Case Report
Nonhemorrhagic Adrenal Infarction With Magnetic Resonance Imaging Features During Pregnancy Jeffrey P. Guenette, MD, and Servet Tatli, MD BACKGROUND: Adrenal infarction is an infrequent cause of severe abdominal pain during pregnancy. The magnetic resonance imaging (MRI) features of adrenal infarction have not previously been thoroughly described. CASES: A 20-year-old woman, gravida 1 para 0, presented at 27 4/7 weeks of gestation with sudden-onset right upper quadrant and flank pain. A 29-year-old woman, gravida 2 para 1, presented at 17 5/7 weeks of gestation with suddenonset right abdominal and flank pain and again at 35 5/7 weeks of gestation with sudden-onset severe left flank and upper quadrant pain. In both patients, unilateral adrenal infarction was diagnosed on contrast-enhanced computed tomography after initial nondiagnostic ultrasonography and MRI. Clinical presentation and MRI features of nonhemorrhagic adrenal infarction are described. CONCLUSION: Nonhemorrhagic adrenal infarction may be an underdiagnosed cause of acute abdominal pain during pregnancy and can be diagnosed with MRI. (Obstet Gynecol 2015;126:775–8) DOI: 10.1097/AOG.0000000000000884
A
drenal infarction in pregnancy is a rare clinical entity. It can cause sudden, severe, unremitting upper right or left quadrant pain similar to other acute intraabdominal pathologies such as biliary colic, renal colic, and appendicitis. Diagnosis of nonhemorrhagic adrenal infarction can be made with computed tomography (CT),1–3 but, ideally, CT would be avoided in pregnant patients because it involves exposure of the fetus to radiation. Similarly, a single report suggests that it From the Brigham & Women’s Hospital, Department of Radiology, Harvard Medical School, Boston, Massachusetts. Corresponding author: Jeffrey P. Guenette, MD, Brigham & Women’s Hospital, Department of Radiology, Harvard Medical School, Boston, MA 02115; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2015 by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0029-7844/15
VOL. 126, NO. 4, OCTOBER 2015
Teaching Points 1. Consider adrenal infarction in the differential of acute severe upper abdominal pain during pregnancy. 2. Magnetic resonance imaging features of nonhemorrhagic adrenal infarction include diffuse enlargement of the adrenal gland, decreased signal intensity on T2-weighted images, and edema in the adjacent retroperitoneum. 3. Consider hematology evaluation in the setting of adrenal infarction.
may be possible to diagnose nonhemorrhagic adrenal infarction with contrast-enhanced magnetic resonance imaging (MRI),4 but MRI contrast is usually avoided in pregnant patients as a result of potential fetal toxicity. A search of MEDLINE (English language; 1966 to November 2014; search terms: “adrenal infarction” and “MRI”) revealed no thorough description of nonhemorrhagic adrenal infarction with noncontrast MRI. We present two patients with nonhemorrhagic adrenal infarction in pregnancy in the setting of severe pain refractory to narcotics after inconclusive ultrasonography and MRI examinations. We present the noncontrast MRI features of adrenal infarction, which were identified on retrospective review. We hope that increased awareness of adrenal infarction as a cause of acute abdominal pain in pregnancy and improved knowledge of its MRI features may allow timely diagnosis of this pathology.
CASES A 20-year-old woman, gravida 1 para 0, at 27 4/7 weeks of gestation presented to our obstetrics and gynecology triage with sudden-onset postprandial right upper quadrant and right flank pain. She also had two bouts of nonbloody and nonbilious emesis. She was afebrile, slightly hypertensive, and had moderate right costovertebral angle and upper quadrant tenderness. No guarding, rebound, or fundal tenderness was noted. Pelvic examination revealed a long, closed cervix and normal fetal heart tracing. There were no contractions on tocodynamanometer. Laboratory values were notable only for a white blood cell count of 16.5 K per microliter and a urinalysis with 2+ glucose and 1+ leukocyte esterase. An abdominal ultrasonogram showed no significant abnormality but was limited by the patient’s body habitus and gravid state. Although the etiology of her pain was not clear, her symptoms were treated with 10 mg oral oxycodone and 1 mg intravenous (IV) hydromorphone. Because her symptoms were not relieved with this treatment, she was admitted and evaluated with MRI to exclude appendicitis with the following protocol: T1-weighted ultrafast gradient echo axial sequence (repetition time 3.43, echo time
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
775
1.61 ms, slice thickness: 3 mm), in- and out-of-phase T1weighted sequence (repetition time 6.87, echo time 4.76 ms, slice thickness 4 mm), and T2-weighted ultrafast spin echo sequence (repetition time 1,000, echo time 92 ms, slice thickness 5 mm). No IV contrast was administered given her pregnancy. On MRI, no evidence of appendicitis or other source of pain was noted. Therefore, persisting symptoms prompted abdominal CT with IV contrast, which showed a diffusely enlarged and hypoenhancing right adrenal gland surrounded by mild stranding and a small amount of retroperitoneal fluid with a normal-appearing left adrenal gland, consistent with right adrenal gland infarction. No other abnormality was present. As a result of the CT findings, a retrospective review of the MRI was performed and a small amount of retroperitoneal fluid around a diffusely enlarged and slightly T2-hypointense right adrenal gland was noted (Fig. 1). The left adrenal gland appeared normal in size and signal. Hypercoagulation factors including protein C and antithrombin III levels were within normal limits. She had a slightly low protein S level of 50% (reference 70–134%). Factor II was wild type. Antiphospholipid antibody workup was negative. The patient was started on a heparin drip, discharged on fondaparinux, and delivered a healthy 6-pound 9-ounce neonate at 39 4/7 weeks of gestation. She did not experience adrenal insufficiency. A 29-year-old woman, gravida 2 para 1, at 17 5/5 weeks of gestation presented to our emergency department with suddenonset severe right-sided abdominal and flank pain radiating to the back and scapula accompanied by nausea. She was afebrile and slightly hypotensive (82/47 mm Hg). Her examination was notable for epigastric and right upper quadrant tenderness without a Murphy sign. Laboratory values were notable for a white blood cell count of 10.8 K per microliter, D-dimer 846 ng/mL (normal range 110–250 ng/mL), and a urinalysis with 1+ ketones, six red blood cells, and 12 hyaline casts. An abdominal ultrasonogram was normal except for a trace amount of fluid seen in Morrison’s pouch. An MRI was performed with a similar protocol to the previous patient
with the addition of a T2-weighted fat saturated true fast imaging with steady-state precession (repetition time 379.09, echo time 1.42 ms, slice thickness 5 mm) sequence. MRI confirmed trace fluid in Morrison’s pouch (Fig. 2) and showed a 6-cm fibroid in the right uterine fundus. The symptoms were attributed to the uterine fibroid and she was treated with indomethacin and hydromorphone. She was discharged on hospital day 5 with improved pain control. At 35 5/7 weeks of gestation, she again presented to our obstetrics and gynecology triage with sudden-onset severe flank and upper quadrant pain but this time on the left side with similar clinical symptoms and physical examination findings. An abdominal ultrasonogram was normal. A repeat MRI was performed and the only notable finding was a small amount of perirenal fluid. The patient was admitted and, because the pain was exacerbated by breathing, a chest CT scan was performed the next day to evaluate for pulmonary embolism. Although negative for pulmonary embolism, mild left adrenal thickening and lack of adrenal enhancement were noted in addition to left perirenal edema that was most pronounced around the left adrenal gland, all consistent with left adrenal gland infarction. The right adrenal gland was normal in size and enhancement. Retrospective review of MRI revealed a small amount of retroperitoneal edema around a mildly enlarged left adrenal gland, which appeared slightly hypointense on T2-weighted images (Fig. 3). Retrospective review of the MRI from the prior episode also showed similar findings involving the right adrenal gland. The patient was started on a heparin drip. Hypercoagulation studies of activated protein C, protein C, and antithrombin III levels were within normal limits, whereas a protein S level of 41% (reference 70–134%) was slightly low. Factor VIII level was slightly high at 178% (reference 50–150%). Factor II and factor VIII were both wild type. Antiphospholipid antibody workup was negative. As a result of the patient’s persistent requirement of hydromorphone, an uneventful cesarean delivery was performed on hospital day 7 and a healthy 7 pound 1-ounce Fig. 1. A woman aged 20 years (gravida 1 para 0) at 27 4/7 weeks of gestation with sudden-onset right upper quadrant and right flank pain. A. Axial, T2-weighted, ultrafast spin echo magnetic resonance image shows increased signal intensity (white arrows), suggestive of edema, around the enlarged right adrenal gland (red arrows) and a normal left adrenal gland (blue arrow). B. Contrast-enhanced computed tomography image in the axial plane shows a diffusely enlarged and hypoattenuating right adrenal gland (red arrows) surrounded by mild stranding and a small amount of fluid (white arrow). Note a normal left adrenal gland (blue arrow). Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
776
Guenette and Tatli
Adrenal Infarction During Pregnancy
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
Fig. 2. A woman aged 29 years (gravida 2 para 1) at 17 5/7 weeks of gestation with sudden-onset severe right-sided abdominal and flank pain. A. Axial, T1-weighted noncontrast magnetic resonance image shows a diffusely enlarged right adrenal gland (red arrows). Note a normal left adrenal gland (blue arrow). B. Axial, T2-weighted, fat-saturated magnetic resonance image shows a region of increased retroperitoneal signal (white arrows) around the enlarged right adrenal gland (red arrows) and superior pole of the right kidney. Note a normal left adrenal gland (blue arrow). Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
neonate was delivered with Apgar scores of 6 and 8. The patient was discharged on enoxaparin for anticoagulation with resolved left upper quadrant pain. The patient did not experience adrenal insufficiency after either episode.
DISCUSSION Adrenal infarction is a rare clinical finding in pregnancy with descriptions limited to case reports and small series. The first report of adrenal infarction in
pregnancy dates to 1947.5 Since then, 16 cases have been reported. Only one of those cases occurred without hemorrhage.6 Pathology studies have demonstrated that both hemorrhagic and nonhemorrhagic adrenal infarction are secondary to adrenal vein thrombosis.7 Recognition of adrenal infarction on MRI in pregnant patients is particularly important because pregnant patients are not typically evaluated with CT as a result of radiation
Fig. 3. The same patient as in Figure 2 at 35 5/7 weeks of gestation with sudden-onset severe left flank pain. A. Axial, T2weighted, ultrafast spin echo magnetic resonance image shows prominent left adrenal gland (red arrow) with decreased T2 signal surrounded by increased signal intensity (white arrows) indicating local retroperitoneal edema. Note intrauterine fetus anteriorly. B. Pulmonary embolism protocol contrast-enhanced axial computed tomography image shows a diffusely enlarged, hypoattenuating left adrenal gland (red arrows) with a small amount of surrounding fluid (white arrows), consistent with adrenal infarction. Note a normal in size and normally attenuating right adrenal gland (blue arrows), which was involved in a previous episode. Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
VOL. 126, NO. 4, OCTOBER 2015
Guenette and Tatli
Adrenal Infarction During Pregnancy 777
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
risks to the fetus and because ultrasonography may be limited as a result of the challenging location of the gland, patient body habitus, and gravid uterus. In both patients presented here, MRI was performed when ultrasound findings were inconclusive and CT was then performed when MRI findings were inconclusive. Computed tomography imaging findings of acute nonhemorrhagic adrenal infarction have been described and include diffuse adrenal gland enlargement, hypoenhancement, and adjacent fat stranding and fluid in the retroperitoneum.1–3 Although these findings have not been correlated pathologically, development of acute adrenal insufficiency in patients with bilateral involvement provides physiologic confirmation of the diagnosis. Our patients did not develop adrenal insufficiency because only a single adrenal gland was infarcted during each episode. However, contrast-enhanced CT imaging features were identical to that of previously described cases and confirm the MRI features. We did not administer IV contrast (gadolinium) during MRI. If gadolinium had been given, it was likely that the adrenal glands could have been hypoenhancing relative to the normal side, which could have been prompted the diagnoses of acute adrenal gland infarction earlier. However, as a result of potential fetal toxicity, in current clinical practice, gadolinium is generally not used for MRI studies obtained during pregnancy. Therefore, nonenhanced MRI features, which we described here, become more important. Adrenal hemorrhage would typically present with T1 hyperintensity8 and a recent case report by Bockhorny et al9 provides a thorough discussion of adrenal hemorrhage in pregnancy. Adrenal neoplasms develop gradually and do not likely cause acute pain. The findings of chronic adrenal infarction are distinctly different than acute infarction and include shrunken and possibly calcified glands without surrounding edema or stranding.10 Management of adrenal infarction consists of narcotics for pain relief. Because adrenal infarction is caused by adrenal vein thrombosis7 and the resulting presumption that these patients may be at high risk of additional thrombotic events, hematology consultation and initiation of anticoagulation have been practiced. Conservative management without anticoagulation has been advocated based on a single conservatively managed case in which no further thrombotic events or adverse sequelae were identified.11 In one of our patients, the patient had metachronous bilateral adrenal gland infarctions with resolution of function and of normal imaging features of the first infarcted gland at
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the time of the contralateral infarction. A similar case has been previously reported.12 It is unclear whether recovery of gland function is a common feature after infarction. Regardless, the theoretical risk of additional thrombotic events, including metachronous contralateral adrenal vein thrombosis leading to acute adrenal insufficiency, certainly exists. Ultimately, no evidence exists at this time to definitively recommend either anticoagulation or conservative management. In summary, adrenal infarction should be on the obstetrician’s and radiologist’s differential when evaluating a pregnant patient with severe abdominal pain. Given the increasingly frequent use of noncontrast MRI during pregnancy, the adrenal glands should be carefully evaluated not only for hemorrhage, but also for the features of infarction, which include diffuse enlargement of the adrenal gland, decreased signal intensity on T2-weighted images, and edema in the adjacent retroperitoneum. REFERENCES 1. Ames DE, Asherson RA, Ayres B, Cassar J, Hughes GR. Bilateral adrenal infarction, hypoadrenalism and splinter haemorrhages in the “primary” antiphospholipid syndrome. Br J Rheumatol 1992; 31:117–20. 2. Thuerl C, Altehoefer C, Spyridonidis A, Laubenberger J. Imaging findings in the rare catastrophic variant of the primary antiphospholipid syndrome. Eur Radiol 2002;12:545–8. 3. Wheatley T, Gallagher S, Dixon AK. Adrenal insufficiency and bilateral adrenal enlargement: demonstration by computed tomography. Postgrad Med J 1985;61:435–8. 4. Riddell AM, Khalili K. Sequential adrenal infarction without MRI-detectable hemorrhage in primary antiphospholipidantibody syndrome. AJR Am J Roentgenol 2004;183:220–2. 5. Arnold G, Richer AG, Lepore JJ. Suprarenal hemorrhage in pregnancy; report of a case with a review of the literature. N Engl J Med 1949;240:1040–3. 6. Green PA, Ngai IM, Lee TT, Garry DJ. Unilateral adrenal infarction in pregnancy. BMJ Case Rep 2013;2013. pii:bcr2013009997. 7. Fox B. Venous infarction of the adrenal glands. J Pathol 1976; 119:65–89. 8. Hoeffel C, Legmann P, Luton JP, Chapuis Y, Fayet-Bonnin P. Spontaneous unilateral adrenal hemorrhage: computerized tomography and magnetic resonance imaging findings in 8 cases. J Urol 1995;154:1647–51. 9. Bockorny B, Posteraro A, Bilgrami S. Bilateral spontaneous adrenal hemorrhage during pregnancy. Obstet Gynecol 2012;120:377–81. 10. Doppman JL, Gill JR Jr, Nienhuis AW, Earll JM, Long JA Jr. CT findings in Addison’s disease. J Comput Assist Tomogr 1982;6:757–61. 11. Gavrilova-Jordan L, Edmister WB, Farrell MA, Watson WJ. Spontaneous adrenal hemorrhage during pregnancy: a review of the literature and a case report of successful conservative management. Obstet Gynecol Surv 2005;60:191–5. 12. Ryan MF, Murphy JP, Jay R, Callum J, MacDonald D. MRI diagnosis of bilateral adrenal vein thrombosis. Br J Radiol 2003;76:566–9.
Adrenal Infarction During Pregnancy
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
Nonhemorrhagic Adrenal Infarction With Magnetic Resonance Imaging Features During Pregnancy Jeffrey P. Guenette, MD, and Servet Tatli, MD BACKGROUND: Adrenal infarction is an infrequent cause of severe abdominal pain during pregnancy. The magnetic resonance imaging (MRI) features of adrenal infarction have not previously been thoroughly described. CASES: A 20-year-old woman, gravida 1 para 0, presented at 27 4/7 weeks of gestation with sudden-onset right upper quadrant and flank pain. A 29-year-old woman, gravida 2 para 1, presented at 17 5/7 weeks of gestation with suddenonset right abdominal and flank pain and again at 35 5/7 weeks of gestation with sudden-onset severe left flank and upper quadrant pain. In both patients, unilateral adrenal infarction was diagnosed on contrast-enhanced computed tomography after initial nondiagnostic ultrasonography and MRI. Clinical presentation and MRI features of nonhemorrhagic adrenal infarction are described. CONCLUSION: Nonhemorrhagic adrenal infarction may be an underdiagnosed cause of acute abdominal pain during pregnancy and can be diagnosed with MRI. (Obstet Gynecol 2015;126:775–8) DOI: 10.1097/AOG.0000000000000884
A
drenal infarction in pregnancy is a rare clinical entity. It can cause sudden, severe, unremitting upper right or left quadrant pain similar to other acute intraabdominal pathologies such as biliary colic, renal colic, and appendicitis. Diagnosis of nonhemorrhagic adrenal infarction can be made with computed tomography (CT),1–3 but, ideally, CT would be avoided in pregnant patients because it involves exposure of the fetus to radiation. Similarly, a single report suggests that it From the Brigham & Women’s Hospital, Department of Radiology, Harvard Medical School, Boston, Massachusetts. Corresponding author: Jeffrey P. Guenette, MD, Brigham & Women’s Hospital, Department of Radiology, Harvard Medical School, Boston, MA 02115; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2015 by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0029-7844/15
VOL. 126, NO. 4, OCTOBER 2015
Teaching Points 1. Consider adrenal infarction in the differential of acute severe upper abdominal pain during pregnancy. 2. Magnetic resonance imaging features of nonhemorrhagic adrenal infarction include diffuse enlargement of the adrenal gland, decreased signal intensity on T2-weighted images, and edema in the adjacent retroperitoneum. 3. Consider hematology evaluation in the setting of adrenal infarction.
may be possible to diagnose nonhemorrhagic adrenal infarction with contrast-enhanced magnetic resonance imaging (MRI),4 but MRI contrast is usually avoided in pregnant patients as a result of potential fetal toxicity. A search of MEDLINE (English language; 1966 to November 2014; search terms: “adrenal infarction” and “MRI”) revealed no thorough description of nonhemorrhagic adrenal infarction with noncontrast MRI. We present two patients with nonhemorrhagic adrenal infarction in pregnancy in the setting of severe pain refractory to narcotics after inconclusive ultrasonography and MRI examinations. We present the noncontrast MRI features of adrenal infarction, which were identified on retrospective review. We hope that increased awareness of adrenal infarction as a cause of acute abdominal pain in pregnancy and improved knowledge of its MRI features may allow timely diagnosis of this pathology.
CASES A 20-year-old woman, gravida 1 para 0, at 27 4/7 weeks of gestation presented to our obstetrics and gynecology triage with sudden-onset postprandial right upper quadrant and right flank pain. She also had two bouts of nonbloody and nonbilious emesis. She was afebrile, slightly hypertensive, and had moderate right costovertebral angle and upper quadrant tenderness. No guarding, rebound, or fundal tenderness was noted. Pelvic examination revealed a long, closed cervix and normal fetal heart tracing. There were no contractions on tocodynamanometer. Laboratory values were notable only for a white blood cell count of 16.5 K per microliter and a urinalysis with 2+ glucose and 1+ leukocyte esterase. An abdominal ultrasonogram showed no significant abnormality but was limited by the patient’s body habitus and gravid state. Although the etiology of her pain was not clear, her symptoms were treated with 10 mg oral oxycodone and 1 mg intravenous (IV) hydromorphone. Because her symptoms were not relieved with this treatment, she was admitted and evaluated with MRI to exclude appendicitis with the following protocol: T1-weighted ultrafast gradient echo axial sequence (repetition time 3.43, echo time
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
775
1.61 ms, slice thickness: 3 mm), in- and out-of-phase T1weighted sequence (repetition time 6.87, echo time 4.76 ms, slice thickness 4 mm), and T2-weighted ultrafast spin echo sequence (repetition time 1,000, echo time 92 ms, slice thickness 5 mm). No IV contrast was administered given her pregnancy. On MRI, no evidence of appendicitis or other source of pain was noted. Therefore, persisting symptoms prompted abdominal CT with IV contrast, which showed a diffusely enlarged and hypoenhancing right adrenal gland surrounded by mild stranding and a small amount of retroperitoneal fluid with a normal-appearing left adrenal gland, consistent with right adrenal gland infarction. No other abnormality was present. As a result of the CT findings, a retrospective review of the MRI was performed and a small amount of retroperitoneal fluid around a diffusely enlarged and slightly T2-hypointense right adrenal gland was noted (Fig. 1). The left adrenal gland appeared normal in size and signal. Hypercoagulation factors including protein C and antithrombin III levels were within normal limits. She had a slightly low protein S level of 50% (reference 70–134%). Factor II was wild type. Antiphospholipid antibody workup was negative. The patient was started on a heparin drip, discharged on fondaparinux, and delivered a healthy 6-pound 9-ounce neonate at 39 4/7 weeks of gestation. She did not experience adrenal insufficiency. A 29-year-old woman, gravida 2 para 1, at 17 5/5 weeks of gestation presented to our emergency department with suddenonset severe right-sided abdominal and flank pain radiating to the back and scapula accompanied by nausea. She was afebrile and slightly hypotensive (82/47 mm Hg). Her examination was notable for epigastric and right upper quadrant tenderness without a Murphy sign. Laboratory values were notable for a white blood cell count of 10.8 K per microliter, D-dimer 846 ng/mL (normal range 110–250 ng/mL), and a urinalysis with 1+ ketones, six red blood cells, and 12 hyaline casts. An abdominal ultrasonogram was normal except for a trace amount of fluid seen in Morrison’s pouch. An MRI was performed with a similar protocol to the previous patient
with the addition of a T2-weighted fat saturated true fast imaging with steady-state precession (repetition time 379.09, echo time 1.42 ms, slice thickness 5 mm) sequence. MRI confirmed trace fluid in Morrison’s pouch (Fig. 2) and showed a 6-cm fibroid in the right uterine fundus. The symptoms were attributed to the uterine fibroid and she was treated with indomethacin and hydromorphone. She was discharged on hospital day 5 with improved pain control. At 35 5/7 weeks of gestation, she again presented to our obstetrics and gynecology triage with sudden-onset severe flank and upper quadrant pain but this time on the left side with similar clinical symptoms and physical examination findings. An abdominal ultrasonogram was normal. A repeat MRI was performed and the only notable finding was a small amount of perirenal fluid. The patient was admitted and, because the pain was exacerbated by breathing, a chest CT scan was performed the next day to evaluate for pulmonary embolism. Although negative for pulmonary embolism, mild left adrenal thickening and lack of adrenal enhancement were noted in addition to left perirenal edema that was most pronounced around the left adrenal gland, all consistent with left adrenal gland infarction. The right adrenal gland was normal in size and enhancement. Retrospective review of MRI revealed a small amount of retroperitoneal edema around a mildly enlarged left adrenal gland, which appeared slightly hypointense on T2-weighted images (Fig. 3). Retrospective review of the MRI from the prior episode also showed similar findings involving the right adrenal gland. The patient was started on a heparin drip. Hypercoagulation studies of activated protein C, protein C, and antithrombin III levels were within normal limits, whereas a protein S level of 41% (reference 70–134%) was slightly low. Factor VIII level was slightly high at 178% (reference 50–150%). Factor II and factor VIII were both wild type. Antiphospholipid antibody workup was negative. As a result of the patient’s persistent requirement of hydromorphone, an uneventful cesarean delivery was performed on hospital day 7 and a healthy 7 pound 1-ounce Fig. 1. A woman aged 20 years (gravida 1 para 0) at 27 4/7 weeks of gestation with sudden-onset right upper quadrant and right flank pain. A. Axial, T2-weighted, ultrafast spin echo magnetic resonance image shows increased signal intensity (white arrows), suggestive of edema, around the enlarged right adrenal gland (red arrows) and a normal left adrenal gland (blue arrow). B. Contrast-enhanced computed tomography image in the axial plane shows a diffusely enlarged and hypoattenuating right adrenal gland (red arrows) surrounded by mild stranding and a small amount of fluid (white arrow). Note a normal left adrenal gland (blue arrow). Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
776
Guenette and Tatli
Adrenal Infarction During Pregnancy
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
Fig. 2. A woman aged 29 years (gravida 2 para 1) at 17 5/7 weeks of gestation with sudden-onset severe right-sided abdominal and flank pain. A. Axial, T1-weighted noncontrast magnetic resonance image shows a diffusely enlarged right adrenal gland (red arrows). Note a normal left adrenal gland (blue arrow). B. Axial, T2-weighted, fat-saturated magnetic resonance image shows a region of increased retroperitoneal signal (white arrows) around the enlarged right adrenal gland (red arrows) and superior pole of the right kidney. Note a normal left adrenal gland (blue arrow). Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
neonate was delivered with Apgar scores of 6 and 8. The patient was discharged on enoxaparin for anticoagulation with resolved left upper quadrant pain. The patient did not experience adrenal insufficiency after either episode.
DISCUSSION Adrenal infarction is a rare clinical finding in pregnancy with descriptions limited to case reports and small series. The first report of adrenal infarction in
pregnancy dates to 1947.5 Since then, 16 cases have been reported. Only one of those cases occurred without hemorrhage.6 Pathology studies have demonstrated that both hemorrhagic and nonhemorrhagic adrenal infarction are secondary to adrenal vein thrombosis.7 Recognition of adrenal infarction on MRI in pregnant patients is particularly important because pregnant patients are not typically evaluated with CT as a result of radiation
Fig. 3. The same patient as in Figure 2 at 35 5/7 weeks of gestation with sudden-onset severe left flank pain. A. Axial, T2weighted, ultrafast spin echo magnetic resonance image shows prominent left adrenal gland (red arrow) with decreased T2 signal surrounded by increased signal intensity (white arrows) indicating local retroperitoneal edema. Note intrauterine fetus anteriorly. B. Pulmonary embolism protocol contrast-enhanced axial computed tomography image shows a diffusely enlarged, hypoattenuating left adrenal gland (red arrows) with a small amount of surrounding fluid (white arrows), consistent with adrenal infarction. Note a normal in size and normally attenuating right adrenal gland (blue arrows), which was involved in a previous episode. Guenette. Adrenal Infarction During Pregnancy. Obstet Gynecol 2015.
VOL. 126, NO. 4, OCTOBER 2015
Guenette and Tatli
Adrenal Infarction During Pregnancy 777
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
risks to the fetus and because ultrasonography may be limited as a result of the challenging location of the gland, patient body habitus, and gravid uterus. In both patients presented here, MRI was performed when ultrasound findings were inconclusive and CT was then performed when MRI findings were inconclusive. Computed tomography imaging findings of acute nonhemorrhagic adrenal infarction have been described and include diffuse adrenal gland enlargement, hypoenhancement, and adjacent fat stranding and fluid in the retroperitoneum.1–3 Although these findings have not been correlated pathologically, development of acute adrenal insufficiency in patients with bilateral involvement provides physiologic confirmation of the diagnosis. Our patients did not develop adrenal insufficiency because only a single adrenal gland was infarcted during each episode. However, contrast-enhanced CT imaging features were identical to that of previously described cases and confirm the MRI features. We did not administer IV contrast (gadolinium) during MRI. If gadolinium had been given, it was likely that the adrenal glands could have been hypoenhancing relative to the normal side, which could have been prompted the diagnoses of acute adrenal gland infarction earlier. However, as a result of potential fetal toxicity, in current clinical practice, gadolinium is generally not used for MRI studies obtained during pregnancy. Therefore, nonenhanced MRI features, which we described here, become more important. Adrenal hemorrhage would typically present with T1 hyperintensity8 and a recent case report by Bockhorny et al9 provides a thorough discussion of adrenal hemorrhage in pregnancy. Adrenal neoplasms develop gradually and do not likely cause acute pain. The findings of chronic adrenal infarction are distinctly different than acute infarction and include shrunken and possibly calcified glands without surrounding edema or stranding.10 Management of adrenal infarction consists of narcotics for pain relief. Because adrenal infarction is caused by adrenal vein thrombosis7 and the resulting presumption that these patients may be at high risk of additional thrombotic events, hematology consultation and initiation of anticoagulation have been practiced. Conservative management without anticoagulation has been advocated based on a single conservatively managed case in which no further thrombotic events or adverse sequelae were identified.11 In one of our patients, the patient had metachronous bilateral adrenal gland infarctions with resolution of function and of normal imaging features of the first infarcted gland at
778
Guenette and Tatli
the time of the contralateral infarction. A similar case has been previously reported.12 It is unclear whether recovery of gland function is a common feature after infarction. Regardless, the theoretical risk of additional thrombotic events, including metachronous contralateral adrenal vein thrombosis leading to acute adrenal insufficiency, certainly exists. Ultimately, no evidence exists at this time to definitively recommend either anticoagulation or conservative management. In summary, adrenal infarction should be on the obstetrician’s and radiologist’s differential when evaluating a pregnant patient with severe abdominal pain. Given the increasingly frequent use of noncontrast MRI during pregnancy, the adrenal glands should be carefully evaluated not only for hemorrhage, but also for the features of infarction, which include diffuse enlargement of the adrenal gland, decreased signal intensity on T2-weighted images, and edema in the adjacent retroperitoneum. REFERENCES 1. Ames DE, Asherson RA, Ayres B, Cassar J, Hughes GR. Bilateral adrenal infarction, hypoadrenalism and splinter haemorrhages in the “primary” antiphospholipid syndrome. Br J Rheumatol 1992; 31:117–20. 2. Thuerl C, Altehoefer C, Spyridonidis A, Laubenberger J. Imaging findings in the rare catastrophic variant of the primary antiphospholipid syndrome. Eur Radiol 2002;12:545–8. 3. Wheatley T, Gallagher S, Dixon AK. Adrenal insufficiency and bilateral adrenal enlargement: demonstration by computed tomography. Postgrad Med J 1985;61:435–8. 4. Riddell AM, Khalili K. Sequential adrenal infarction without MRI-detectable hemorrhage in primary antiphospholipidantibody syndrome. AJR Am J Roentgenol 2004;183:220–2. 5. Arnold G, Richer AG, Lepore JJ. Suprarenal hemorrhage in pregnancy; report of a case with a review of the literature. N Engl J Med 1949;240:1040–3. 6. Green PA, Ngai IM, Lee TT, Garry DJ. Unilateral adrenal infarction in pregnancy. BMJ Case Rep 2013;2013. pii:bcr2013009997. 7. Fox B. Venous infarction of the adrenal glands. J Pathol 1976; 119:65–89. 8. Hoeffel C, Legmann P, Luton JP, Chapuis Y, Fayet-Bonnin P. Spontaneous unilateral adrenal hemorrhage: computerized tomography and magnetic resonance imaging findings in 8 cases. J Urol 1995;154:1647–51. 9. Bockorny B, Posteraro A, Bilgrami S. Bilateral spontaneous adrenal hemorrhage during pregnancy. Obstet Gynecol 2012;120:377–81. 10. Doppman JL, Gill JR Jr, Nienhuis AW, Earll JM, Long JA Jr. CT findings in Addison’s disease. J Comput Assist Tomogr 1982;6:757–61. 11. Gavrilova-Jordan L, Edmister WB, Farrell MA, Watson WJ. Spontaneous adrenal hemorrhage during pregnancy: a review of the literature and a case report of successful conservative management. Obstet Gynecol Surv 2005;60:191–5. 12. Ryan MF, Murphy JP, Jay R, Callum J, MacDonald D. MRI diagnosis of bilateral adrenal vein thrombosis. Br J Radiol 2003;76:566–9.
Adrenal Infarction During Pregnancy
OBSTETRICS & GYNECOLOGY
Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
