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At least 865 records · Page 48Linked to original sources
PubMed2000

Cardiorespiratory function is significantly improved following corrective surgery for severe pectus excavatum. Proposed treatment guidelines.

BACKGROUND: Pectus excavatum is a congenital chest abnormality which may become more marked during childhood and teenage growth. Young teenagers with severe PE deformities often become short of breath during strenuous exercise and complain of becoming easily fatigued. The pathophysiologic effects of PE remain controversial because they have been difficult to measure, especially in young children. Symptomatic patients often have significant subjective improvement during exercise following PE repair and there is a major cosmetic impact on teenagers with severe deformities. This study was designed to measure pulmonary and cardiac function with exercise before and after corrective surgery. METHODS: Pulmonary function testing and incremental exercise testing were performed in 36 adolescents with pectus excavatum (PE) and 10 age-matched, healthy control subjects. Six months after corrective surgery was performed, 15 of these PE patients and 6 control subjects were re-evaluated for pulmonary function. RESULTS: Before surgery, PE subjects had a lower forced vital capacity (FVC) than controls; there was no change in FVC after surgery. Before surgery, 58% of PE patients had subjective complaints of exercise limitation; 66% of the patients were significantly improved after surgery. PE subjects exercised at a similar workload to controls. Respiratory parameters during exercise were similar between the two groups before surgery, indicating that exercise was not limited by restrictive lung disease. After surgery, PE subjects exercised longer and had a higher oxygen pulse than before surgery, whereas controls showed no such changes. Although some PE subjects showed mild restrictive lung function, surgical repair did not influence this mild degree of restriction. CONCLUSIONS: After corrective surgery, PE patients have increased exercise tolerance and a higher oxygen pulse. Oxygen pulse is a measure of cardiac output. Results suggest that PE repair improves cardiopulmonary function during vigorous exercise. Based upon these studies, and our experience in the treatment of more than 700 surgical patients with pectus excavatum over a 40-year period, guidelines for the diagnosis and management of children with pectus excavatum are proposed.

Adolescent↗
PubMed2006

Family study of the inheritance of pectus excavatum.

BACKGROUND: The most common congenital deformity of the chest wall is pectus excavatum, a malformation that is present in between 1 in 400 and 1 in 1000 live births and causes the body of the sternum to be displaced, producing a depression. There are many different shapes of the pectus, and multiple factors probably contribute to the final form. The etiology of pectus excavatum is uncertain, but a familial tendency has been found in clinical experience, where it may be seen in more than one sibling. Pectus excavatum is commonly associated with connective tissue disorders such as Marfan and Ehlers Danlos syndromes. Extensive literature review failed to identify articles documenting families with multiple affected members. PURPOSE: The purpose of this study was to collect evidence that pectus excavatum is familial and may be an inherited disorder. METHODS: Using the Children's Surgical Specialty Group database at Children's Hospital of The King's Daughters, families with more than one affected individual were selected. With Institutional Review Board-approved informed consent, 34 families agreed to participate. Family histories were obtained, and a 4-generation pedigree was constructed for each family. Forty questions were asked about each individual's medical history, and comprehensive systems review included features of connective tissue-related problems. Inheritance patterns for each family were determined by pedigree analysis. RESULTS: A total of 14 families suggested autosomal dominant inheritance, 4 families suggested autosomal recessive inheritance, and 6 families suggested X-linked recessive inheritance. Ten families had complex inheritance patterns. Pectus excavatum occurred more frequently in males than in females (1.8:1). Long arms, legs, and fingers; high-arched palate; mitral valve prolapse; heart arrhythmia; scoliosis; double jointedness; flexibility; flat feet; childhood myopia; poor healing; and easy bruising were commonly associated with pectus excavatum. CONCLUSIONS: Pedigree analysis of 34 families provides evidence that pectus excavatum is an inherited disorder, possibly of connective tissue. Although some families demonstrate apparent Mendelian inheritance, most appear to be multifactorial.

Female↗
PubMed2002

Early experience with the Nuss minimally invasive correction of pectus excavatum in adults.

Nuss described a minimally invasive technique for correcting pectus excavatum in children. A curved stainless-steel bar is inserted behind the sternum through the chest cavity with the convex surface face down, then rotated 180 degrees to elevate the sternum and correct the deformity. The procedure gained wide acceptance in children. The purpose of this study was to determine if the procedure is effective in adults. Only patients with symptoms limiting lifestyle, chest wall indices higher than 3.25, and demonstrable cardiac compression on echocardiography were accepted. Between April 1998 and January 2001, 14 adults aged 19 to 46 underwent assessment for the Nuss minimally invasive technique for correction of pectus excavatum. Eight patients, 19 to 32 years of age, met the stated criteria for acceptance. The comorbidities were 2 asymmetrical deformities, 2 scolioses, 1 previous pectus repair, and 1 previous breast augmentation. The patients were informed of the benefits and disadvantages of both the Ravitch and the Nuss procedures. All patients except the first had talked to one or more adults who previously had the procedure. Follow-up was 7 to 37 months (mean 22.1 months). Four patients have had their bars removed and maintained correction. Success of the operation was based on relief of cardiac compression, alleviation of symptoms, and adequate pain control. Operating time was 1 to 2:05 hours (mean 1:32 hours). Complications were pneumothorax in one patient, urinary retention in 2, and left lower lobe atelectasis in 5. Complications did not prolong hospitalization. Postoperative epidural analgesia was discontinued after 2 to 4 days (mean 2.8 days). Average daily pain scores were between 1.6 and 3.7 on a scale of 0 to 10. Hospital stay was 3 to 5 days (mean 4 days). Relief of symptoms and increase in activity were obtained in all patients. Relief of cardiac compression was demonstrated in the 6 patients who have had postoperative echocardiograms. Patients returned to normal activity 2 to 4 weeks postoperatively (mean 2.3 weeks). Duration of pain medicine was 2 to 4 weeks in 6 patients and 2 and 4 months for the other 2 (mean 5.5 weeks). There were 2 late complications related to the bar, but without loss of correction. The early experience with the Nuss minimally invasive pectus excavatum repair in adults is encouraging. The procedure is effective for correcting pectus excavatum in selected patients. Early results are dependent upon adequate bar stabilization and pain control. The long-term results in adults are unknown.

Adult↗
PubMed1984

Laryngomalacia. A review and case report of surgical treatment with resolution of pectus excavatum.

Laryngomalacia has been described in the medical literature for over 100 years. During this time many different names have been used for the same condition. There have been numerous discussions of symptoms, cause, pathogenesis, relationships to other conditions, hereditary factors, and treatment. The purpose of this study is to historically review the literature, clarify terminology, and describe the clinical aspects of the disease. We also summarize and make conclusions regarding cause, pathogenesis, genetic and disease relationships, prognosis, and treatment. Finally, we present a case report of severe laryngomalacia and describe a surgical technique used to treat it.

Epiglottis↗
PubMed2005

Effect of pectus excavatum deformity on cardiorespiratory fitness in adolescent boys.

OBJECTIVE: To determine the magnitude of the effects of pectus excavatum deformity on endurance fitness and cardiorespiratory functional reserve in adolescent boys. DESIGN: Cross-sectional comparison of cardiac and ventilatory variables at rest and during a maximal cycle exercise test. SETTING: Pediatric exercise-testing laboratory. PARTICIPANTS: Twelve boys (mean +/- SD age, 14.1 +/- 1.8 years; age range, 11.8-18.0 years) with moderate-to-severe pectus excavatum deformity (mean +/- SD Haller index, 3.95 +/- 0.88) and 20 control boys (mean +/- SD age, 12.5 +/- 0.4 years; age range, 12.1-13.5 years) without musculoskeletal deformity. MAIN OUTCOME MEASURES: Endurance fitness (physical work capacity); respiratory rate, tidal volume, and minute ventilation; and cardiac output and stroke volume by Doppler echocardiography. RESULTS: Patients with pectus deformity had significantly lower endurance fitness than controls (mean +/- SD physical work capacity, 2.60 +/- 0.28 W . kg(-1) vs 3.11 +/- 0.45 W . kg(-1)) and reduced mean +/- SD values for maximal cardiac index (10.6 +/- 1.6 L . min(-1) vs 12.0 +/- 2.2 L . min(-1)) and peak tidal volume (3.02 +/- 0.27 mL . kg(-1) . 10(-2) vs 3.46 +/- 0.30 mL . kg(-1) . 10(-2)). However, considerable overlap was observed in these values between the 2 groups. CONCLUSIONS: As a group, boys with pectus excavatum deformity have lower endurance fitness than controls, and this is associated with reduced cardiac output and tidal volume responses to exercise. However, the wide variability of these measures makes it difficult to assign pectus deformity as a cause of exercise intolerance in individual patients.

Adolescent↗
PubMed1978

Pectus excavatum repair using autologous perichondrium for sternal support.

The support of the lower sternum following repair of pectus excavatum using autologous perichondrium from the lower costal cartilages to supplement the standard Ravitch's repair has been used effectively in 29 children during the past 14 years. This technique helps prevent recurrent sternal depression, particularly when the operation is performed in preadolescent children. Most adolescents and adults with pectus deformities as well as patients requiring reoperation will benefit from an internal prosthesis for the sternum that supplements the perichondrial support to reduce the frequency of subsequent sternal depression. Using the described operative technique, an excellent cosmetic and functional result was obtained in 27 children. Recurrent sternal depression of mild degree that occurred in two patients in the first four months postoperatively was related to trauma and possibly to decreased vascular supply to the lower sternum but neither required additional therapy. The internal mammary vessels should be preserved with the lower sternum whenever feasible.

Adolescent↗
PubMed1999

Interesting inferior vena cava.

This article presents, as diagnostic problems, computed tomographic (CT) images from a patient with unusual cross-sectional anatomy at the thoraco-abdominal region. The introduction and discussion emphasize the solution and explain the underlying clinical condition and resultant radiographic and computed tomographic imaging features.

Aged↗
PubMed1990

Mild form of Jeune syndrome in two sisters.

Jeune syndrome is characterized by respiratory distress, osseous dysplasia, and short stature. Patients generally die during the first months of life. However, some cases with milder clinical manifestations have been described; these cases show characteristic renal involvement of different severity. The authors report on two cases of the mild form of Jeune syndrome in sisters.

Asphyxia Neonatorum↗
PubMed1994

Newly recognized autosomal recessive faciothoracoskeletal syndrome.

We report on 2 brothers, born to consanguineous parents presenting thin/long face, small ears, blepharophimosis, malar hypoplasia, long neck, pectus excavatum, brachycamptodactyly, and sacral dimple. We suspect that these patients represent a previously undescribed autosomal recessive syndrome.

Abnormalities, Multiple↗