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Strangulated Diaphragmatic Hernia in a Patient with Osteogenesis Imperfecta

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Strangulated Diaphragmatic Hernia in a Patient with Osteogenesis Imperfecta Strangulated diaphragmatic hernia in a patient with osteogenesis imperfecta pain. The day before admission se- limits. It appeared from a chest Paul A. Spence,* MD vere pain had developed in the left x-ray film that the left hemidia- Zane Cohen,t MD, FRCS[C] upper quadrant; it was followed by phragm was elevated and that there Tomas A. Salerno,t MD, FRCS[C] colic in the midline of the lower was a gas-containing structure be- abdomen. She was nauseated but neath it. Intravenous pyelography had not vomited. Several hours after yielded normal results. Roentgenog- Hernias are common in patients with the onset of a dull ache in her left raphy of the upper gastrointestinal osteogenesis imperfecta, but to our shoulder she presented to hospital. tract demonstrated a normal esoph- knowledge there have been no reports Osteogenesis imperfecta had been agus, stomach and duodenum. The of diaphragmatic hernias in such diagnosed when she was a child. She gastroesophageal junction was below patients. We describe a patient with had suffered 14 major long-bone the diaphragm. The gas-containing osteogenesis imperfecta in whom a fractures and had undergone re- structure was then seen to be above diaphragmatic hernia contained a placement of the femoral and hu- the diaphragm, and a herniation of strangulated segment of the splenic meral heads. She had worn a hear- the splenic flexure of the colon was flexure of the colon. Resection of the ing aid for many years. suspected. necrotic tissue and transverse colos- At the time of admission she was Four days after her admission the tomy resulted in an uneventful recov- in distress. Her vital signs were abdominal pain became more severe, ery. stable, and her temperature was and the next day she became lethar- 37°C. Her sclerae were blue, and gic, her temperature rose to 38°C Si les hernies sont banales dans l'os- her legs were bowed. Her chest had and her respiratory rate was 40/min. teopsathyrose, nous n'avons pas con- an unusual, barrel-like deformity. She was then transferred to our naissance qu'on en ait dej?a rapporte Her breath sounds and bowel sounds hospital. At the time of arrival she interessant le diaphragme, comme were normal. No abdominal re- was in acute respiratory distress and dans le cas present, otu nous avons bound, guarding or rigidity was required intubation and mechanical observe une incar&eration de l'angle present, and there was no evidence ventilation. Her abdomen was dis- splenique du colon. Guerison sans of an inguinal hernia. The results of tended and tender, and bowel sounds encombre apres resection des tissus rectal examination were normal. were absent. Her leukocyte count necroses et colostomie transverse. Her leukocyte count was 8.4 X was 20.0 X 1 09/L. Resuscitation 109/L and her hemoglobin level 146 with lactated Ringer's solution was Osteogenesis imperfecta is a diffuse g/L. Serum electrolyte, amylase and performed and antibiotic therapy connective tissue disorder associated glucose levels were all within normal begun. Chest roentgenography dem- with abnormal tissue collagen.' Al- though inguinal and umbilical hernias are relatively frequent in patients with this disease, previous investigators have been unable to identify any association of osteogen- esis imperfecta with diaphrag- matic hernia.2'3 We describe a woman with osteogenesis imperfecta who presented with a strangulated dia- phragmatic hernia that required ur- gent surgery. Case report A 65-year-old woman was admit- ted to hospital because of abdominal From the Division of General Surgery, To- ronto General Hospital, and the Division of Cardiovascular and Thpracic Surgery, St. Michael's Hospital, University of Toronto *Resident in general surgery tAssociate professor of surgery Reprint requests to: Dr. Zane Cohen, Eaton 9-21 1, Toronto General Hospital, 100 Col- Fig. 1-Barium-containing segment of colon in left side of chest. Note multiple old lege St., Toronto, Ont. M5G I L7 fractures of ribs. - For prescribing information see page 1407 CAN MED ASSOC J, VOL. 131, DECEMBER 1, 1984 1369 Intermediate Prescribing Information arrhythmia can develop. Profound shock can occur in severe overdosage. Treatment: No known specific antidote. Evacuate onstrated a barium-containing seg- *Apresoline tablets gastric content, taking adequate precautions against ment of colon in the chest (Fig. 1). (hydralazine hydrochloride) aspiration and for protection of the airway; if general conditions permit, activated charcoal slurry is instilled. Surgery revealed a defect 4 cm in Antihypertensive Agent These procedures may have to be omitted or carried out diameter in the posterolateral aspect Actions after cardiovascular status has been stabilized, since Hydralazine hydrochloride exerts its hypotensive action they might precipitate cardiac arrhythmias or increase of the left diaphragm. It contained a by reducing vascular resistance through direct relaxation the depth of shock. of vascular smooth muscle. Support of the cardiovascular system is of primary impor- strangulated segment of the splenic Indications tance. Shock should be treated with volume expanders flexure of the colon; perforation had APRESOLINE Oral: Essential hypertension. without resorting to use of vasopressors, if possible. APRESOLINE is used in conjunction with a diurectic If a vasopressor is required, a type that is least likely to occurred at many sites. The and/or other antihypertensive drugs but may be used precipitate or aggravate cardiac arrhythmia should be 15-cm-long necrotic segment was re- as the initial agent in those patients in whom, in the used, and the E.C.G. should be monitored while they judgment of the physician, treatment should be started are being administered. sected and a transverse colostomy with a vasodilator. Digitalization may be necessary. Renal function must APRESOLINE Parenteral: Severe hypertension when be monitored and supported as required. performed. The distal colon was the drug cannot be given orally or when there is an No experience has been reported with extracorporeal oversewn and left in situ. urgent need to lower blood pressure (e.g. toxemia of or peritoneal dialysis. pregnancy or acute glomerulonephritis). It should be used Dosage and Administration The patient's recovery was un- with caution in patients with cerebral vascular accidents. Adjust dosage according to individual blood pressure eventful. Contraindications response. Hypersensitivity to hydralazine, coronary artery disease, Orally: Inftial: 10 mg 4 times daily for the first 2 to 4 mitral valvular rheumatic heart disease, and acute days, 25 mg 4 times daily for the remainder of the first dissecting aneurysm of the aorta. week, 50 mg 4 times daily for the second and subse- Discussion Warnings quent weeks of treatment. Hydralazine may produce in a few patients a clinigal Maintenance: adjust dosage to lowest effective levels. picture simulating systemic lupus erythematosus, inl such Following titration, some patients may be maintained Inguinal and umbilical hernias cases treatment should be discontinued immediately. on a twice daily schedule. are common in patients with os- Long-term treatment with adrenocorticosteroids may Usual maximum daily dose is 200 mg, up to 300 mg be necessary. Complete blood counts, L.E. cell prepara- daily may be required in some patients. In such cases teogenesis imperfecta,2'3 but we tions, and antinuclear antibody titer determinations are a lower dosage of APRESOLINE combined with a indicated before and periodically during prolonged ther- thiazide, reserpine or both, or with a beta-adrenergic- could find no evidence from the apy with hydralazine and if patient develops arthralgia, blocking agent may be considered. When combining literature that this disease has ever fever, chest pain, continued malaise or other unexplained therapy, individual titration is essential to ensure that signs or symptoms. If the results of these tests are the lowest possible therapeutic dose of each drug is been associated with diaphragmatic abnormal, treatment should be discontinued. administered. hernia. The posterolateral location Usage in Pregnancy Parenterally: patients should be hospitalized. Usual Animal studies indicate that high doses of hydralazine dose is 20-40 mg I.M. or by slow I.V. injection or I.V. of the diaphragmatic defect in our are teratogenic. Although there is no positive evidence drip, repeated as necessary. Patients with marked of adverse effects on the human fetus, hydralazine renal damage may require a lower dosage. patient (the site of the embryonic should be used during pregnancy only if the benefit For I.V. drip, the ampoule(s) should be added to 5% pleuroperitoneal canal and Boch- clearly justifies the potential risk to the fetus. sorbitol solution, physiological saline or Ringer solution; Precautions glucose solution is not suitable for this purpose. dalek's hernia in infants) suggests Caution is advised in patients with suspected coronary- Blood pressure levels should be monitored. It may that an anatomic defect may have artery disease, as it may precipitate angina pectoris or begin to fall within a few minutes after injection, with congestive heart failure, and it has been implicated in an average maximal decrease occurring in 10 to 80 allowed the initial development of the production of myocardial infarction. minutes. In cases with a previously existing increased the The "hyperdynamic" circulation caused by APRESOLINE intracranial pressure, lowering the blood pressure may hernia, with weak connective may accentuate specific cardiovascular inadequacies, increase cerebral ischemia. tissue permitting propagation. A sim- e.g. may increase pulmonary artery pressure in patients Most patients can be transferred to oral APRESOLINE with mitral valvular disease. within 24 to 48 hours. ilar combination of factors - an May reduce the pressor responses to epinephrine. Availability anatomic defect plus weak connec- Postural hypotension may result. Tablets of 10 mg: yellow, uncoated, biconvex, scored, Use with caution in patients with cerebral vascular and imprnted FA" on one side and "CIBA" on the other. tive tissue - probably accounts for accidents and in patients with advanced renal damage.
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