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Spontaneous thymic hemorrhage in the neonate: Report of two cases
Spontaneous Thymic Hemorrhage in the Neonate: Report of Two Cases By Morton M. Woolley, Hart Isaacs, Jr., George Lindesmith, Dorothy M. Vollmer, and Stephen Van Adelsberg
T
HE CLINICAL, RADIOGRAPHIC, and surgical findings of two infants are similar and unique enough to warrant the following report: CASE REPORT
Case 1 S.Y. (Glendale Adventist Hospital No. 175226). A female infant was born on May 19, 1961 weighing 2.9 kg. Pregnancy and delivery were uncomplicated. The infant fed well for I day then developed poor feeding, pallor, and gradually increasing respiratory distress. A chest x-ray taken at 36 hr of age (Fig. 1) revealed the left chest to be o p a q u e with an associated shift of the mediastinum to the right. Physical examination revealed poor breath s o u n d s on the left, pallor, and respiratory distress. Hemoglobin was recorded as 9.4 g, and no additional laboratory procedures were performed at that time. She was given 1 cc of Phytonadione, intramuscularly. A chest tube was placed in the left pleural space and 60 cc of dark blood was removed. The infant's respiratory distress decreased immediately. During the next 24 hr she was given 100 cc of whole blood. There was a small a m o u n t of blood loss t h r o u g h the chest tube; however, the respiratory distress gradually recurred and another x-ray revealed increasing opacity of the left hemithorax and increasing mediastinal shift to the right. The patient was obviously collecting blood in the left hemithorax. A left t h o r a c o t o m y was performed on May 21, 1961 and the left chest was found to contain 45 cc of blood. The left lung was completely collapsed. The t h y m u s was diffusely h e m o r r h a g i c and the left mediastinal pleura had ruptured so that the blood from the t h y m u s had escaped into the left pleural space. The enlarged hemorrhagic t h y m u s was dissected from the pericardium and removed. The right pleural space was entered during the dissection; therefore, a chest catheter was placed in each pleural space for postoperative drainage. The patient received 80 cc of blood during surgery and postoperative hemoglobin was 13.5 g. The postoperative course was uneventful, the chest tubes were removed on the second postoperative day and the patient was discharged from the hospital on the sixth postoperative day after the skin sutures had been removed. The t h y m u s weighed 25 g. Gross and microscopic e x a m i n a t i o n showed only diffuse h e m o r r h a g e with extravascation into the intralobular connective tissue. There was no evidence of tumor. The patient's subsequent course was uneventful.
From the Childrens Hospital of Los Angeles, The Glendale Adventist Hospital, Glendale, Calif. and the University of Southern California School of Medicine, Department of Surgery, Pediatrics, and Pathology, Los Angeles, Calif. Morton M. Woolley, M.D.: Head, Division of Pediatric Surgery, Department of Surgery at the Childrens Hospital of Los Angeles; Associate Clinical Professor of Surgery, University of Southern California, School of Medicine, Los Angeles, Calif. Hart Isaacs, M.D.: Pathologist, Department of Pathology at the Childrens Hospital of Los Angeles; Assistant Professor of Pathology, University of Southern California, School of Medicine, Los Angeles, Calif. George Lindesmith, M.D.: Head, Division of Thoracic Surgery, Department of Surgery at the Childrens Hospital of Los Angeles, Los Angeles, Calif. Dorothy Vollmer, M.D.: Pediatrician, Glendale, CaliJl Stephen Van Adelsberg, M.D.: Attending Physician, Department of Pediatrics at the Childrens Hospital of Los Angeles, Los A ngeles, Calif. Address for reprint requests." Morton M. WooHey, M.D., Head, Division of Pediatric Surgery, Childrens Hospital of Los Angeles, P. O. Box 54700, Los Angeles, Calif 90054. 9 1974 by Grune & Stratton, lnc. Journal of Pediatric Surgery, Vol. 9. No. 2 (April), 1974
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Fig. 1. Case 1. Chest x-ray taken at 36 hr of age showing opaque left chest and mediasfinal shift to the right.
Case 2 R.P. (CHLA No. 478881). A l-mo-old male infant was admitted on February 28, 1973 with a 2-day history of irritability, dyspnea, and pallor. The infant's birth weight was 3.3 kg; pregnancy and vaginal delivery were without complication. The infant had been feeding and gaining weight normally until the onset of the present illness. Examination of the infant included the following: Weight 4.0 kg, temperature 35.3, pulse 154, respirations 58, blood pressure 124/54. Skin was pale, there were decreased breath sounds over the left chest. Laboratory tests revealed the following: hemoglobin, 7.2 g; white blood count, 28,100; fibrinogen, 330 rag/100 ml (normal 200 400 rag/100 ml); partial thromboplastin time 66 sec (normal less than 40 sec), prothrombin 56~. The patient bled excessively from venipuncture and heel-stick sites. He was given the following therapy to control bleeding and to replace blood volume: Phytonadione, 1 mg; fresh-
Fig. 2. Case 2. (A) Chest x-ray of case 2 shewing widened mediastinum and left pleural opacity. (B) Photomicrograph of thymus, case 2.
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frozen plasma, 15 cc per kg; sedimented red cells, 10 cc per kg. After these medications the external bleeding stopped. A chest x-ray revealed a mediastinal mass and opacity of the left chest (Fig. 2A). The patient was operated upon through a median s t e r n o t o m y incision and an hemorrhagic t h y m u s was removed. The left pleural space contained 60 cc of blood as a result of rupture of the left mediastinal pleura. There was no problem with hemostasis at the time of operation and no postoperative bleeding. The w o u n d healed per p r i m u m . The patient was discharged from the hospital 9 days after operation, on March 9, 1973. Grossly the t h y m u s contained large areas of capsular and lobular hemorrhage. Microscopic sections revealed foci of recent h e m o r r h a g e a n d acute involution. Other than this there was no histologic abnormality. (Fig. 1B). DISCUSSION
To our knowledge these are unique case histories of patients with spontaneous bleeding into the thymus. Each of the patients presented within the neonatal period with no specific etiology for the bleeding. In each instance the left mediastinal pleura ruptured allowing left h e m o t h o r a x to occur. In the second patient, a coagulopathy was identified. N o coagulation studies were performed on the first patient who may well have had a coagulation defect which was corrected by the use of whole blood and vitamin K. The x-rays are quite similar, so that this diagnosis can p r o b a b l y be m a d e in the future in patients who present with anemia, respiratory distress, and this x-ray appearance.
T
HE CLINICAL, RADIOGRAPHIC, and surgical findings of two infants are similar and unique enough to warrant the following report: CASE REPORT
Case 1 S.Y. (Glendale Adventist Hospital No. 175226). A female infant was born on May 19, 1961 weighing 2.9 kg. Pregnancy and delivery were uncomplicated. The infant fed well for I day then developed poor feeding, pallor, and gradually increasing respiratory distress. A chest x-ray taken at 36 hr of age (Fig. 1) revealed the left chest to be o p a q u e with an associated shift of the mediastinum to the right. Physical examination revealed poor breath s o u n d s on the left, pallor, and respiratory distress. Hemoglobin was recorded as 9.4 g, and no additional laboratory procedures were performed at that time. She was given 1 cc of Phytonadione, intramuscularly. A chest tube was placed in the left pleural space and 60 cc of dark blood was removed. The infant's respiratory distress decreased immediately. During the next 24 hr she was given 100 cc of whole blood. There was a small a m o u n t of blood loss t h r o u g h the chest tube; however, the respiratory distress gradually recurred and another x-ray revealed increasing opacity of the left hemithorax and increasing mediastinal shift to the right. The patient was obviously collecting blood in the left hemithorax. A left t h o r a c o t o m y was performed on May 21, 1961 and the left chest was found to contain 45 cc of blood. The left lung was completely collapsed. The t h y m u s was diffusely h e m o r r h a g i c and the left mediastinal pleura had ruptured so that the blood from the t h y m u s had escaped into the left pleural space. The enlarged hemorrhagic t h y m u s was dissected from the pericardium and removed. The right pleural space was entered during the dissection; therefore, a chest catheter was placed in each pleural space for postoperative drainage. The patient received 80 cc of blood during surgery and postoperative hemoglobin was 13.5 g. The postoperative course was uneventful, the chest tubes were removed on the second postoperative day and the patient was discharged from the hospital on the sixth postoperative day after the skin sutures had been removed. The t h y m u s weighed 25 g. Gross and microscopic e x a m i n a t i o n showed only diffuse h e m o r r h a g e with extravascation into the intralobular connective tissue. There was no evidence of tumor. The patient's subsequent course was uneventful.
From the Childrens Hospital of Los Angeles, The Glendale Adventist Hospital, Glendale, Calif. and the University of Southern California School of Medicine, Department of Surgery, Pediatrics, and Pathology, Los Angeles, Calif. Morton M. Woolley, M.D.: Head, Division of Pediatric Surgery, Department of Surgery at the Childrens Hospital of Los Angeles; Associate Clinical Professor of Surgery, University of Southern California, School of Medicine, Los Angeles, Calif. Hart Isaacs, M.D.: Pathologist, Department of Pathology at the Childrens Hospital of Los Angeles; Assistant Professor of Pathology, University of Southern California, School of Medicine, Los Angeles, Calif. George Lindesmith, M.D.: Head, Division of Thoracic Surgery, Department of Surgery at the Childrens Hospital of Los Angeles, Los Angeles, Calif. Dorothy Vollmer, M.D.: Pediatrician, Glendale, CaliJl Stephen Van Adelsberg, M.D.: Attending Physician, Department of Pediatrics at the Childrens Hospital of Los Angeles, Los A ngeles, Calif. Address for reprint requests." Morton M. WooHey, M.D., Head, Division of Pediatric Surgery, Childrens Hospital of Los Angeles, P. O. Box 54700, Los Angeles, Calif 90054. 9 1974 by Grune & Stratton, lnc. Journal of Pediatric Surgery, Vol. 9. No. 2 (April), 1974
231
:)32
WOOLLEY ET AL.
Fig. 1. Case 1. Chest x-ray taken at 36 hr of age showing opaque left chest and mediasfinal shift to the right.
Case 2 R.P. (CHLA No. 478881). A l-mo-old male infant was admitted on February 28, 1973 with a 2-day history of irritability, dyspnea, and pallor. The infant's birth weight was 3.3 kg; pregnancy and vaginal delivery were without complication. The infant had been feeding and gaining weight normally until the onset of the present illness. Examination of the infant included the following: Weight 4.0 kg, temperature 35.3, pulse 154, respirations 58, blood pressure 124/54. Skin was pale, there were decreased breath sounds over the left chest. Laboratory tests revealed the following: hemoglobin, 7.2 g; white blood count, 28,100; fibrinogen, 330 rag/100 ml (normal 200 400 rag/100 ml); partial thromboplastin time 66 sec (normal less than 40 sec), prothrombin 56~. The patient bled excessively from venipuncture and heel-stick sites. He was given the following therapy to control bleeding and to replace blood volume: Phytonadione, 1 mg; fresh-
Fig. 2. Case 2. (A) Chest x-ray of case 2 shewing widened mediastinum and left pleural opacity. (B) Photomicrograph of thymus, case 2.
THYMIC HEMORRHAGE
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frozen plasma, 15 cc per kg; sedimented red cells, 10 cc per kg. After these medications the external bleeding stopped. A chest x-ray revealed a mediastinal mass and opacity of the left chest (Fig. 2A). The patient was operated upon through a median s t e r n o t o m y incision and an hemorrhagic t h y m u s was removed. The left pleural space contained 60 cc of blood as a result of rupture of the left mediastinal pleura. There was no problem with hemostasis at the time of operation and no postoperative bleeding. The w o u n d healed per p r i m u m . The patient was discharged from the hospital 9 days after operation, on March 9, 1973. Grossly the t h y m u s contained large areas of capsular and lobular hemorrhage. Microscopic sections revealed foci of recent h e m o r r h a g e a n d acute involution. Other than this there was no histologic abnormality. (Fig. 1B). DISCUSSION
To our knowledge these are unique case histories of patients with spontaneous bleeding into the thymus. Each of the patients presented within the neonatal period with no specific etiology for the bleeding. In each instance the left mediastinal pleura ruptured allowing left h e m o t h o r a x to occur. In the second patient, a coagulopathy was identified. N o coagulation studies were performed on the first patient who may well have had a coagulation defect which was corrected by the use of whole blood and vitamin K. The x-rays are quite similar, so that this diagnosis can p r o b a b l y be m a d e in the future in patients who present with anemia, respiratory distress, and this x-ray appearance.