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(Letters to the Editor are welcomed. They may report new clinical or laboratory observations and new developments in medical care or may contain comments on recent contents of the Journal. They will be published, if found suitable, as space permits. Like other material submitted for publication, letters must be typewritten, double-spaced, and submitted in duplicate. They must not exceed two typewritten pages in length. No more than five references and one figure or table may be used. See "Information for Authors" for format of references, tables, and figures. Editing, possible abridgment, and acceptance remain the prerogative of the Editors.)

Recurrence of Bacterial Meningitis in a Patient With Crouzon Syndrome and Pseudomeningocele

To the Editor. Crouzon syndrome is an autosomal dominant disease associated with a mutation in the fibroblast growth-factor receptor-2 gene; it is characterized by shallow orbits, premature craniosynostosis, maxillary hypoplasia, and growth hormone deficiency. (1-3) Repeated reconstructive surgery is needed to repair congenital craniofacial abnormalities as the patient grows. The development of a pseudomeningocele is an unusual complication of craniofacial surgery. (4) In this report, we present the first case of Crouzon syndrome with a postoperative pseudomeningocele and purulent meningitis.

The patient was a 25-year-old Japanese man. His parents had no apparent signs of Crouzon syndrome, but he had shown signs of the syndrome since birth. Skull radiographs were used to diagnose Crouzon syndrome. His stature was within normal limits. Four separate reconstructive craniofacial surgeries had been performed as the patient grew. The day before admission to our hospital, he complained of headache, nausea, and high fever. These symptoms worsened suddenly, and his level of consciousness decreased. He was admitted to another hospital and received antibiotic therapy. The next morning, when he was transferred to our hospital, his consciousness level was 3 on the Glasgow Coma Scale. Physical examination revealed an absent light reflex, normal-sized pupils, dilated superficial vein of the head, neck stiffness, and severe exophthalmos, especially on left. The patient's temperature was 39[degrees]C, blood pressure was 134/80 mm Hg, heart rate was 108/mm, and respiratory rate was 17/mm. Laboratory data on admiss ion revealed the following values: white blood cell count, 32,900/[micro]L with 89% neutrophils; sodium, 126 mEq/L; potassium, 4.4 mEq/L; antidiuretic hormone, 4.0 pg/mL; and C-reactive protein, 34.6 mg/dL. Aspertate aminotransferase, alanine aminotransferase, blood urea nitrogen, and creatinine levels were all within normal limits. Analysis of his cerebrospinal fluid (CSF) showed the following values: neutrophils, 11,532 /mL; pressure, >400 mm [H.sub.2]O; protein concentration, 392 mg/dL; glucose concentration, 0. Bacteria were not detected in the CSF, urine, or blood, probably due to antibiotic therapy administered before he was admitted to our hospital. Changes in the cranial bones and bilateral hydrocephalus were detected by computed tomography.

The patient was intubated due to his comatose state. The diagnoses of serious bacterial meningitis and inappropriate secretion of antidiuretic hormone were made. Cerebrospinal fluid was drained, and the patient was treated with intrathecal gentamicin (10 mg/day) and intravenous cefotaxime sodium (3 g/day). The patient's level of consciousness increased, and his symptoms gradually improved. White blood cell count, cerebrospinal fluid analysis, and C-reactive protein level were all normal by the 24th hospital day.

The patient was briefly discharged on 42nd hospital day, but he again complained of headache, nausea, and fever, and immediately returned to our hospital. Acute bacterial meningitis was diagnosed on the basis of CSF findings, and antibiotic therapy was started without CSF drainage. All of his symptoms and signs improved within 5 days of the initiation of therapy. Magnetic resonance imaging (MRI) of the head was performed to determine the cause of his recurrent meningitis; 2 giant pseudomeningoceles were detected, 1 in the left postorbital space and 1 in the right ethmoidal sinus (Figure). We speculated that the infection was induced by unidentified bacteria, which invaded through the ethmoidal pseudomeningocele. The orbital pseudomeningocele could cause severe exophthalmos on the left side. Additional resection of the pseudomeningocele with cranial reconstruction was performed. More than 2 years after that surgery, the patient has had no recurrence of his bacterial infection.

Postoperative pseudomeningocele is an unusual complication of craniofacial surgery in congenital craniosynostosis. (4) To our knowledge, Crouzon syndrome in conjunction with an infected pseudomeningocele has not previously been reported. To accomodate his growth, our patient had undergone 4 surgeries for cranial reconstruction, which probably caused the pseudomeningocele in his cranial base. In addition, when he had bacterial meningitis twice within 2 months, it was necessary to investigate the cause of his susceptibility to bacterial infection. As suspected, pseudomeningoceles were detected by cerebral MRI, and resection of the meningoceles protected him from a recurrence of acute bacterial meningitis.

Our case shows that prompt diagnosis and treatment, including drainage of CSF and antibiotic therapy, are extremely effective and shorten the treatment period in a patient with meningitis caused by a pseudomeningocele. Because cefotaxime sodium penetrates into the CSF, (5) the drug is useful for the treatment of severe meningitis. If meningitis recurs after only a short time, coexistence of a pseudomeningocele should be suspected.

Masami Ohmori, MD, PhD

Hitoshi Karnio, MD

Koh-ichi Inokuchi, MD

Haruhiko Tsutsumi, MD, PhD

Advanced Tertiary Emergency Medical Center

Saitama Medical Center

Saitama 350-8550, Japan

Akio Fujimura, MD, PhD

Department of Clinical Pharmacology

Jichi Medical School

Tochigi 329-0498, Japan


(1.) Reardlon W, Wintcr RM, Ruthland P, et al: Mutations in the fibroblast growth factor receptor 2 gene cause Crouzon syndrome. Nat Genet 1994; 8:98-103

(2.) Neilson KM, Friesel RE: Constitutive activation of fibroblast growth factor receptor-2 by a point mutation associated with Crouzon syndrome. J Biol Chem 1995; 44:26037-26040

(3.) Field CR, Leiber A, Toniges C: Crouzon syndrome with short stature. Am J Med Sci 1991; 302:101-102

(4.) Elias DL, Kawamoto HK Jr: Psetidomeningocele: an unusual complication of craniofacial surgery. Plast Reconstr Surg 1992; 90:484-486

(5.) Sato M, Echizenya K: Studies on the transfer of cefotaxime into the cerebrospinal fluid in the field in surgery. Recent Advances in Chemotherapy: Proceedings of the 14th International Congress of Chemotherapy. Ishigami J (ed). Tokyo, Japan, University of Tokyo Press, Vol 2, 1985, pp 1042-1043

Lidocaine Patch 5% and the Management of Chronic Pain *

To the Editor: In the October 2000 issue of the Southern Medical Journal, Khouzam (1) provided a general review of current approaches to the management of chronic pain in the primary care setting. In my practice at a pain clinic, I see many patients who require the types of interdisciplinary intervention and multiple treatment modalities described by Dr. Khouzam. I would add a relatively new modality, the topical lidocaine patch 5% (Lidoderm), to the list of treatments that could be useful in managing certain chronic neuropathic pain states encountered in primary care practice. (2) For many patients, the lidocaine patch is exceptionally effective in the treatment of postherpetic neuralgia (P1-IN), for which it is indicated in the United States. (3,4) This patch has the additional benefits of limiting drug exposure to only the local treatment area and providing a physical barrier that increases the comfort of patients with allodynia, the painfully heightened sensitivity of skin that can cause substantial disru ption in patients' lives.

Successful clinical application of the lidocaine patch to types of chronic pain other than P1-IN have recently been described. (5) At our pain center, we have broadened our use of this agent to treat patients with complaints of chronic low back pain, cervical spine pain, and myofascial pain. Each of these painful conditions is obviously relevant to a primary care practice. Our experience with the lidocaine patch has shown us that this agent has the potential to fill important roles in the management of PHN, as well as other common chronic pain states. Of course, prospective, well-controlled clinical trials will be required to assess whether these and other potential indications are effectively treated with the lidocaine patch. Nonetheless, currently available clinical information suggests that the lidocaine patch could be considered in any discussion of the management of chronic pain in primary care practice.

Charles E. Argoff, MD

Cohn Pain Management Center

North Shore University Hospital at Syosset

22l Jericho Turn Pike

1st Floor West Wing

Syosset, NY 11791


(1.) Khouzam HR: Chronic pain and its management in primary care. South Med J 2000; 93:946-952

(2.) Argoff CE: New analgesics for neuropathic pain: the lidocaine patch. Clin J Pain 2000; 16 (suppl 2):S62-S66

(3.) Galer BS, Rowbotham MC, Perander J, et al: Topical lidocaine patch relieves postherpetic neuralgia more effectively than a vehicle topical patch: results of an enriched enrollment study. Pain 1999; 80:533-538

(4.) Rowbotham MC, Davies PS, Verkempinck C, et al: Lidocaine patch: double-blind controlled study of a new treatment method for post-herpetic neuralgia Pain 1996; 65:3944

(5.) Devers A, Galer BS: Topical lidocaine patch relieves a variety of neuropathic pain conditions: an open-label study. ClinJPain 2000; 16:205-208

Paraganglioma of the Organ of Zuckerkandi With Electrocardiographic Changes Mimicking Acute Myocardial Infarction During the Early Postoperative Period

To the Editor: Few cases of tumors of the organ of Zuckerkandl with electrocardiogram (ECG) patterns suggestive of acute myocardial infarction related to the effect of excess circulating catecholamines are described in the literature. (1) We present a case of a 41-year-old black woman with a history of asthma, migraines, and sustained hypertension. Computed tomography (CT) of the abdomen showed a 3.6 x 2.7 cm mass immediately inferior to the duodenum at the aortic bifurcation. Metaidobenzylguanidine (MIBG) scan showed abnormal activity just to the right of the low thoracic or upper lumbar spine region. Clinical laboratory analysis revealed the following values: serum dopamine, 101 pg/mL (normal, 100 to 440 pg/mL); epinephrine, 4 pg/mL (normal, <15 pg/mL), norepinephrine, 96 pg/mL (normal, 11 to 86 pg/mL); total free catecholamines, 201 pg/mL (normal <540 pg/mL); urine metanephrine, 0.4 mg/24 hrs (normal, <1.3 mg/24 hrs). These findings were consistent with the diagnosis of extra-adrenal retroperitoneal paraga nglioma. She had resection of the tumor, which was located just anterior to the aorta near the origin of the inferior mesenteric artery. The maximal blood pressure level observed during the surgery was 180/110 mm 1-1g. Grossly, the tumor was well circumscribed, weighed 23.2 grams, and measured 4.5 x 4.0 x 3.1 cm. It was brownish tan, well vascularized, and encapsulated, with a rubbery consistency. The tumor was characterized microscopically by proliferation of a homogenous population of round to oval cells, with eosinophilic, granular cytoplasm containing a round, slightly vesicular nucleus with a single nucleolus. The histopathologic findings were consistent with the diagnosis of extra-adrenal paraganglioma of the organ of Zuckerkandl.

On the evening of the first postoperative day, the patient had midsternal chest pain that radiated down her right arm. Cardiac enzyme levels were markedly elevated: creatinine kinase (CK), 4,444 U/L (normal, 25 to 190 U/L); CKMB, 31.2 ng/mL (normal, <5.0 ng/mL); troponin I, 92.5 ng/mL (normal, <0.4 ng/mL). Electrocardiogram showed S-T elevation in leads II, III, and [aV.sub.f] consistent with inferior injury or acute infarct. Electrocardiogram changes suggestive of myocardial infarction reverted to normal within 3 hours. A MIBI scan with technetium Tc 99m showed normal myocardial perfusion. The patient was successfully managed by a cardiology team and was referred to her cardiologist for follow-up after discharge.

The area known as the organ of Zuckerkandl encompasses all extra-adrenal chromaffin tissue near the origin of the inferior mesenteric artery and the aortic bifurcation. The symptoms of a tumor arising in the organ of Zuckerkandl may be related either to excessive catecholamine secretion or to the growth of a retroperitoneal mass. Catecholamines, particularly norepinephrine, are known to have a toxic effect on the myocardium, which may account for such cardiovascular manifestations as myocardial infarction, serious arrhythmias, congestive heart failure, cardiomyopathy (active catecholamine myocarditis), dissecting aneurysm, and sudden death. A classic pathologic feature most commonly observed in a heart exposed to the toxic effects of catecholamine is myccardial contraction-band necrosis, resulting in cell death due to cytosolic calcium overload. The reported ECG abnormalities associated with pheochromocytoma include left-ventricular-hypertrophy pattern, arrhythmias (sometimes occurring without hypertension an d likely to be induced by exertion), variable and transient repolarization disturbances, and hemodynamic manifestations of myocardial involvement (catecholamine cardiomyopathy). (4) None of the ECG changes described are specific, but when they occur in association with the onset of symptoms and signs of increased circulating catecholamines, they acquire greater diagnostic significance. Particularly interesting ECG patterns, consistent with those observed in acute myocardial infarctions, were reported in some patients with pheochromocytoma. (2-5) These ECG changes were probably the result of cardiotoxic effects of excess circulating catecholamines.

We presented a case of extra-adrenal norepinephrine-secreting paraganglioma of the organ of Zuckerkandl in a patient who had surgical removal of the tumor with subsequent development of symptoms and ECG changes mimicking those of myocardial infarction. The surgical procedure may have resulted in massive mobilization of catecholamines that caused damage to the myocardium, with ECG pattern simulating acute inferior myocardial infarction. Direct relationship with the surgical procedure, resulting in massive influx of catecholamines in circulation and elevation of blood pressure; rapid stabilization of the clinical changes and ECG changes with adrenergic blockade; and normal perfusion MIBI scan favored the diagnosis of a catecholamine-induced myocardial damage.

Acute catecholamine secretion may induce chest pain and abnormal ECG changes, mimicking an ischemic episode. Catecholamine-induced myocardial damage should, therefore, be included in the differential diagnosis of acute coronary syndrome occurring in the immediate postoperative period in patients with norepinephrine-producing tumors of the organ of Zuckerkandl.

Irma Mikolaenko, MD

Michael G. Conner, MD

Division of Anatomic Pathology

University of Alabama at Birmingham

506 Kracke Bldg

619 S 19th St

Birmingham, AL 35233-6823


(1.) Costa J, Brandao A, Coreira MA, et al: Extra-adrenal pheochromocytoma simulating acute myocardial infarction. Rev Port Cardiol 1999; 18:1025-1029

(2.) Haas CJ, Tzagournis M, Boudoulas H: Pheochromocytoma: catecholamine-mediated electrocardiographic changes mimicking ischemia. Am Heart J 1988, 116:1363-1365

(3.) Murai K, Hirota K, Niskikimi T, et al: Pheochromocytoma with electrocardiographic change mimicking angina pectoris, and cyclic change in direct arterial pressure. a case report. Angiology 1991; 42:157-161

(4.) Shub C, Cueto-Garcia L, Sheps SG, et al: Echocardiographic finding in pheochromocytoma, Am J Cardiol 1986; 57:971-975

(5.) Cheng TO, Bashour TT: Striking electrocardiographic changes associated with pheochromocytoma masquerading as ischemic heart disease. Chest 1976; 70:397-399

Staphylococcus aureus Bacteremia in Patients With Neutropenia *

To the Editor: Staphylococcus aureus is a common but serious pathogen in both neutropenic and nonneutropenic patients, (1) and the incidence of S aureus bacteremia (SAB) has been steadily increasing over the past 15 years among neutropenic patients. Immunocompromised patients, (1) particularly neutropenic patients, (2,3) may be especially susceptible to complications of SAB.

Patients in this retrospective cohort analysis were adults admitted to Duke University Medical Center between September 1994 and February 1999, diagnosed with SAB within 48 hours of having an absolute neutrophil count (ANC) < 1000 cells/[mm.sup.3]. The control group consisted of a prospectively collected cohort of 525 consecutive nonneutropenic patients hospitalized over the same period. We reviewed the patients' medical records to obtain baseline data regarding demographics, immune function, the course of infection, and death or recurrent infection. We defined a clinically significant episode of SAB as more than 1 blood culture positive for S aureus, or a single blood culture yielding S aureus in a patient with clinical evidence of infection. The primary outcome in our study was an adverse event, which we defined as recurrent S aureus infection, or death from any cause within 12 weeks of the onset of SAB. We also evaluated the clinical characteristics of SAB that were associated with adverse events in neutro penic patients. Statistical significance was determined using Fisher exact test, logistic regression, and Kaplan Meier analysis, as appropriate.

We identified 60 neutropenic patients with SAB during our study period. Fifty-five (91.7%) patients were neutropenic as a result of chemotherapy for malignancies, including leukemia (n = 17), lymphoma (n = 14), breast cancer (n = 7), lung cancer (n = 3), melanoma (n= 3), and other miscellaneous malignancies (n = 11). The other causes of neutropenia included immunosuppressive therapy for organ transplantation, treatment of neurosarcoidosis, and bone marrow suppression secondary to malnutrition and sepsis. Neutropenic patients were significantly more likely than nonneutropenic patients to be white, receiving corticosteroid therapy, and non-hemodialysis-dependent. At the time of first positive blood culture for S aureus in the neutropenic group, the median ANG was 600 cells/[ mm.sup.3] (interquartile range, 100-2700 cells/[ mm.sup.3]). The median duration of neutropenia (ANC < 1000 cells/[ mm.sup.3]) was 8 days (interquartile range, 3-16 days).

Neutropenic patients experienced a higher mortality rate (44.7% vs 22.1%; P < .01) and had a higher rate of adverse events (55.3% vs 31.5%; P < .01) than nonneutropenic patients, and these findings persisted after adjusting for diabetes mellitus diagnosis, hemodialysis dependence, and steroid use. Forty-five percent (95% confidence interval [ CI] = 0.30-0.59) of neutropenic patients remained free of adverse events for 90 days, compared with 71% (95% CI = 0.67-0.75) of the nonneutropenic group (P < .001) (Figure).

Neutropenic patients with methicillin-resistant S aureus (MRSA) bacteremia were 3.91 (95% CI = 1.30-11.74) times more likely than nonneutropenic patients to have an adverse event within 12 weeks of the onset of bacteremia (P = .02). Sources of infection that were non-removable (odds ratio [ OR] = 15.00; 95% CI = 3.44-65.36; P < .01) or unknown (OR = 10.13; 95% CI = 1.77-57.91; P = .01) were also associated with adverse events. Each of these variables was independently associated with adverse events and showed no confounding influence when analyzed together. Furthermore, these variables remained significant when controlling separately for age, sex, and race. Failure of the neutrophil count to recover to > 500 cells/[ mm.sup.3] was also associated with adverse events (P < .0001). The type of cancer did not correlate with adverse events.

Our study has yielded several important observations. Neutropenic patients with SAB have a significantly worse clinical outcome than nonneutropenic patients with SAB. Within the neutropenic cohort, risk factors associated with adverse events included MRSA infection, non-catheter-related infections, and failure of neutrophil counts to recover. Among neutropenic patients with SAB, those whose ANC rose above 500 cells/[ mm.sup.3] were significantly less likely to die or develop recurrent staphylococcal infections than were those whose ANC did not recover. These findings underscore the critical role of neutrophils in host defense against bacterial infection, (1) and are consistent with findings in murine models of granulocytopenic hosts infected with S aureus. (2) The important role of recombinant granulocyte colony-stimulating factor (C-CSF) in the management of neutropenic patients is also highlighted by these findings. When given to neutropenic patients, C-CSF increases the number of circulating host neutroph ils, improves the microbicidal activity of these neutrophils, improves the clinical response to antibiotic therapy, and reduces the associated mortality rate. (3)

In our study, S aureus bacteremia from a noncatheter source was associated with adverse events in neutropenic patients. This is consistent with the findings of previous investigators, and also applies to patients who are not immunocompromised. For example, Lautenschlager et a1 (4) found a higher mortality rate among nonneutropenic patients with complicated deep-tissue SAB (40%) than among those with uncomplicated SAB (24%; P< .01). The data indicate that both neutropenic and nonneutropenic patients with deep-tissue, nonremovable, or unknown sources of S aureus infection have a worse prognosis than patients with intravascular, catheter-associated SAB.

Finally, we found that neutropenic patients with MRSA bacteremia had higher rates of mortality from all causes and of recurrent staphylococcal infection than those without MRSA infection. This association of MRSA bacteremia with a higher all-cause mortality rate is likely due to the fact that MRSA infection is primarily acquired by chronically ill, hospitalized patients with multiple comorbid conditions and a poor overall prognosis. Reliance upon vancomycin therapy for the treatment of this infection might also contribute to higher complication rates among MRSA-infected patients. As an antistaphylococcal agent, vancomycin has been associated with a high clinical failure rate, (5) especially in the presence of undrained foci of infection or foreign bodies.

We have demonstrated that, in patients with SAB, neutropenic patients have significantly worse outcomes than nonneutropenic patients, and that identifiable clinical characteristics are associated with this poor clinical outcome. Treating clinicians should aggressively manage neutropenic patients with deep-tissue or unknown sources of SAB in order to minimize the chance of metastatic infection and death.

Manish A. Shah, MD

Linda Sanders, MPH

Kevin Lanclos, MD

Anna Lisa Chamis, MD

Department of Medicine

Jon Gockerman, MD

Division of Hematology/Oncology

Dannah Wray, MD

G. Ralph Corey, MD

Vance G. Fowler, Jr., MD

Division of Infectious Diseases

Duke University Medical Center

Box 3281

Durham, NC 27710


(1.) Kuritzkes DR: Neutropenia, neutrophil dysfunction, and bacterial infection in patients with human immunodeficiency virus disease: the role of granulocyte colony-stimulating factor. Clin Infect Dis 2000; 30:256-260

(2.) Verdrengh M, Tarkowski A: Role of neutrophils in experimental septicemia and septic arthritis induced by Staphylococcus aureus. Infect Immun 1997; 65:2517-2521

(3.) Aviles A, Guzman R, Garcia EL, et al: Results of a randomized trial of granulocyte colony-stimulating factor in patients with infection and severe granulocytopenia. Anticanncer Drugs 1996; 7:392-397

(4.) Lautenschlager S, Herzog C, Zimmerli W: Course and outcome of bacteremia due to Staphylococcus aureus. evaluation of different clinical case definitions. Clin Infect Dis 1993;16:567-573

(5.) Wong SA, Ho PL, Woo PC, et al: Bacteremia caused by staphylococci with inducible vancomycin heteroresistance. Clin Infect Dis 1999; 29:760-767

Incidental Finding of Bilateral Adrenal Calcification in a Patient With Congenital Heart Disease

To the Editor. We treated a patient with severe valvular aortic stenosis and coarctation who incidentally was found to have bilateral adrenal calcification. A 3-month-old male was admitted to the hospital with respiratory difficulty and easy fatigability with feeding, which started soon after birth. His birth history was unremarkable. Dyspnea and tachypnea had started in his first days of life. His skin color was pale and femoral pulses were weak. There was a grade III/VI systolic ejection murmur at the second intercostal space to the right of sternum, and bilateral fine rales were heard on lung auscultation. The liver was palpable 3 cm below the right costal margin. Diagnoses based on electrocardiography were bicuspid aortic valve, valvular aortic stenosis, coarctation of the aorta, patent ductus arteriosus, and atrial septal defect. Cardiac catheterization and balloon aortic valvuloplasty were done. A follow-up chest x-ray film, including upper abdomen, showed bilateral adrenal calcification (Figure). Abdo minal ultrasonography revealed bilateral adrenal calcification without an associated mass lesion. The serum cortisol level was 28.51 [micro]g/dL (normal, 7 to 25 [micro]g/dL and adrenocorticotropic hormone (ACTH) level was 21.4 pg/mL (normal, 9 to 25 pg/mL). He had surgery for repair of aortic coarctation and ligation of patent ductus arteriosus. His postoperative course was uneventful, with no fluid or electrolyte disturbances.

The most common cause of adrenal calcification in children is adrenal hemorrhage. In neonates, adrenal hemorrhage usually occurs in the setting of difficult labor and birth asphyxia. Incidental finding of asyptomatic adrenal calcification has rarely been reported, (1,2) and the pathogenesis of adrenal hemorrhage has not been fully explained. In their series of 26 patients with the condition, Jarvis and Seaman (1) concluded that birth trauma and anoxia are important causes of adrenal calcification. None of their patients had congenital heart disease. The main causative factors of adrenal calcification are asphyxia resulting in venous congestion, or trauma during delivery or postnatal resuscitation; those especially prone to the condition include premature infants, large infants, and infants of diabetic mothers. (1,3,4) Any event leading to hypoxia may direct blood toward the central nervous system, heart, and adrenal glands. Congestion and injury of the endothelial cells lead to hemorrhage. Our patient had no history of perinatal distress or asphyxia, and his birth did not involve a difficult labor. Left-heart obstructive lesions, such as valvular aortic stenosis and coarctation of the aorta, are important causes of circulatory collapse and cardiac failure in neonates, (5) and may result in multiorgan failure. Spasmodic constriction and relaxation of the ductus arteriosus lead to a variable clinical picture. Constriction of the ductus arteriosus in the first days after birth is responsible for the cardiopulmonary decompensation in these infants. These factors may cause the redistribution of blood flow, leading to congestion of the adrenal gland. We believe that the hemodynamic compromise created by left-heart obstruction in this infant may well be the underlying cause of adrenal hemorrhage resulting in bilateral adrenal calcification. Our patient must have survived the neonatal period due to patency of the ductus arteriosus.

An incidental finding of adrenal calcification in infants or children alerts the physician to a possible tumor, such as neuroblastoma. In the absence of an associated mass, such a calcification is usually the result of perinatal adrenal hemorrhage. Adrenal hemorrhage is almost always associated with normal adrenal function. (1) The fact that normal neonatal adrenal glands excrete only small amounts of steroids during the first week of life probably accounts for the low incidence of adrenal insufficiency in the case of hemorrhage of the gland. The relative preservation of the cortex of the gland may also be a factor in normal functioning. (1)

We suggest that congenital heart malformations presenting in the early neonatal period with circulatory insufficiency or heart failure may precipitate adrenal hemorrhage and subsequent calcification.

Birgul Varan, MD

Arda Saygili, MD

Kursad Tokel, MD

Department of Pediatric Cardiology

Hakan Atalay, MD

Sukru Mercan, MD

Department of Cardiovascular Surgery

Tuba Cemil, MD

Department of Pediatrics

Baskent University Faculty of Medicine

10. Sokak

06490 Bachcelievler

Ankara, Turkey


(1.) Jarvis JL, Seaman WB: Idiopathic adrenal calcification in infants and children. Am J Roentgenol Rad Ther Nucl Med 1959;82:510-520

(2.) Ozturk H, Karnak I, Demirbag S, et al: Adrenal calcification in children: a case report. J Pediatr Surg 1999;34:481-482

(3.) Eklof O: Large asymptomatic adrenal hematomas in the neonate. Acta Radiol Diagn 1971;11:481-488

(4.) Black J, Williams DI: Natural history of adrenal hemorrhage in the newborn. Arch Dis Child 1973;48:183-190

(5.) Talner NS: The physiology of congenital heart disease. The Science and Practice of Pediatric Cardiology. Garson A Jr, Bricker JT, Fisher DJ, et al (eds). Baltimore, Williams and Wilkins, 2nd Ed, 1998, pp 1107-1118

Number at Risk

Non-Neutropenic Neutropenic
 525 47
 496 40
 470 36
 445 32
 423 29
 413 27
 403 27
 389 25
 382 23
 374 21

Comparison of adverse-event-free survival between neutropenic and
nonneutropenic patients with Staphylococcus aureus bacteremia
(Kaplan-Meier method). (Analysis excludes 13 neutropenic patients who
had polymicrobial bacteremia.)

Note: Table made from line graph

* This work was supported by ENDO Pharmaceuticals Inc. 223 Wilmington West Chester Pike, Chadds Ford, PA 19317.

* This study was supported by grant AI-01647 from the National Institutes of Health to Dr. Fowler.
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Date:Jul 1, 2002
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