateral massive adrenal hemorrhage. Med Clin North Am 1995; 79(1): 107-29. Aron DC, Findling JW, Tyrrell JB. Glucocorticoids and adrenal androgens. In: Greenspan's Basic and Clinical Endocrinology. Gardner DG, Shoback D (eds). Mc Graw Hill 2007; pp 367-378. Caron P, Chabannier MH, Cambus JP, Fortenfant F, Otal P, Suc JM. Definitive adrenal insufficiency due to bilateral adrenal hemorrhage and primary antiphospholipid syndrome. J Clin Endocrinol Metab 1998; 83(5): 1437-9. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

PSAT045 Spontaneous Adrenal Hemorrhage with Partial Adrenal Insufficiency in a Patient Anticoagulated with Apixaban for Antiphospholipid Syndrome

Abstract Background Addison's disease secondary to adrenal hemorrhage is a rare complication of antiphospholipid syndrome (APLS). Anticoagulation with a direct oral anticoagulant (DOAC) might increase the risk of adrenal hemorrhage. We report a patient who developed bilateral adrenal hemorrhage after starting apixaban for deep venous thrombosis (DVT) and pulmonary embolism (PE) in the setting of latent APLS. Clinical Case A 45-year-old man without significant past medical history presented with a left lower extremity DVT and PE. He had no personal or family history of previous trauma, bleeding or clotting events. He was discharged on apixaban. Three days later he was readmitted with bilateral flank pain. Computerized tomography (CT) imaging showed bilateral adrenal gland edema. He was hemodynamically stable. Due to persistent right flank pain and concern for ileus, CT was repeated and revealed findings concerning for adrenal hemorrhage. He subsequently complained of asthenia. Further testing revealed a reduced morning cortisol of 1.4 ug/dL (reference: 6.0 - 18.4 ug/dL) and an elevated morning ACTH of 149 pg/mL (7.2-63.3 pg/mL). Plasma dehydroepiandrosterone sulphate (DHEA-S) was reduced at 39 mcg/dL (70 - 495 mcg/dL), and epinephrine and norepinephrine were undetectable. Morning plasma aldosterone was undetectable and renin activity was 0.76 ng/mL/h (0.25 - 5.82 ng/mL/h). He was started on stress dose steroids and discharged on maintenance hydrocortisone without reinitiating anticoagulation. Two weeks later, he re-presented with a right lower extremity DVT. An APLS panel revealed elevated anti-beta2 glycoprotein 1 IgG and anti-cardiolipin IgG. Laboratory work-up for other causes of thrombosis, including inherited and acquired thrombophilias, anatomical vascular obstruction, paroxysmal nocturnal hemoglobinuria (PNH), heparin-induced thrombocytopenia (HIT), and myeloproliferative neoplasms (MPN), was unrevealing. Warfarin was started with an international normalized ratio (INR) target range of 2-3. Hydrocortisone was continued at a total daily dose of 30mg. Since discharge, his morning cortisol levels have been low at 4.4-4.9 ug/dL and ACTH has remained high at 92-137 pg/mL, but aldosterone has been variable at <1 to 9 ng/dL, and renin has been normal at 0.35 ng/mL/h. This suggests what transpired had been microthrombi-associated intra-adrenal hemorrhage involving the zona fasciculata with sparing of the glomerulosa. Conclusion This case highlights DOAC-associated adrenal hemorrhage in a patient with underlying APLS, an under-appreciated complication. The extent to which the various layers of the adrenal gland are affected may vary. Since guidelines on the management of patients with partial adrenal insufficiency are lacking, further reporting is needed on long term outcomes and adrenal function of these patients. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

Postmortem PF4 antibodies confirm a rare case of thrombosis thrombocytopenia syndrome associated with ChAdOx1 nCoV-19 anti-COVID vaccination.

We report a case of cerebral venous sinus thrombosis, bilateral adrenal hemorrhage, and thrombocytopenia in a 70-year-old man found dead. He had previously received the ChAdOx1 nCoV-19 vaccine (Vaxzevria®, AstraZeneca) 18 days before, and had since developed unspecific and undiagnosed characteristics of what proved to be a rare case of vaccine-associated thrombocytopenia with thrombosis syndrome (TTS). He was found dead 1 week after the beginning of symptoms (day 25 post-vaccine). Autopsy yielded venous hemorrhagic infarction with the presence of thrombi within dural venous sinuses, and extensive hemorrhagic necrosis of the central part of the adrenal glands. Antibodies against platelet factor 4 (PF4) were strongly positive in postmortem fluids, as measured with an enzyme-linked immunosorbent assay (ELISA). This difficult diagnosis is usually made during the patient’s lifetime. After eliminating differential diagnoses, we concluded on a fatal case of vaccine-induced immune TTS with positive anti-PF4 antibodies in cadaveric blood, 3 weeks after ChAdOx1 nCoV-19 vaccination. Specific search for anti-PF4 antibodies in cadaveric blood appears therefore paramount to assess postmortem cases of TTS associated with anti-COVID vaccines.

Multimodality Imaging Findings in Waterhouse-Friderichsen Syndrome: A Case Report and a Short Literature Review

Waterhouse-Friderichsen syndrome is a rare disease characterized by bleeding into the adrenal glands, and its symptoms may vary from non-specific abdominal pain to multi-organ failure. Commonly related to a variety of infectious diseases among which sepsis from Neisseria meningitidis is the most frequent, many other etiologies have been described, and in some rare cases, even no apparent cause can be identified. We report a rare case of an idiopathic bilateral adrenal hemorrhage in a 70-year-old male who presented to our emergency department with non-specific abdominal pain. Ultrasonography Scan (US) and Computed Tomography (CT) findings were crucial for making an early diagnosis, which resulted in a short hospitalization time and, eventually, a complete recovery of the adrenal function. After discharge, Magnetic Resonance Imaging (MRI) was also performed, providing a more complete ensemble of multimodality imaging findings that can be associated with this rare syndrome.

Scrotal Hematoma Caused by Neonatal Bilateral Adrenal Hemorrhage: A Case Report

Scrotal Hematoma Caused by Neonatal Bilateral Adrenal Hemorrhage: A Case Report

A case of ChAdOx1 vaccine-induced thrombocytopenia and thrombosis syndrome leading to bilateral adrenal haemorrhage and adrenal insufficiency

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Unilateral Adrenal Infarction in Pregnancy: A Case Report

Adrenal infarction is usually associated with bilateral adrenal hemorrhage in the setting of antiphospholipid syndrome or hemodynamic variation. Few cases of unilateral non-hemorrhagic adrenal infarction have been described in the literature. Here, we report a case occurring during pregnancy. A 27-year-old woman was infected by coronavirus four months ago and presented at 35 weeks of gestation with sudden-onset right abdominal pain without contractions. Unilateral adrenal infarction was diagnosed following computed tomography. It showed an enlarged right adrenal, without hyperenhancement. The patient’s adrenal hormonal function was normal. Accurate diagnosis of non-hemorrhagic adrenal infarction remains difficult as its clinical presentation is not specific. It can only be performed with adrenal imaging. Magnetic resonance imaging shows diffuse enlargement of one or both adrenals and edema on T2-weighted images. Anticoagulation therapy may be discussed. Patients should be evaluated between 3 and 6months after the event to assess adrenal size and function. In summary, non-hemorrhagic adrenal infarction during pregnancy is probably underdiagnosed and obstetricians should be aware of this diagnostic difficulty.

A case of ChAdOx1 vaccine-induced thrombocytopaenia and thrombosis syndrome leading to bilateral adrenal haemorrhage and adrenal insufficiency

A case of ChAdOx1 vaccine-induced thrombocytopaenia and thrombosis syndrome leading to bilateral adrenal haemorrhage and adrenal insufficiency

Neonatal bilateral adrenal hemorrhage and adrenal insufficiency accompanied by Subgaleal hematoma: a case report with brief review of literature

BackgroundNeonatal adrenal hemorrhage (NAH) is an almost infrequent phenomenon (0.2–0.55%). Mechanical compression and alterations of venous pressure during delivery are considered the most probable explanations. Approximately 10% of the cases might have bilateral involvement. Clinical symptoms include abdominal mass, poor feeding, vomiting, prolonged jaundice, and anemia. Subgaleal hemorrhage (SGH) is one of the most clinically remarkable and potentially hazardous postnatal cranial injuries.Case presentationAn early-term Iranian male neonate who was born through spontaneous vaginal delivery and experienced shoulder dystocia was diagnosed with bilateral NAH leading to adrenal insufficiency requiring glucocorticoid and mineralocorticoid supplementation. The SGH and jaundice were other postnatal complications. Serial monthly abdominal and brain ultrasound revealed complete regression of lesions after 70 days. However, after 16 months, the neonate has been still treated with hydrocortisone and fludrocortisone for the adrenal insufficiency diagnosis. He has a lower limit weight for age; however, developmental milestones have been appropriate for age.Discussion and conclusionAdrenal hemorrhage and SGH should be examined and looked for, particularly with proven evidence of difficult delivery and asphyxia in at-risk newborns. Clinical and ultrasound follow-up is mandatory for the assessment of hemorrhage resolution and conservative management. The early detection and treatment of adrenal insufficiency by laboratory examination is strongly recommended in bilateral cases. Furthermore, the early recognition of postnatal SGH to prevent clinical and neurological outcomes seems essential.

A case of ChAdOx1 vaccine-induced thrombocytopenia and thrombosis syndrome leading to bilateral adrenal haemorrhage and adrenal insufficiency.

SummaryVaccine-induced thrombosis and thrombocytopenia (VITT) after vaccination against SARS-CoV-2 with the adenoviral vector-based vaccines ChAdOx1 and Ad26.COV2.S has been associated with adrenal pathology, such as bilateral adrenal vein thrombosis, adrenal cortex haemorrhage and adrenal insufficiency in 6% of patients. We report the case of a 23-year-old woman who presented at 8 days after ChAdOx1 vaccination with a low platelet count of 43 × 109/L, raised d dimers >100 000 ng/mL and multiple lobar and segmental pulmonary emboli. Anti-platelet factor 4 antibodies were detected confirming definite VITT in accordance with the UK diagneostic criteria. At 16 days post-vaccine, further imaging showed bilateral adrenal haemorrhage, non-occlusive splenic vein thrombosis and right ventricular thrombosis. Her cortisol level was <25 nmol/L. She was treated with anticoagulation, plasmapheresis, immunosuppression and steroid replacement. She had high anti-spike titre and positive anti-nucleocapsid titres for SARS-CoV-2. She developed seizures secondary to posterior reversible encephalopathy, requiring intensive care. After 4 weeks in hospital, she was discharged on warfarin, hydrocortisone and fludrocortisone replacement. Short synacthen tests 3 and 9 months later showed no recovery of adrenal function, although magnetic resonance imaging of the adrenal glands showed resolving adrenal haemorrhage. Adrenal insufficiency secondary to bilateral adrenal vein thrombosis and adrenal haemorrhage should be suspected in patients with VITT and treated promptly. Adrenal vein thrombosis can occur either as the initial presentation of VITT or days to weeks after the development of thrombosis in other sites. Further studies are required to provide insight on adrenal function recovery after VITT.Learning pointsAdrenal insufficiency secondary to bilateral adrenal vein thrombosis and adrenal cortex haemorrhage should be suspected in patients with vaccine-induced thrombosis and thrombocytopenia (VITT) and treated promptly.Adrenal vein thrombosis can occur as the initial presentation of VITT or even days to weeks later after the development of thrombosis in other more classic sites (e.g. pulmonary or cerebral vasculature).Completion of vaccination schedule against SARS-CoV-2 post-VITT using an mRNA-based vaccine should be recommended to patients post-VITT as mRNA-based vaccines have not been associated with VITT but confer protection against SARS-CoV-2.There is paucity of data regarding the potential for recovery of adrenal function after bilateral adrenal haemorrhage in the context of VITT, and thus, more studies are needed to inform clinical practice.The need for disease registries for rare conditions, such as VITT, is crucial as direct cooperation and sharing of information by clinicians might enable quicker identification of disease patterns than would have been possible via established reporting tools of adverse events.

Adrenal gland haemorrhages following motor vehicle accident with resultant adrenal insufficiency.

SummaryAdrenal gland haemorrhage is an uncommon, yet likely under-diagnosed complication of high-impact trauma, such as motor vehicle accidents (MVA). It usually occurs with multi-trauma and is associated with additional injuries to the ribs, liver, kidney, spleen and vertebrae. Trauma cases with resultant adrenal gland injury have higher mortality rates. Primary adrenal insufficiency as a result of bilateral adrenal haemorrhage is potentially fatal. We report three cases of life-threatening adrenal insufficiency following adrenal injuries sustained in MVA’s. Case 1 was a 60-year-old-male who presented with acute haemodynamic instability on admission. Case 2 was an 88-year-old female on anticoagulation for atrial fibrillation, who developed haemodynamic instability 10 days into her admission. Case 3 was a 46-year-old male who developed hyponatraemia 2 weeks post-MVA. All were commenced on stress dose hydrocortisone replacement with improvement in clinical status. Only case 1 has had complete adrenal axis recovery, whereas the other patients remain on maintenance hydrocortisone replacement. Our cases demonstrate acute and subacute presentations of adrenal insufficiency following traumatic bilateral adrenal haemorrhages and highlight the importance of assessing adrenal morphology and function in any trauma patient with haemodynamic instability or hyponatraemia.Learning pointsAdrenal gland haemorrhage is an under-diagnosed consequence of high-impact trauma.Trauma patients with adrenal haemorrhage have a significantly increased mortality risk.Bilateral adrenal gland haemorrhage can result in life-threatening adrenal insufficiency requiring urgent glucocorticoid replacement.Biochemical assessment of the adrenocortical axis should be considered in all patients presenting with high-impact trauma following motor vehicle accidents.Given the potential for delayed presentation, any patients with new haemodynamic instability should have repeat biochemistry and/or imaging performed, even if initial adrenal imaging and investigations were normal.