History Of Exercise Intolerance Research Articles

Case Report of a Dog Heavily Infected With Hepatozoonosis

Canine hepatozoonosis is a widespread tick-borne protozoan disease and generally affects domestic dogs. Diagnosis can be made via PCR, serology, and direct observation of the gamonts of the parasite on stained blood smears. In the present case, a four-year-old female crossbred hunting dog presented to Balikesir University Veterinary Faculty clinics of internal medicine with a history of exercise intolerance, and weight loss for ten days. The dog was highly infected and showed anemia, thrombocytopenia, and neutrophilia. In radiographic analysis, unilateral pulmonary pathology was determined. This case report can be important for clinicians who live in Turkey to recognize the parasite.

Clinical and Laboratory Findings on Glycogen Storage Disease Type V: Results from a Retrospective Observational Study in a Tertiary Hospital.

Introduction - Glycogen storage disease type V (GSDV, MIM #232600) is an autosomal recessive metabolic myopathy caused by pathogenic variants in the PYGM gene. The characteristic symptoms of exercise intolerance, myalgia, and cramps, which improve after a few minutes of rest, are frequently unrecognized in affected children. When there is clinical suspicion, the initial approach with a forearm exercise test has diagnostic value by detecting low post-exercise plasma lactate-to-ammonia ratio values. The diagnostic algorithm is followed by genetic testing if the results suggest myophosphorylase deficiency. Methods - This was a retrospective observational study conducted based on reviewing medical records of patients with GSDV in a tertiary hospital. We assessed demographic variables, including the timing of onset and diagnosis, relevant clinical characteristics, and whether genetic testing was performed, including its results. Results/Case Report - Our goal was to review the GSDV cases in our center to assess our cohort's diagnostic timing and clinical and genetic characteristics. We identified 28 patients from 24 families, three with consanguinity. The mean age at the time of the study was 43 years. While most (26/28; 93%) recalled their first symptoms in childhood/adolescence, only 25% (7/28) were diagnosed then. All patients had exercise intolerance and CK elevation, while about half reported the second wind phenomenon. Genetic testing was performed in 22 patients, revealing biallelic PYGM variants (9 homozygous, 13 compound heterozygous) as the most common (p.R50*). Conclusion - GSDV is rare and presents in the pediatric age, with subtle manifestations often underestimated for decades. A late diagnosis may negatively impact the psychosocial development of affected children. It is essential to recognize some unique features that facilitate diagnosis: history of exercise intolerance, the second wind sign, and high resting serum CK levels. Identifying the disease-causing variants in PYGM is currently the gold standard for diagnosis as it is less invasive than performing a muscle biopsy, and may promptly diagnose the condition and avoid wrongful labelling of patients.

A Case of Left Atrial Myxoma Diagnosed on Routine Preoperative Evaluation

Cardiac tumors are very rare and myxoma is the most common primary cardiac tumor. Cardiac myxomas frequently originate from the left atrium and predominantly affect women. We present a case of a 65-year-old male who was admitted for hernia surgery and underwent preoperative evaluation. On evaluation, he was found to have a diastolic murmur on auscultation and had a history of exercise intolerance, and a transthoracic echocardiogram was done. It revealed a large mass in the left atrium attached to the interatrial septum suggestive of left atrial myxoma. He then underwent surgical resection of the tumor. The lack of recognition of this condition by appropriate history and physical exam can lead to a delay in diagnosis and treatment and the risk of cardiovascular complications.

Diagnóstico ecocardiográfico de defeito do septo interventricular com agravo de síndrome de Eisenmenger em cão adulto – relato de caso

Interventricular septal defect (IVSD) is a congenital heart disease characterized by communication or non-closure of the interventricular septum in the embryonic or postnatal period, causing a failure in the separation between the systemic and pulmonary circulation. The severity of the hemodynamic consequences depends on the extent of the defect and the magnitude of the flow through it. Blood flow commonly occurs from left to right, because systemic vascular resistance is greater than pulmonary resistance. This report describes the echocardiographic findings in a 4-year-old male Poodle treated at the Veterinary Hospital with a history of exercise intolerance, cyanosis and episodes of syncope. On echocardiography, perimembranous IVSD was diagnosed with atrial and right ventricular dilatation, discontinuity of the interventricular septum in the subaortic region measuring approximately 5 mm. In the Doppler evaluation, reverse turbulent flow was evidenced, characterizing shunt from right to left (Eisenmenger syndrome). Despite preserved systolic function, abnormal relaxation was observed. Doppler echocardiography was a fundamental complementary imaging test for the diagnosis of this congenital heart disease since its suspicion is not commonly raised in adult patients. Doppler echocardiography allowed us to identify the defect, define its size and extension, determine the direction of blood flow as well as its hemodynamic consequences.

The phenotypic and genotypic features of a Scottish cohort with McArdle disease

This retrospective study evaluated the phenotypic and genotypic features of 14 patients with McArdle disease attending the West of Scotland adult muscle clinic. Although all patients experienced exercise-induced cramps, exercise intolerance and hyperCKaemia, only 71% (n = 10) experienced the second wind phenomenon, rhabdomyolysis and/or myoglobinuria. We observed a high rate of fixed muscle weakness (50%; n = 7), coronary artery disease (36%; n = 5), and psychological comorbidity (50%; n = 7). Although 79% had symptom onset in the first decade of life, the mean age at presentation and at genetic diagnosis was 43.8 years and 47.7 years, respectively. 93% had at least one copy of the common PYGM pathogenic variant, c.148C > T, p.(Arg50*), with 50% (n = 7) of the cohort being homozygous. Our cohort highlights the phenotypic variability seen in McArdle disease and underscores the potential for late-onset presentations. It emphasises the need for improved awareness and recognition of this condition amongst neurologists, rheumatologists and general physicians. A history of exercise intolerance and second wind phenomenon may not always be volunteered by the patient, underscoring the need to ask specific questions in clinic to extrapolate the relevant symptoms in this patient cohort.

Diagnosis of Myxomatous Mitral Valve Disease (MMVD) in Aged Dogs

Background: Aging is a biological process, which affects many body systems and increases susceptibility to disease that is associated with a reduction in immune response, a decline in cognitive function and a diminished functional reserve of the cardiovascular, pulmonary and renal systems. Diseases of heart or cardiovascular system represents a major component of geriatric medicine among which, myxomatous mitral valve disease is more common among geriatric dogs. The present study is keeping on record about the diagnosis of myxomatous mitral valve disease in geriatric dogs in and around Hyderabad, Telangana state.Methods: A total of1695 geriatric dogs presented to various clinics including university veterinary clinical complex of Hyderabad, Telangana state during 2015 to 2020 were considered for this study. After confirming that they were free from any other systemic disease, medium to large size breed dogs above 8 years with the history of exercise intolerance, dyspnoea and cough were selected. Diagnostic protocol included, auscultation, chest x-ray and 2d-echocardiography to confirm mitral valve degeneration. Result: Myxomatous mitral valve disease was diagnosed in 427/695 geriatric dogs (61.43%) and were showing hyperechoeic thick mitral valve leaflets with kinking and prolapse. Further, echocardiography also revealed dilated left atrium and ventricle, hyperkinesis of interventricular septum, mitral valve degeneration and rupture of chordae tendineae with valuvular regurgitation.

Novel TTN mutations and muscle imaging characteristics in congenital titinopathy.

ObjectiveWe present clinical features, muscle imaging findings, and genetic characteristics of five unrelated Chinese patients with congenital titinopathy, emphasizing the diagnostic role of muscle MRI.MethodsFive patients who recessive titinopathies were recruited. All patients received muscle biopsies. Mutations were detected by panel massively parallel sequencing and confirmed by Sanger sequencing. Western blotting of muscle proteins was performed. Leg muscle MRIs were performed in four patients.ResultsFour patients aged 1–4 years old showed delayed motor development from early infancy, while a 17‐year‐old boy showed only a 1‐year history of exercise intolerance. Physical examination showed proximal weakness in three patients. Muscle biopsies demonstrated multiple myopathological changes, including increased internalized nuclei, multicores, central cores, and dystrophic changes. Genetic sequencing revealed compound heterozygous or homozygous novel TTN mutations, including six frameshift mutations, one nonsense mutation, two missense mutations, one splicing mutation, and one small nonframeshift deletion. Protein analyses revealed significant decrease of full‐length titin in all patients. Thigh muscle MRIs in four patients showed prominent fatty infiltration in the upper portion of semitendinosus and the peripheral portion of gluteus medius, while the sartorius and gracilis were relatively preserved.InterpretationThese cases provided further evidence that TTN mutations are likely responsible for an increasing proportion of congenital myopathies than currently recognized. The novel mutations reported expand the mutation spectrum of the TTN gene. There is a characteristic pattern of muscle involvement in congenital titinopathy regardless of clinical or pathological phenotype, providing valuable clues for guiding a genetic diagnosis workup.

Nuss Procedure for a Patient with Negative Haller Index

Introduction Minimally invasive repair for pectus excavatum (MIRPE) is controversial in extremely severe cases of pectus excavatum (PE) and an open repair is usually favored. Our aim is to describe a case of a patient with an extremely severe PE that underwent a minimally invasive approach. Case report An 8-year-old girl with severe sternum depression was assessed. She had a history of exercise intolerance, nocturnal dyspnea, fatigue, and shortness of breath. Chest computed tomography showed that sternum depression was posterior to the anterior vertebral column; therefore, Haller and correction index could not be measured. Spirometry indicated an obstructive ventilation pattern (forced expiratory volume in 1 second = 74.4%), and echocardiogram revealed a dilated inferior vena cava, mitral valve prolapse with normal ventricular function. After multidisciplinary committee evaluation, a MIRPE approach was performed. All symptoms had disappeared at the 3-month postoperative follow-up; the desired sternum shape was achieved and normalization of cardiopulmonary function was observed. The Nuss bars were removed after a 2-year period. After 18-month follow-up, the patient can carry out normal exercise and is content with the cosmetic result. Conclusion Nuss procedure is feasible in our 8-year-old patient. In this case, both the Haller and correction index were not useful to assess the severity of PE. Therefore, under these circumstances, other radiologic parameters have to be taken into consideration for patient evaluation.

A case of an unexplained eosinophilic myocarditis in a dog

An 8-year-old spayed female Munsterlander was evaluated for a chronic low grade fever and a two month history of exercise intolerance. On physical examination, tachycardia and a grade II/VI right systolic heart murmur were detected. Echocardiography revealed marked thickening of the atrial and ventricular walls with mixed echogenicity and concentric hypertrophy of the left and right ventricles and equivocal systolic dysfunction. Serum cardiac troponin I level was markedly elevated. Endomyocardial biopsy was attempted; however, the patient arrested during the procedure and resuscitation was unsuccessful. Post-mortem examination revealed severe, chronic atrial and ventricular eosinophilic myocarditis associated with marked interstitial fibrosis. Serological testing, histopathology and immunohistochemistry staining did not reveal an underlying infectious agent or neoplasm. To our knowledge, this is the first reported case of primary eosinophilic myocarditis in the absence of a peripheral eosinophilia and multi-organ eosinophilic inflammation in a dog.

Anesthesia case of the month. Diaphragmatic herniation.

History A 7-year-old sexually intact female 7.4-kg (16.3-lb) Jack Russell Terrier with a history of exercise intolerance, coughing, and weight loss was referred to the Companion Animal Clinic of the Faculty of Veterinary Medicine, Ghent University. The dog had been hit by a car 2 years previously. Thoracic radiographs were taken immediately after the accident at a local veterinary clinic, and a diagnosis of pleural effusion was made. The dog was subsequently hospitalized for several days. After discharge, the dog never completely regained its former activity level. Over the months preceding referral, the dog gradually became exercise intolerant, lost weight, and started to cough. Food and water intake were normal, and neither vomiting nor diarrhea was reported. The dog was currently not receiving any medications. On physical examination, the dog was bright, alert, and responsive, with a body condition score of 2 of 5. On thoracic auscultation, muffling over the entire right and left ventral aspects of the thorax was noticed; however, in the left dorsal thoracic quarter, lung and heart sounds could be evaluated. Respiratory rate was 64 breaths/min, and heart rate was 108 beats/min. No murmurs or arrhythmias were detected. Metatarsal pulses were strong, symmetric, and regular. Mucous membranes were pink, and capillary refill time was normal. Rectal temperature was 39.1°C (102.4°F). On thoracic radiographs (laterolateral and dorsoventral views), a diaphragmatic hernia was visible, with presence of the stomach, small intestines, and liver in the thoracic cavity. Results of a preanesthetic CBC, serum biochemical analysis, and measurement of electrolyte concentrations were within reference limits. A 22-gauge catheter was placed aseptically in the right cephalic vein. The dog was hospitalized overnight for observation, and surgery (herniorraphy and elective ovariectomy) was scheduled for the next morning. Case Management Results of preanesthetic clinical examination on the morning of surgery were unchanged from the day of admission, and the dog was classified as American Society of Anesthesiologists class III (ie, severe systemic disease). The dog was preoxygenated (2 L/min via a face mask) for 5 minutes while propped up on a tilted table. Premedication consisted of fentanyl (5 g/kg [2.27 g/lb],

Performance of serum and dried blood spot acylcarnitine profiles for detection of fatty acid β-oxidation disorders in adult patients with rhabdomyolysis

Plasma/serum and dried blood spot (DBS) acylcarnitine profiles (ACPs) are key to the diagnosis of mitochondrial fatty acid β-oxidation disorders (FAODs). Despite their significant clinical applications, limited published data exists to compare their sensitivities and specificities. We retrospectively evaluated these two methods in adult patients with a history of rhabdomyolysis; investigated for an underlying FAOD. A retrospective study was completed for adult patients (investigated between 2003 and 2011) meeting the inclusion criteria of a history of recurrent rhabdomyolysis or one episode of rhabdomyolysis with a history of exercise intolerance. All subjects underwent investigations for an underlying FAOD including DBS and serum ACP analysis concurrently collected during a symptom-free period, and skin biopsy for cultured fibroblast fatty acid oxidation studies or enzyme activity measurement, as indicated, with or without molecular confirmation. Their medical records were reviewed, and the performance of the two methods were compared. Seven out of 31 subjects (22.6%) were diagnosed with an underlying FAOD. Long chain acylcarnitines were more markedly elevated in serum samples from confirmed CPTII cases (n = 4) as compared to matched DBS profiles. The sensitivity and specificity of DBS ACP was 71.4% (95% CI, 0.30-0.95) and 100% (95% CI, 0.79-1.00), respectively, compared to a sensitivity of 100% (95% CI, 0.56-1.00) and a specificity of 94.7% (95% CI, 0.72-1.00) for serum ACP. FAODs appear to be a common cause of recurrent rhabdomyolysis or rhabdomyolysis with a history of exercise induced myalgia. At least historically, FAODs maybe underdiagnosed in adults with rhabdomyolysis. This study suggests that serum ACP might be more sensitive than DBS ACP for detection of an underlying FAOD in adults with rhabdomyolysis while asymptomatic.

G.P.115 Recurrent myoglobinuria and deranged acyl-carnitines due to mutation of the COII gene of mtDNA

Abstract Mitochondrial myopathies generally present with exercise intolerance in which the typical symptoms constitute breathlessness and fatigue on exercise. Exercise-induced rhabdomyolysis and myoglobinuria occur very rarely. This case presents the story of a 43-year old man with a life-long history of exercise intolerance, associated with recurrent episodes of exercise-induced myoglobinuria. His parents sought medical assistance when he as a young boy could not keep up with his younger sister when walking. He was believed to be affected by multiple acyl-CoA dehydrogenase deficiency (MADD), because of his symptoms, and because his acyl-carnitine profiles looked typical for MADD. He was treated with riboflavin, which however, worsened his symptoms. Results: On testing, the patient had a normal neurological examination, except for mild weakness in hip flexion bilaterally. Resting plasma lactate ranged from normal to 3.8 mmol/L. A muscle biopsy showed only mild myopathic changes, no abnormal lipid accumulation, approximately 40% COX-negative fibers, and a few ragged blue fibers and multiple fibers with subsarcolemmal accumulation of mitochondria. Biochemical assessment showed a selective complex IV deficiency (20% of normal). No mutation in the three genes responsible for MADD could be found, but sequencing of the mitochondrial genome revealed a novel, frame-shifting mutation in the COII subunit gene (8156dupG). The mutation load in muscle was 56%. The mutation was not present in fibroblasts or blood from the patient. In accordance with the exercise intolerance, the patient had a peak oxygen uptake of 23 ml O 2 /min/kg on cycle testing, which is about 60% of normal for the age. Mutations in COII of mtDNA have only been reported in a handful of patients, and the present case demonstrates that a phenotype of a disorder of lipid oxidation can be mimicked with this defect, with recurrent episode of myoglobinuria and acyl-carnitine profiles reminiscent of MADD.

ALCAPA Presents in an Adult with Exercise Intolerance but Preserved Cardiac Function.

Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital anomaly that usually manifests as severe left-sided heart failure and mitral valve insufficiency during the first one to two months of life. The majority of these cases die in infancy if not corrected early upon presentation. Adulthood presentation is rare and most of the untreated patients who reach adulthood present with left ventricular dysfunction, severe mitral regurgitation, and sometimes myocardial infarction. Here we report a case of a 20-year-old woman with a history of exercise intolerance since childhood that was misinterpreted as asthma until a 2D-Echo revealed ALCAPA with RCA collaterals to the left anterior descending artery, preserved LV ejection fraction, and absence of apparent mitral valve abnormality. One month after the ALCAPA diagnosis, she successfully underwent surgical reconstruction of left main and pulmonary artery without any major complications. She had normal left ventricular function without apparent ischemic cardiac symptoms eighteen months after procedure.

Treatment of traumatic tracheal collapse with extraluminal titanium mesh screens

SummaryA 12‐year‐old Thoroughbred gelding presented with a history of exercise intolerance and inspiratory stridor. Physical examination revealed a blunt crush injury to the ventral neck and underlying trachea of unknown duration. Tracheoscopy revealed a grade 3 tracheal collapse. Surgical management of the case involved placement of extraluminal titanium mesh screens circumferentially around the trachea for complete support. During the period of hospitalisation, the only post operative complication was seroma formation, which resolved with routine care. Follow‐up after 5 years revealed no further evidence of exercise intolerance, inspiratory dyspnoea or stridor. The horse returned to his previous level of work as a show hunter.

Révélation d’une maladie de Mc Ardle par une intolérance musculaire à l’effort associée à une rhabdomyolyse sévère

McArdle's disease (MAD) is a rare hereditary myopathy secondary to a deficit in myophosphorylase, an essential enzyme for the use of muscular glycogen reserves. Exercise intolerance to a variable degree is the fundamental manifestation. Muscular enzymes are usually normal or slightly elevated, except during episodes of rhabdomyolysis. Generally, the electromyogram has poor sensitivity for the diagnosis of exercise myopathies. The muscular biopsy can be misleadingly normal. The role of MRI in the diagnosis of MAD is not well clarified in the literature. We report the case of a 16-year-old patient, hospitalized in July 2008 for exercise intolerance. On admission, he was asymptomatic and the physical examination was non contributive. Serum creatine kinase levels and renal function measures were normal. Cycloergometer exercise testing unmasked the disease. EMG and muscular biopsies were normal. During the second hospitalization, this time for rhabdomyolysis, T2 weighted MRI of the thighs showed high intensity signals from the gracilis muscles. The control MRI, made after 2 weeks of rest, was normal. Right gracilis muscle biopsy demonstrated excess glycogen with myophosphorylase deficiency, establishing the diagnosis of MAD. MAD is a rare metabolic myopathy to consider in patients with a history of exercise intolerance. The muscle biopsy can be misleadingly normal and should be, to our opinion, be guided by MRI findings.

Association between bronchoalveolar lavage cytologic features and airway reactivity in horses with a history of exercise intolerance

SUMMARY Objective To correlate indices of airway reactivity to bronchoalveolar lavage (BAL) fluid cytologic features in horses with a recent decline in exercise tolerance. Animals 20 actively working horses from 2 to 24 years old. Procedure Bronchoalveolar lavage fluid samples were obtained and analyzed. Forced oscillatory mechanics (1-7 Hz) technique was used for measurements of total respiratory system resistance (RRS), compliance (CRS), and resonant frequency (fres). Changes in RRS (1 Hz) during histamine challenge were used to generate histamine dose-response curves, from which the provocative concentrations that evoked a 75 or 100% increase in baseline RRS (PCRRS75 and PCRRS100, respectively) were determined. Age, sex, baseline lung mechanics, and BAL cytologic findings were correlated with PCRRS75 and PCRRS100. Results No horse of the study had clinical signs or history of obstructive pulmonary disease or increased percentage (> 7%) of neutrophils in bronchoalveolar lavage fluid samples. Mean (± SEM) RRS, CRS, and fres were 0.67 ± 0.06 cm of H2O/L/s, 0.52 ± 0.04 L/cm H2O, and 2.46 ± 0.02 Hz, respectively. There was no correlation between age or sex, and RRS, CRS, fres, PCRRS75, or PCRRS100. There was a significant correlation (rs = −0.78, P < 0.001) between percentage of BAL fluid mast cells and PCRRS75 or PCRRS100, but correlation with other cell types and indices of airway reactivity were not observed. Conclusion The strong association between mast cell percentage in BAL fluid and airway reactivity in this group suggests that mast cell products may contribute to bronchospasm, airway wall thickening, and/or loss of elastic recoil, which underlie airway hyperreactivity. Alternatively, mast cells may contribute to nonspecific airway reactivity in horses through unknown mechanisms. (Am J Vet Res 1998;59:176–181)

Evaluation of ventricular septal defects in horses using two-dimensional and Doppler echocardiography.

Ventricular septal defects (VSDs) were diagnosed in 27 horses; in 26 affected horses systolic murmurs were detected over both sides of the chest. Holodiastolic decrescendo murmurs were also detected in 5 horses. Standardbreds and Arabian horses were over-represented, while Thoroughbred horses were under-represented, when compared to the hospital population (P < 0.0001). Five horses had previously raced successfully, one 2-year-old was training successfully and close to racing, and 4 horses had competed successfully in other types of competition. Eleven horses had a history of exercise intolerance or poor performance, 5 horses were stunted and 3 horses were in congestive heart failure at the time of presentation. The VSD murmur was detected as an incidental finding in 14 horses. Membranous VSD were most commonly detected (in 23/27 affected horses) and were typically found underneath the septal leaflet of the tricuspid valve and the right and/or noncoronary leaflet of the aortic valve. Muscular VSDs were much less common and were located in any portion of the muscular septum. The VSDs ranged in size from 1-4.6 cm (maximal diameter) in affected horses. A left to right shunt through the VSD was detected in 26/27 affected horses with Doppler echocardiography. The peak velocity of shunt flow detected through the VSD was 0-5.8 m/s. The interventricular pressure gradients estimated from the peak shunt velocity obtained with Doppler echocardiography were 0-135 mmHg. Right ventricular pressures estimated with Doppler echocardiography were 15-84 mmHg, similar to invasively obtained measurements of right ventricular pressure in 80% of horses in which right sided cardiac catheterisation was performed (n = 5). Left ventricular and left atrial volume overload was detected in the majority (23/27) of horses. Right atrial and right ventricular volume overload was severe in 3 horses with muscular VSDs, mild in 1 horse with a perimembranous VSD, and mild in 2 horses with membranous VSDs. Concurrent left ventricular dysfunction was detected in 2 horses. Aortic valve prolapse was seen in 7 horses associated with the membranous location of the VSD; 6 of these horses had very mild (1+) or mild (2+) aortic regurgitation. Severe (4+) aortic regurgitation was present in one horse, severe mitral regurgitation in 2 horses, severe tricuspid regurgitation in 3 horses, and severe pulmonary regurgitation was detected in 2 horses. Mitral valve prolapse, tricuspid valve dysplasia, a flail aortic valve leaflet, and a bicuspid pulmonary valve were additional findings detected in one horse each. Post mortem examinations were performed in 8 horses which confirmed the echocardiographic findings. Sixteen out of 27 horses had a history of racing or competing successfully either before or after the diagnosis of the VSD. Two horses were useful pleasure horses, 3 horses presented in congestive heart failure, 1 horse developed signs of congestive heart failure, 2 horses were lost to follow-up and 2 are still alive but are small and stunted. The successful racehorses usually had a membranous VSD that measured < or = 2.5 cm in its largest diameter and a peak velocity of shunt flow through the VSD of > or = 4 m/s. Two-dimensional (2-D) and Doppler echocardiography is useful in assessing the haemodynamic significance of VSDs in horses and can be used to help formulate a prognosis for life and performance.

Severe Rhabdomyolysis in Well Conditioned Athletes

Two well conditioned high school athletes developed rhabdomyolysis after rather modest exercise in activities for which they had not specifically trained-weight lifting and playing bongo drums. Neither patient had a history of exercise intolerance or rhabdomyolysis before. The extent of the rhabdomyolysis in the first patient was extreme (CPK 181,690), and he did not develop renal impairment. The second patient's course was complicated by the use of a contrast study, and he developed marked renal impairment.

Cramps following exercise

A 13-year old boy presented with a 10-year history of severe muscle cramps experienced an hour after prolonged exercise. There was no history of exercise intolerance or myoglobinuria. A muscle biopsy showed a lipid myopathy and a deficiency of muscle carnitine palmityl transferase. He has responded to a high carbohydrate, low fat diet with added carbohydrate intake preceding extensive exercise. Diagnosis of this entity before an episode of rhabdomyolysis is unusual.

Role of Hyaline Membrane Disease in Production of Later Childhood Lung Abnormalities

Between 1961 and 1970, 177 survivors of hyaline membrane disease have been followed clinically and roentgenologically for a minimum of three years to determine the long-term pulmonary outcome. Of these, 153 have been seen for at least six years and 61, for 11 years or more. A total of 175 batteries of pulmonary function tests have been performed on 129 children at age 7 and/or 11 years, Final roentgenograms showed fibrosis in 12 instances, and these changes were positively correlated with the severity of the initial disease, the use of a positive pressure respirator (but not negative pressure respirator), time on the respirator, and length of time in O2, but not high O2 concentrations. Residual symptoms in the first years of life were also correlated with positive pressure respirator use, length of use, and with secondary infection. Pulmonary function tests showed minimal abnormalities in 12 children when last seen. None of these children had a history of exercise intolerance or chronic respiratory symptoms. The ultimate pulmonary history of this population of patients is unknown, but overt childhood lung disease following hyaline membrane disease is not a frequent occurrence.