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Pathological Case of the Month

Pathological Case of the Month Abstract A 1450-G WHITE MALE infant was delivered by cesarean section after 30 weeks' gestation. Fetal ultrasound showed polyhydramnios and possible complex congenital heart disease. At delivery, the infant required intubation and significant peak inflating pressures with 100% oxygen to achieve good color. Severe respiratory distress, hepatomegaly, and decreased perfusion developed. An echocardiogram revealed a dilated right ventricle, patent ductus arteriosus, and decreased cardiac function. Surfactant instillation, high-frequency ventilation, and tube thoracostomy were administered. Cardiac function was marginal despite inotropes. Subsequent improvement in the cardiorespiratory status allowed extubation on the 13th day of life. Intermittent hypertension of unclear origin was first noted on the 10th day of life, which was minimally responsive to diuretics and angiotensin-converting enzyme inhibitors. On the 25th day of life, a chest radiograph revealed cardiomegaly, pulmonary edema, and, for the first time, probable calcification of the abdominal aorta (Figure 1). An echocardiogram showed a thick, poorly functioning References 1. Bryant JH, White WA. A case of calcification of the arteries and obliterative endarteritis associated with hydronephrosis in a child aged six months . Guy's Hosp Rep . 1901;55:17-28. 2. Marrott PK, Newcombe KD, Becroft DM, Friedlander DH. Idiopathic infantile calcification with survival into adult life . Pediatr Cardiol . 1984;5:119-122.Crossref 3. Stryker WA. Arterial calcification in infancy with special reference to coronary arteries . Am J Pathol . 1946;22:1007-1020. 4. Anderson KA, Burbach JA, Fenton LJ, Jaqua RA, Barlow JF. Idiopathic arterial calcification of infancy in newborn siblings with unusual light and electron microscopic manifestations . Arch Pathol Lab Med . 1985;109:838-842. 5. Juul S, Ledbetter D, Wight TN, Woodrum D. New insights into idiopathic infantile arterial calcinosis: three patient reports . AJDC . 1990;144:229-233. 6. Bacon JF. Arterial calcification in infancy . JAMA . 1964;188:933-935.Crossref 7. Swischuck LE. Imaging of the Newborn, Infant, and Young Child . 3rd ed. Baltimore, Md: Williams and Wilkins; 1989:345-347. 8. Van Reempts PJ, Boven KJ, Spitaels SE, Roodhooft AM, Vercruyssen ELT, Van Acker KJ. Idiopathic arterial calcification of infancy . Calcif Tissue Int . 1991; 48:1-6.Crossref 9. Meradji M, De Villeneuve VH, Huber J, De Bruijn WC, Pearse RG. Idiopathic arterial calcification in siblings: radiologic diagnosis and successful treatment . J Pediatr . 1978;92:401-405.Crossref 10. Milner JS, Heitner R, Thomson P, et al. Hypertension as the major problem of idiopathic arterial calcification of infancy . J Pediatr . 1984;105:934-938.Crossref 11. Sholler GF, Yu JS, Bale PM, Hawker RE, Celermajer JM, Kozlowski K. Generalized arterial calcification of infancy: three case reports, including spontaneous regression with long-term survival . J Pediatr . 1984;105:257-260.Crossref 12. Dehner, LP. Pediatric Surgical Pathology . 2nd ed. Baltimore, Md: Williams and Wilkins; 1987:296-297. 13. Van Dyck M, Proesmans W, Van Hollebeke E, Marchal G, Moerman PH. Idiopathic infantile arterial calcification with cardiac, renal and central nervous system involvement . Eur J Pediatr . 1989;148:374-377.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Pediatrics & Adolescent Medicine American Medical Association

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Publisher
American Medical Association
Copyright
Copyright © 1996 American Medical Association. All Rights Reserved.
ISSN
1072-4710
eISSN
1538-3628
DOI
10.1001/archpedi.1996.02170260105019
Publisher site
See Article on Publisher Site

Abstract

Abstract A 1450-G WHITE MALE infant was delivered by cesarean section after 30 weeks' gestation. Fetal ultrasound showed polyhydramnios and possible complex congenital heart disease. At delivery, the infant required intubation and significant peak inflating pressures with 100% oxygen to achieve good color. Severe respiratory distress, hepatomegaly, and decreased perfusion developed. An echocardiogram revealed a dilated right ventricle, patent ductus arteriosus, and decreased cardiac function. Surfactant instillation, high-frequency ventilation, and tube thoracostomy were administered. Cardiac function was marginal despite inotropes. Subsequent improvement in the cardiorespiratory status allowed extubation on the 13th day of life. Intermittent hypertension of unclear origin was first noted on the 10th day of life, which was minimally responsive to diuretics and angiotensin-converting enzyme inhibitors. On the 25th day of life, a chest radiograph revealed cardiomegaly, pulmonary edema, and, for the first time, probable calcification of the abdominal aorta (Figure 1). An echocardiogram showed a thick, poorly functioning References 1. Bryant JH, White WA. A case of calcification of the arteries and obliterative endarteritis associated with hydronephrosis in a child aged six months . Guy's Hosp Rep . 1901;55:17-28. 2. Marrott PK, Newcombe KD, Becroft DM, Friedlander DH. Idiopathic infantile calcification with survival into adult life . Pediatr Cardiol . 1984;5:119-122.Crossref 3. Stryker WA. Arterial calcification in infancy with special reference to coronary arteries . Am J Pathol . 1946;22:1007-1020. 4. Anderson KA, Burbach JA, Fenton LJ, Jaqua RA, Barlow JF. Idiopathic arterial calcification of infancy in newborn siblings with unusual light and electron microscopic manifestations . Arch Pathol Lab Med . 1985;109:838-842. 5. Juul S, Ledbetter D, Wight TN, Woodrum D. New insights into idiopathic infantile arterial calcinosis: three patient reports . AJDC . 1990;144:229-233. 6. Bacon JF. Arterial calcification in infancy . JAMA . 1964;188:933-935.Crossref 7. Swischuck LE. Imaging of the Newborn, Infant, and Young Child . 3rd ed. Baltimore, Md: Williams and Wilkins; 1989:345-347. 8. Van Reempts PJ, Boven KJ, Spitaels SE, Roodhooft AM, Vercruyssen ELT, Van Acker KJ. Idiopathic arterial calcification of infancy . Calcif Tissue Int . 1991; 48:1-6.Crossref 9. Meradji M, De Villeneuve VH, Huber J, De Bruijn WC, Pearse RG. Idiopathic arterial calcification in siblings: radiologic diagnosis and successful treatment . J Pediatr . 1978;92:401-405.Crossref 10. Milner JS, Heitner R, Thomson P, et al. Hypertension as the major problem of idiopathic arterial calcification of infancy . J Pediatr . 1984;105:934-938.Crossref 11. Sholler GF, Yu JS, Bale PM, Hawker RE, Celermajer JM, Kozlowski K. Generalized arterial calcification of infancy: three case reports, including spontaneous regression with long-term survival . J Pediatr . 1984;105:257-260.Crossref 12. Dehner, LP. Pediatric Surgical Pathology . 2nd ed. Baltimore, Md: Williams and Wilkins; 1987:296-297. 13. Van Dyck M, Proesmans W, Van Hollebeke E, Marchal G, Moerman PH. Idiopathic infantile arterial calcification with cardiac, renal and central nervous system involvement . Eur J Pediatr . 1989;148:374-377.Crossref

Journal

Archives of Pediatrics & Adolescent MedicineAmerican Medical Association

Published: Jan 1, 1996

References