Neurol Med Chir (Tokyo) 43, 85¿87, 2003 Surgical Treatment of Spontaneous Intracerebral Hemorrhage in a Full­term Infant With Coagulopathy —Case Report— Shunichi YOKOYAMA, Kenji TAKAYAMA, Takahiro MURAKAMI, Yasunari ISHIKAWA, Naoki FUJIWARA*, and Hitoshi GANAHA* Divisions of Neurosurgery and *Pediatrics, Okinawa Chubu Hospital, Gushikawa, Okinawa Abstract An 11­week­old male infant presented with intracerebral hemorrhage associated with coagulopathy manifesting as left hemiparesis, lethargy, and vomiting. Computed tomography demonstrated extensive right frontoparietal intracerebral hemorrhage extending into the ventricular system. Liver function tests revealed abnormal values of transaminases and bilirubin. Blood coagulation studies showed prolonged prothrombin time (PT) and activated partial thromboplastin time (APPT). PT and APTT im­ mediately normalized after the administration of vitamin K and fresh frozen plasma. Right parietal craniotomy and evacuation of the hematoma were performed because of the deterioration in conscious­ ness and left hemiparesis. No vascular abnormality was observed in the hematoma cavity. After sur­ gery, he became alert and the left hemiparesis improved. There is a risk of intracerebral hemorrhage due to vitamin K deficiency even if prophylactic administration of vitamin K was given. Surgical treat­ ment should be considered for the treatment of infantile spontaneous intracerebral hemorrhage, espe­ cially if neurological deterioration is present. Key words: coagulopathy, infant, intracerebral hemorrhage, Introduction June 24, 2002; Accepted vitamin K deficiency administration of vitamin K. Case Report Spontaneous intracerebral hemorrhage in full­term neonates and infants is rare, but occurs in the ab­ sence of identifiable risk factors in most cases.3,5,11, 13,15) The risk factors for intracranial hemorrhage in neonates are premature birth, birth asphyxia, and birth trauma.13) Spontaneous intracerebral hemor­ rhage in early infancy usually occurs in breast­fed infants at 3 to 8 weeks of age, and is usually caused by vitamin K deficiency, whereas other causes are rare.14) The incidence of vitamin K deficiency in infancy has decreased remarkably in Japan, due to the in­ creasingly widespread prophylactic administration of vitamin K during the neonatal period, but cases of vitamin K deficiency during or after the neonatal period still occur.7) We report a case of intracerebral hemorrhage associated with coagulopathy treated by surgery in a full­term (À2500 g birth weight, 37 weeks gestation­ al age) infant who had received prophylactic Received surgery, An 11­week­old male infant was born on May 9, 2001, at 38 weeks of gestation, by spontaneous vagi­ nal delivery to a 40­year­old woman. The pregnancy, labor, and delivery were uncomplicated. Vitamin K was administered on the day of birth, and at age 5 days and 1 month. He presented with lethargy and vomiting on July 28, 2001 at the emergency department of Chubu Hospital. Physical examination found he was lethar­ gic, with a head circumference of 40 cm and isocor­ ic pupils, but no evidence of head trauma. Neurolog­ ical examination found slight left hemiparesis. He had mild jaundice. Transfontanel ultrasonography in the emergency room revealed a hyperechoic mass in the fron­ toparietal region. Computed tomography (CT) con­ firmed extensive right frontoparietal intracerebral hemorrhage extending into the ventricular system (Fig. 1A). Laboratory studies revealed a white blood cell count of 14,500/ml with a normal differential October 16, 2002 85 S. Yokoyama et al. 86 Fig. 1 A: Preoperative computed tomography (CT) scan demonstrating extensive right fron­ toparietal intracerebral hemorrhage. B: Postoperative CT scan confirming evacua­ tion of the hematoma. count and a platelet count of 375,000/ml. Liver function tests showed aspartate aminotransferase 176 IU/l (normal 10–34 IU/l), alanine aminotrans­ ferase 182 IU/l (normal 7–35 IU/l), total bilirubin 6.3 mg/dl (normal 0.2–1.2 mg/dl), and direct biliru­ bin 4.8 mg/dl (normal 0.0–0.4 mg/dl). Results of blood coagulation studies were abnormal, with a prothrombin time (PT) of 56.4 sec (normal 10–13 sec) and an activated partial thromboplastin time (APPT) of 101.8 sec (normal 23–35 sec). The hepaplastin test showed the coagulation activity was less than detect­ able (normal 70–130%). PT and APTT immediately normalized after the administration of vitamin K and fresh frozen plasma. The patient underwent a right parietal craniotomy and evacuation of the hematoma because of the de­ terioration in consciousness and left hemiparesis. The hematoma was evacuated without difficulty during hemostasis. No vascular abnormality was ob­ served in the hematoma cavity. Postoperative CT confirmed the evacuation of the hematoma (Fig. 1B). The postoperative course was uneventful. He became alert and the left hemiparesis improved. The values of PT, APTT, and the hepa­ plastin test remained in the normal range without further treatment. Neurological examination found only very slight left hemiparesis at age 13 months. Discussion Infants with vitamin K deficiency are breast­fed infants of 3 to 8 weeks of age, and have a bleeding tendency which disappears after vitamin K treat­ ment.14) Our patient was breast­fed, showed a bleed­ ing diathesis, and vitamin K and fresh frozen plasma corrected his abnormal PT and APTT values, consis­ tent with vitamin K deficiency.6) Liver function tests suggested a hepatic disorder. Defects in the entero­ hepatic circulation interfere with the absorption of fat­soluble vitamins in the ileum.6) Coagulopathy probably occurred in our patient because of the malabsorption of vitamin K caused by the transient liver dysfunction, and resulted in intracerebral hemorrhage. Therefore, there is a risk of intra­ cerebral hemorrhage despite the prophylactic administration of vitamin K. Clinical symptoms of perinatal intracerebral hemorrhage, irrespective of the specific location of the bleeding, include cardiorespiratory problems, arterial hypotension, and neurological disturbances such as lethargy, seizures, and loss of archaic reflexes.1) The initial symptoms in our patient were lethargy and vomiting. Cranial ultrasonography pro­ vides rapid and accessible diagnostic information in patients with these nonspecific symptoms which may not be immediately available with other neuro­ imaging methods.12) Care should be taken in the diagnosis of infantile intracerebral hemorrhage be­ cause the clinical symptoms may be vague and non­ specific. Transfontanel ultrasonography and subse­ quent CT can provide a rapid and accurate diagno­ sis.1) The prognosis for patients treated conservatively is usually good even if extensive hemorrhage is ob­ served by the initial CT.2,4,10,12,14) However, subse­ quent developmental progression was poor in some patients despite adequate control of intracranial hypertension.8) Surgical evacuation was not im­ portant in the management of the majority of previ­ ous patients.9,10,15) However, the majority of a large series of full­term neonates and infants with in­ tracerebral hemorrhage were managed surgically.15) The primary indication for surgical intervention was extensive hemorrhage with radiographic evi­ dence of mass effect in a symptomatic infant. The very young brain requires good anesthesiology and adequate hemostasis is difficult to achieve.15) Craniotomy in young infants requires an appropri­ ate team of experienced personnel. We operated on our patient because of the deterio­ ration in consciousness and the finding of left hemiparesis in the emergency room. The hematoma was evacuated without difficulty during hemostasis. Patients treated by surgery at an early stage have had variable outcome.14) Neurological examination in our patient at age of 13 months was normal except for very slight left hemiparesis. The indications for surgical treatment are controversial, because the significance of surgical intervention is difficult to establish due to the lack of studies on the use of sur­ Neurol Med Chir (Tokyo) 43, February, 2003 Intracerebral Hemorrhage in an Infant gery in this disease. Intracerebral hemorrhage may have effects on long­term development, so further investigation is needed to establish whether surgical treatment is more important than previously thought. 8) References 9) 1) 2) 3) 4) 5) 6) 7) Bal áeriaux D, Ticket D, Dony D, Leanmart L: The con­ tribution of CT to perinatal intracranial hemorrhage including that accompanying apparently uncompli­ cated delivery at full term. Neuroradiology 19: 273–277, 1980 Bergman I, Bauer RE, Barmada MA, Latchaw RE, Taylor HG, David R, Painter MJ: Intracranial hemor­ rhage in the full­term neonatal infant. Pediatrics 75: 488–496, 1985 Cartwright GW, Culbertson K, Schrener RL, Garg BP: Changes in clinical presentation of term infants with intracranial hemorrhage. Dev Med Child Neurol 21: 730–737, 1979 Chaplin ER Jr, Goldstein GW, Norman D: Neonatal seizures, intracerebral hematoma, and subarachnoid hemorrhage in full­term infants. 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Brain Dev 6: 435–443, 1984 Sandberg DI, Lamberti­Pasculli M, Drake JM, Humphreys RP, Rutka JT: Spontaneous intraparen­ chymal hemorrhage in full­term neonates. Neurosur­ gery 48: 1042–1049, 2001 Address reprint requests to: S. Yokoyama, M.D., Division of Neurosurgery, Okinawa Chubu Hospital, 281 Aza­ Miyazato, Gushikawa, Okinawa 904–2293, Japan. e­mail: yokoyamaäshunichi—hosp.pref.okinawa.jp.