Nonimmunologic hydrops fetalis--a review of 31 cases

Non-immunologic hydrops fetalis-a review of 31 cases: 31 Patients with non-immunologic hydrops fetalis (NIHF) seen between 1984 and 1987 are described. 13 infants survived. The infants with major congenital malformations and connatal infections died. In 8 of the patients who died a cause for NIHF could not be identified. 10 of the survivors presented chylous ascites and/or chylothorax without major congenital anomalies. 2 infants had fetal tachyarrhythmia and 1 patient showed severe anemia due to fetomaternal hemorrhage.     

Report of three cases: congenital chylothorax and treatment modalities

Chylothorax is the most common cause of pleural effusion in the newborn. We report three patients with congenital chylothorax and discussed the clinical course and treatment options. Cases 1 and 2 with congenital chylothorax were treated by chest tube placement and total parenteral nutrition (TPN), and were fed a formula rich in medium-chain triglyceride. They were discharged home without any sequelae. Our 3rd case with chylothorax did not respond to the conventional therapies. Octreotide infusion was tried without any benefits and necessitated surgical intervention, but the infant developed chronic lung disease requiring nasal oxygen therapy until three months of age. All three patients developed complications of chylothorax treatment like chest tube dysfunction, pneumothorax, nosocomial sepsis, and cholestasis. Management of congenital chylothorax necessitates a multidisciplinary approach. Treatment options include pleural drainage, cessation of enteral feeding and initiation of TPN. Experience with octreotide treatment is limited. Surgery should be reserved for severe and refractory cases.     

Nonimmune Hydrops Fetalis in the Liveborn: Series of 32 Autopsies

Nonimmune hydrops fetalis (NIHF) or generalized soft tissue edema and cavity effusions may be due to cardiovascular diseases, congenital infections, genitourinary malformations, thoracic masses, placental conditions, chromosomal abnormalities, and idiopathic. We report 32 cases of NIHF from among 429 neonates who underwent autopsies (incidence 7.45%). Sixteen cases (50%) had cardiovascular disease; all were due to low output cardiac failure; 7 had structural congenital heart disease. Three of the children with congenital heart disease also had chromosomal abnormalities: 2 had trisomy 18 and 1 had Noonan syndrome. Among myocardial conditions were five subjects with cardiomyopathies (1 of each of the following types): oncocytic, dilated, endocardial fibroelastosis, cardiac glycogenosis, and carnitine deficiency; 3 had myocarditis, and 1 had cardiac rhabdomyomas. Congenital infections were due to cytomegalovirus in 3 cases, bacteria in 2, and parvovirus in 1. The mechanism of NIHF in these cases might be a combination of decreased myocardial contractility due to myocarditis and fetal anemia. Genitourinary diseases were present in 5 newborns: Two had congenital nephrotic syndrome, 1 had VACTER association, 1 had prune-belly syndrome, and 1 had urogenital sinus malformation. Intrathoracic lesions were found in 2 babies (pulmonary sequestration and diaphragmatic hernia). One twin died of volume overload due to twin transfusion syndrome. Only 2 newborns were classified as idiopathic. Our study shows that cardiovascular diseases that lead to heart failure or impaired venous return are more common in the liveborn (50%), whereas congenital infections are more common in the stillborn with NIHF.     

Etiology and management of pediatric chylothorax

OBJECTIVES: To determine the incidence and etiology of chylothorax and to assess our therapeutic management approach. STUDY DESIGN: We reviewed 51 patients diagnosed with chylothorax over a 12-year period. Cause, interval between operation and diagnosis, duration of chylothorax, and total volume loss per weight were recorded. RESULTS: Chylothorax was diagnosed in 46 children after cardiothoracic surgery, giving an incidence of 2.5% (46/1842); in 1 child chylothorax occurred after chest trauma, and in 4 the chylothorax was congenital or a manifestation of lymph angiomatosis. Three etiologic groups were identified: group 1, direct injury to the thoracic duct (33/51 = 65%); group 2, thrombosis and/or high venous pressure in the superior vena cava (14/51 = 27%); and group 3, congenital (4/51 = 8%). Conservative treatment was the only treatment in 80% of the patients. Surgical procedures consisted of 4 ligations of the thoracic duct, placement of 7 pleurodesis shunts, and placement of 2 pleuroperitoneal shunts. Patients in groups 2 and 3 were at higher risk for failure of conservative treatment (P <. 005). Longer duration of chylothorax and higher volume of drainage were present in group 2 compared with group 1 (P <.01). CONCLUSION: Conservative treatment was successful in 80% of the patients with our management approach. Prevention, early recognition, and treatment of potential complications, such as superior vena cava thrombosis or obstruction, may further improve success of conservative treatment. Congenital chylothorax seems different and may require a specific approach.     

Prevalence of nosocomial infections in neonatal intensive care unit patients: Results from the first national point-prevalence survey.

OBJECTIVES: Patients admitted to neonatal intensive care units (NICUs) are at high risk of nosocomial infection. We conducted a national multicenter assessment of nosocomial infections in NICUs to determine the prevalence of infections, describe associated risk factors, and help focus prevention efforts. STUDY DESIGN: We conducted a point prevalence survey of nosocomial infections in 29 Pediatric Prevention Network NICUs. Patients present on the survey date were included. Data were collected on underlying diagnoses, therapeutic interventions/treatments, infections, and outcomes. RESULTS: Of the 827 patients surveyed, 94 (11.4%) had 116 NICU-acquired infections: bloodstream (52.6%), lower respiratory tract (12.9%), ear-nose-throat (8.6%), or urinary tract infections (8.6%). Infants with infections were of significantly lower birth weight (median 1006 g [range 441 to 4460 g] vs 1589 g [range 326 to 5480 g]; P <.001) and had longer median durations of stay than those without infections (88 days [range 8 to 279 days] vs 32 days [range 1 to 483 days]; P <.001). Most common pathogens were coagulase-negative staphylococci and enterococci. Patients with central intravascular catheters (relative risk = 3.81, CI 2.32-6.25; P <.001) or receiving total parenteral nutrition (relative risk = 5.72, CI 3.45-9.49; P <.001) were at greater risk of bloodstream infection. CONCLUSIONS: This study documents the high prevalence of nosocomial infections in patients in NICUs and the urgent need for more effective prevention interventions.     

Management of Refractory Chylothorax After Pediatric Cardiovascular Surgery

We investigated the optimal treatment for refractory chylothorax after pediatric cardiovascular surgery. We retrospectively reviewed the cases of 15 consecutive patients who developed chylothorax after congenital heart surgery performed between December 2004 and November 2010. Among the 15 patients (12 male and 3 female; median age 13.9 months) who developed postoperative chylothorax, 10 recovered with conservative therapy, such as a low-fat diet, medium chain triglyceride–enriched diet, or total parenteral nutrition. Of the remaining 5 patients who underwent surgical treatment followed by conventional therapy, 4 showed improvement, and 1 died from cardiac failure. Surgical treatment was performed at a median of 19 days after diagnosis of chylothorax. Average drainage output of thoracocentesis for the first 5 days before thoracic duct ligation was 33.1 ml/kg/day. Duration of chylous fluid drainage was significantly longer in surgical patients than in patients who recovered with conservative therapy (p < 0.01). Surgical patients tended to be younger with lower body weight. Significant risk factors for surgical intervention were age <4 months, body weight <4 kg, and duration of drainage >10 days. In cases of refractory postoperative chylothorax, surgical therapy such as thoracic duct ligation should be considered when discharge from the drainage tube is >30 ml/kg/day or chylothorax is not improved within 10 days.     

Octreotide as therapeutic option for congenital idiopathic chylothorax: a case series

Background: Octreotide, a somatostatin analogue, is used for the management of patients with refractory chylothorax although its safety and efficacy in neonates have not been evaluated in controlled clinical trials. We present one of the largest case series about the use of octreotide in congenital idiopathic chylothorax. Methods: Six cases of congenital chylothorax (CC) were prospectively collected, who were managed with same unit protocol for octreotide. Mean (SD) gestation was 34.5 (±2.2) weeks, and birthweight was 3410 (±840.4) g. All infants required chest drains from day 1 of life, and the mean (SD) duration of insertion was 36.1 (±8.5) days. Octreotide was commenced at a median age of 13.5 days (range 8–22), given for a median duration of 20 days (range 12–27). The starting dose was 0.5–1 μg/kg/h with an increment of 1–2 μg/kg/day to a maximum of 10 μg/kg/day. Resolution of chylothorax was achieved in five patients, being resistant to treatment in the sixth patient. None had adverse effects from octreotide. Full enteral feeds were reached at a mean age of 44 days. Conclusion: Early commencement of octreotide is recommended although further reports to evaluate the safety and efficacy would add to the profile of this medication in the treatment of CC.     

Value of Autopsy in Nonimmune Hydrops Fetalis: Series of 51 Stillborn Fetuses

Nonimmune hydrops fetalis (NIHF) is used to describe fetuses and newborns with generalized edema and cavity effusions. It is helpful to alert physicians about the presence of anemia, heart failure, and/or hypoproteinemia, but this diagnosis is frequently overlooked. We reviewed the autopsy files from 1990 to 2000, selected all cases with NIHF including clinical information (with maternal laboratory tests and ultrasound), and classified patients by etiology. Among 840 stillborn autopsies during the 11-year period, we found 51 with NIHF (6.07%). The clinical summary had mentioned hydrops in 14 patients and the etiology in another 7 by fetal ultrasonography, but without addressing the possibility of hydrops. In the remaining 30 cases neither hydrops nor an etiology was mentioned. Other pertinent diagnoses were maternal diabetes mellitus (4), congenital heart disease (3), and cystic hygroma (2). The following diagnoses were made in one instance each: cardiac tumor, twin transfusion syndrome, congenital adenomatoid malformation, syphilis, Turner syndrome, and cerebral arteriovenous malformation. Postmortem and placental examination confirmed the following etiologies: congenital infections (17); placental pathology significant enough to explain NIHF (10); cardiovascular diseases (8) (further classified as congenital heart disease [3], rhabdomyoma [1], and vascular malformations [4]); chromosomal abnormalities (6); uncontrolled maternal diabetes (4); intrathoracic lesions (2); prune-belly syndrome (2); and idiopathic NIHF (2). Only 3.9% of the cases studied had no identifiable etiology. The cause of hydrops was confirmed by autopsy in 47 fetuses (92%), which further supports the importance of performing an autopsy. Thirty-two cases (62.74%) had placental abnormalities helpful to the etiology (parvovirus, syphilis, Turner's syndrome, etc.). In 20 instances, the clinical summary had no mention of either hydrops or any of the diseases leading to it. The autopsy in conjunction with placental examination and fetal ultrasound represent the best combination to determine the etiology of NIHF among stillborn fetuses.     

非免疫性胎儿水肿新生儿的临床特征及预后分析

目的 分析非免疫性胎儿水肿（NIHF）新生儿的临床特征、病因及转归情况。方法 回顾性分析23例NIHF新生儿的临床资料及转归。结果 23例NIHF患儿中,早产儿18例（78%）,足月儿5例（22%）;出生窒息12例（52%）,其中重度窒息6例。NIHF病因包括双胎输血综合征（TTTS）8例（35%）,心血管畸形3例（13%）,微小病毒B19感染3例（13%）,先天性乳糜胸2例（9%）,Turner综合征1例（4%）,柯萨奇病毒感染1例（4%）,病因不明5例（22%）。临床治愈13例（57%）,死亡10例,新生儿期病死率为43%。死亡组中早产儿、新生儿窒息、5分钟Apgar评分<8分及心力衰竭比例（分别为100%、100%、60%、60%）明显高于存活组（分别为62%、15%、8%、8%）（P < 0.05）。结论 NIHF新生儿易发生出生窒息;胎龄越小、窒息程度越重、合并心力衰竭者新生儿期死亡风险越大。TTTS中受血儿是NIHF的主要病因。     

How should chylothorax be managed?

The management and complications of chylothorax occurring beyond the neonatal period were reviewed retrospectively. Records from 15 patients treated between 1976 and 1986 were analysed; a combination of thoracocentesis, chest drain insertion, and dietary modification were successful in abolishing chyle leakage in 10 cases. One child died from complications of cardiac surgery rather than from the chylothorax, and surgical intervention was necessary in the remaining four patients and included pleurectomy in three and thoracic duct ligation in the fourth. Lymphopenia, hypoalbuminaemia, hyponatraemia, and weight loss were the most common complications of conservative management and tended to occur in those patients with the longest duration of drainage. Postoperative recovery after pleurectomy and thoracic duct ligation was uneventful. We conclude that conservative management of chylothorax will be successful in most cases. Complications of such a policy are fairly common but rarely serious.     

Somatostatin treatment of congenital chylothorax may induce transient hypothyroidism in newborns

AIM: To describe a group of neonates with congenital, non-traumatic chylothorax, one of whom developed transient hypothyroidism following treatment with somatostatin. METHODS: The charts of seven infants with congenital chylothorax were reviewed in terms of their clinical presentation, the severity of their disease, the complications they presented and the duration of their hospitalization. Their pituitary-thyroid axis function was monitored in particular. RESULTS: The seven infants, all preterm (32-34 wk), suffered from congenital chylothorax and hydrops fetalis diagnosed during the prenatal period. Four were treated by intrauterine drainage, and four had congenital malformations. Hospitalization lasted from 32 to 120 d. Three of the infants suffered from thrombocytopenia, three had chronic lung disease, and one suffered from Gram-negative sepsis. The infant treated with somatostatin initially had normal thyroid function, but later developed primary transient hypothyroidism and was treated with L-thyroxine. The thyroid screening tests for the infants who were not treated with somatostatin were all normal. CONCLUSIONS: Repeated doses of somatostatin were effective in reducing chylus production. Administering this treatment earlier should be considered in order to minimize known complications. The only potential side effect observed was primary transient hypothyroidism. Therefore, careful monitoring of the pituitary-thyroid axis is advised.     

Pleural effusions in the neonate

Introduction: Pleural effusions are rare in the neonate and may be associated to several clinical conditions. Only a few series of pleural effusions in the fetus and newborn are described in the literature.

Aim: This study was undertaken to determine more accurately the causes and prognostic significance of pleural effusions in a population of high-risk neonates.

Materials and methods: A retrospective chart review of 62 neonates admitted to the neonatal intensive care unit of six medical centers in the north of Portugal, between 1997 and 2004, that presented the diagnosis of pleural effusion.

Results: 33M/29F newborns; preterms 47 (76%); GA 33 (25-40) wk; BW 1830 (660-4270) g; C-section 39 (63%). Pleural effusions were congenital in 20 (32%) newborns and acquired in 42 (68%). Congenital pleural effusions occurred as fetal hydrops in 11 (18%) patients and as chylothorax in 9 (15%). In four cases of hydrops, the cause was a congenital chylothorax. Congenital chylothorax (n=13) was the most common (65%) congenital pleural effusion in this study. The incidence of congenital chylothorax was 1:8.600 deliveries and male:female ratio was 2:1. Mortality occurred in five newborns due to pulmonary hypoplasia. Traumatic (iatrogenic) were the most frequent (n=31) acquired pleural effusions. These included 8 (13%) cases of hemothorax and 8 (13%) cases of total parenteral nutrition leakage. Pleural effusions after intra-thoracic surgery were mainly (79%) chylothoraces. There were 11 (26%) non-iatrogenic acquired pleural effusions. No mortality was associated with acquired pleural effusions.

Conclusions: Congenital pleural effusions usually occur as hydrops or congenital chylothorax. Traumatic (iatrogenic) are the most frequent acquired pleural effusions in a tertiary NICU. Pleural effusions after intra-thoracic surgery are mainly chylothoraces. Non-iatrogenic acquired pleural effusions are associated to several clinical conditions, and mortality is usually associated to the underlying condition.     

Improving survival of vulnerable infants increases neonatal intensive care unit nosocomial infection rate

OBJECTIVE: To determine the factors associated with an increasing rate of nosocomial infections in infants with very low birth weights. METHODS: Retrospective review of clinical and nosocomial infection databases for all infants with birth weights of 1500 g or less admitted to an academic neonatal intensive care unit between January 1, 1991, and December 31, 1997 (N = 1184). Two study periods were compared: 1991-1995 and 1996-1997. RESULTS: Among the 1085 infants who survived beyond 48 hours, the proportion who developed nosocomial infections increased from 22% to 31% (P =.001) and the infection rate increased from 0.5 to 0.8 per 100 patient-days (P<.001) during the period from 1996 to 1997. In that same period, the median duration of indwelling vascular access increased from 10 to 16 days (P<.001), and the median duration of mechanical ventilation increased from 7 to 12 days (P<.001). Although the device-specific rate of bloodstream or respiratory infections did not change, the increase in infections was directly attributable to the increasing proportion of infants who required these devices. In both study periods, the peak incidence of initial infection occurred between 10 and 20 days of age. For the entire sample, proportional hazard models identified birth weight, duration of vascular access, and postnatal corticosteroid exposure as significant contributors to the risk of infection. CONCLUSIONS: The increasing number of technology-dependent infants was the primary determinant in the increase of nosocomial infections. Because these infections occur in a small proportion of infants, understanding the host factors that contribute to this vulnerability is necessary to decrease nosocomial infections in neonatal intensive care units.     

儿童先天性肺囊性病胸腔镜手术的围手术期并发症

目的:分析儿童先天性肺囊性病胸腔镜手术围手术期并发症的原因,讨论降低并发症发生率的方法。方法:收集2012年5月至2019年12月于北京儿童医院胸外科进行胸腔镜手术的672例肺囊性病患儿的病例资料。其中,男418例,女254例;中位手术年龄为14个月,手术年龄范围为1~189个月。手术方式根据病变范围选择肺叶切除或肺段...     

Surveillance of nosocomial infections: prospective study in a pediatric intensive care unit. Background, patients and methods

BACKGROUND, PATIENTS AND METHODS: From November 1997 through May 1998, the incidence of nosocomial infections was studied prospectively in a 10-bed multidisciplinary pediatric intensive care unit in Germany. A standardized surveillance [SEKI] system based on the National Nosocomial Infection Surveillance [NNIS] System of the Centers for Disease Control and Prevention [CDC] was used. The CDC definitions for nosocomial infections were adapted to the current practice of pediatric intensive care in Germany. Infection rates were calculated as infections per 100 patients, per 1000 patient-days, and per 1000 device-days (central venous catheters, urinary-catheters, and mechanical ventilation). RESULTS: Fifteen nosocomial infections were recorded in 201 patients during 1035 patient-days. The overall nosocomial infection rates were 7.5/100 patients and 14.5/1000 patient-days. Device-associated nosocomial infection rates for urinary-catheters and mechanical ventilation were 7.2/1000 utilization-days and thus below the 75th percentile of the last NNIS report. Central line infection rates were 10.7/1000 utilization days and therefore above the 75th percentile of the NNIS data (10.2/1000). The median length-of-stay was 5.1 days. CONCLUSIONS: Surveillance data are indispensable for internal and external quality control, and prospective surveillance of nosocomial infections should become an essential component of hospital infection control programs in pediatric intensive care in Germany. The standardized calculation of (device utilization ratios and) device-specific infection rates yields results which can be compared with national and international surveillance data. SEKI meets the criteria of a practice oriented, prospective and standardized surveillance system. Considerable efforts for collecting and interpreting the required data should be balanced against the benefit of prevention of nosocomial infections in this population of critically ill persons.     

新生儿重症监护病房的院内感染638例分析

目的调查NICU院内感染的发生情况,探讨其危险因素,为院内感染的防控提供依据。方法对我科2003年5月至2004年12月,住院的638例新生儿进行院内感染的监控,并进行分析和总结。结果638例新生儿中74例发生88次院内感染,发生率为11．6％;住院日相关的院内感染率为14．9／1000NICU病例一天;导管相关血行感染率为18／1000血管内导管一天（2／111）;呼吸机相关肺炎发生率为63．3／1000机械通气一天（15／237）;平均开始出现感染时间（7．98±4．58）d。发生院内感染者比未感染者的胎龄及出生体重低、住院时间延长。新生儿发生院内感染的危险因素包括胃肠外营养、出生体重≤1500g及呼吸机治疗等（P〈0．05）。感染部位中,以肺炎占首位（45．4％）。院内感染病死率为4．1％。入院后有细菌定植者较无定植者院内感染率高（Х^2=79．7,P〈0．001）。结论充分了解NICU中新生儿发生院内感染的高危因素、尽量减少肠外营养及侵袭性操作的次数和时间、明确NICU中患儿个体细菌的定植情况将有助于控制院内感染并对临床合理用药提供参考。     

宫内发现胎儿胸腔积液 生后呼吸困难

新生儿乳糜胸是新生儿先天性胸腔积液的常见病因,常由多种原因导致胸导管及其分支淋巴管破裂,乳糜液在胸腔内蓄积所致。恶性肿瘤所致新生儿乳糜胸极为罕见。该例为国内首例新生儿纵隔神经母细胞瘤合并乳糜胸。男性患儿宫内即发现左侧胸腔积液,出生时窒息,生后以呼吸困难、全身发绀为主要表现。根据胸腔积液常规和生化诊断为左侧乳糜胸。该患儿经过持续胸腔穿刺置管引流、对症支持等多种治疗,左侧胸腔引流量达90~180?mL/d,考虑为新生儿难治性乳糜胸。生后13?d复查胸片提示左上肺野病灶,胸部CT平扫增强提示后纵隔神经母细胞瘤可能。尸检证实为后纵隔巨大神经母细胞瘤（低分化型）,侵犯C₇~T₆椎管及附近竖脊肌,肝脏及双侧肾上腺有少量肿瘤组织。考虑纵隔肿瘤为乳糜胸的根本病因。     

Octreotide in the treatment of congenital chylothorax

Congenital chylothorax is the accumulation of lymphatic fluid within the pleural space. Cases unresponsive to conservative management usually require surgery. Octreotide has been used successfully to treat post-traumatic chylothoraces in the paediatric and adult population. Its exact mode of action is uncertain but it is believed to reduce lymphatic drainage by a direct action on splanchnic lymph flow. We report a case of congenital chylothorax where surgery was avoided with the compassionate trial of the somatostatin analogue, octreotide. Treatment was associated with prompt respiratory improvement prior to cessation of pleural drainage. Further studies are required to ascertain its true value in congenital chylothorax.     

Microbiological factors associated with neonatal necrotizing enterocolitis: protective effect of early antibiotic treatment

AIM: The incidence of necrotizing enterocolitis (NEC) strongly increased in an neonatal intensive care unit (NICU) in 1997 and 1998 compared with previous years, which coincided with increased incidence of nosocomial sepsis. Specific risk factors related to this NICU and a possible relationship between NEC and nosocomial sepsis were studied retrospectively, including all patients with NEC since 1990 and matched controls. METHODS: Clinical and bacteriological data from the period before the development of NEC and a similar period for the controls were collected retrospectively and corrected for birthweight and gestational age. Statistical analysis was performed by a stepwise regression model. RESULTS: Data of 104 neonates with NEC and matched controls were analysed. The median day of onset of NEC was 12 d (range 1-63 d). Significant risk factors for NEC were: insertion of a peripheral artery catheter [odds ratio (OR) 2.3, 95% confidence interval (95% CI) 1.3-3.9] and a central venous catheter (OR 5.6, 95% CI 3.1-10.1), colonization with Klebsiella sp. (OR 3.4, 95% CI 1.5-7.5) and Escherichia coli (OR 2.1, 95% CI 1.0-4.5), and the occurrence of sepsis, in particular due to coagulase-negative staphylococci (OR 2.6, 95% CI 1.4-5.1). The risk for NEC was decreased after the early use (< 48 h after birth) of amoxicillin-clavulanate and gentamicin (OR 0.3, 95% CI 0.2-0.6). CONCLUSION: Insertion of central venous and peripheral arterial catheters is positively associated with NEC, as is colonization with the Gram-negative bacilli Klebsiella and E. coli and the occurrence of sepsis, particularly due to coagulase-negative staphylococci. Early treatment with amoxicillin-clavulanate and gentamicin is negatively associated with NEC and may be protective against NEC.     

Fertility in male patients treated with neoadjuvant chemotherapy for osteosarcoma

PURPOSE: To evaluate fertility in male patients with osteosarcoma after chemotherapy including high doses of alkylating agents. PATIENTS AND METHODS: Postchemotherapy fertility was evaluated in 96 male patients who received chemotherapy at the authors' institution from 1976 to 1996 for localized bone osteosarcoma of the extremities. Four drugs were administrated (doxorubicin, cisplatin, methotrexate, ifosfamide) at different doses according to six different protocols. Eleven patients were prepubertal and 85 were postpubertal at the time of chemotherapy. The median age of these patients at the time of chemotherapy was 17 years (range 10-42), the median age at the time of the interview was 27 years (range 19-53), and the median follow-up from the end of chemotherapy was 9 years (range 4-17). RESULTS: Of the 96 patients, 26 underwent sperm analysis and 20 showed oligo- or azoospermia. Patients who received high-dose ifosfamide showed a higher incidence of azoospermia. Six patients were normospermic and received no ifosfamide or lower doses of ifosfamide. Eight patients fathered a total of 12 children. No birth defects or congenital anomalies were observed in their offspring. CONCLUSIONS: The rate of sterility was related to ifosfamide dosage. It appears that pubertal maturation is not affected by chemotherapy. No congenital malformations were seen in the children of the few patients who fathered children.