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Cervical emphysema secondary to pneumomediastinum as a complication of childbirth.

Abstract

Pneumomediastinum and cervical emphysema usually occur following esophageal or chest trauma. Rarely do they occur as a complication of childbirth, and only approximately 200 such cases have been reported in the literature worldwide. We describe a new case, and we review the clinical picture, pathophysiology, and management of these conditions. In view of the head and neck symptoms of pneumomediastinum and cervical emphysema during labor--which include dyspnea, cough, sore throat, pain on swallowing, and dysphagia--otolaryngologists might be consulted and should therefore be aware of these conditions in order to recognize and treat them.

Introduction

Pneumomediastinum usually occurs following esophageal or chest trauma. It can also occur spontaneously in association with asthma, excessive coughing, or straining. Cervical emphysema occurs when air moves through tissue planes into subcutaneous areas of the face and neck. These conditions are rare as a complication of childbirth, as only approximately 200 cases of pneumomediastinum and cervical emphysema during labor and delivery have been reported worldwide, (1-3) and their incidence is estimated to be 1 in 100,000 deliveries. (4-7) To our knowledge, these conditions during childbirth have not been previously described in the otolaryngologic literature. In this article, we describe the first such case seen in the 75-year history of Seibo International Catholic Hospital in Tokyo, where approximately 2,000 children are born each year.

Case report

A 32-year-old primigravida was admitted at 40 weeks' gestation for induction of labor following amniorrhexis. She had a history of an untreated herniated lumbar disk. She was a nonsmoker, and neither she nor anyone in her immediate family had a history of pulmonary, laryngeal, or cardiac disease. Her antenatal course had been uncomplicated, and vaginal delivery was anticipated.

The duration of the first stage of labor was 11 hours and 20 minutes, the second stage lasted 3 hours and 8 minutes, and the third stage lasted 6 minutes. The patient strained very forcefully during the second stage. She delivered a live girl weighing 3,708 grams. The infant's Apgar scores were 8 at 1 minute and 9 at 5 minutes. An episiotomy was performed.

At 15 hours following delivery, the patient noticed anterior neck swelling and pain on swallowing. She did not complain of dyspnea. On clinical examination, her pulse rate was 100 beats per minute, her blood pressure was 126/75 mm Hg, and her body temperature was 38[degrees]C.

The patient was referred to the Department of Otolaryngology, where she was noted to have a marked soft swelling and palpable crepitations in her entire neck and supraclavicular area. The otolaryngologic examination, including fiberoptic laryngoscopy, detected neither a perforation nor mucosal swelling in her larynx and pharynx.

Neck and chest x-rays were taken. The neck x-rays showed evidence of bilateral emphysema in the subcutaneous area and around the pharyngeal cavity (figure 1). Pneumomediastinum and subcutaneous emphysema of the supraclavicular area were evident on the chest x-ray (figure 2). The lungs were fully expanded, and no pneumothorax was seen. A Gastrografin esophagography did not detect any evidence of esophageal perforation. Computed tomography (CT) identified evidence of deep cervical emphysema and subcutaneous emphysema from the neck through the chest, in addition to a marked accumulation of air in the mediastinum (figure 3). No bulla or bleb was detected. The mother's leukocyte count was 14,200/[mm.sup.3] on the day of delivery and 13,300/[mm.sup.3] on postpartum day 2.

[FIGURES 1-3 OMITTED]

The mother was administered antibiotics and observed closely on the ward; she was also urged not to cough. Her signs and symptoms of pneumomediastinum and emphysema spontaneously resolved over the next few days, and she was discharged home on postpartum day 8. On the day before discharge, her leukocyte count had fallen to 5,700/[mm.sup.3]. Follow-up x-rays 15 days following delivery revealed no signs of emphysema (figure 4).

[FIGURE 4 OMITTED]

Discussion

Pneumomediastinum and cervical emphysema usually occur following esophageal or chest trauma or surgery to the airway, and they can also occur as a result of straining against a closed glottis, such as during strenuous work or defecation. (8) They rarely occur as complications of childbirth. In fact, the case described here was the first that has been recorded in the 75-year history of our hospital, which is one of the largest maternity hospitals in Japan.

The first case report of subcutaneous emphysema during labor and delivery was published by Simmons in 1783. (9) However, the first published reference to this condition might have been made in 1618, when Louise Bourgeois, midwife to the queen of France, wrote, "I saw that she tried to stop crying out and I implored her not to stop for fear that her neck would swell." (9) The condition was subsequently described by Hamman in 1945. (10)

Women at high risk are typically primigravidas who experience a prolonged and difficult labor and deliver larger than usual babies and women whose deliveries are characterized by cephalopelvic disproportion. (1,4,9,11-13) (Ann exception to this trend was noted by Reeder, who mentioned that the mean length of labor and the mean fetal size in such cases were within normal limits. (11))

In our case, the mother's herniated lumbar disk might have forced her to strain in an uncomfortable position during the second stage of labor. The length of her labor (11.3 hr during the first stage and 3.1 hr during the second stage) was within normal limits, but the fetal weight (3,708 g) was slightly heavier than the average for a neonate.

When pneumomediastinum occurs during labor, it usually does so during the second stage; pneumomediastinum has been reported during the first stage and antenatally during episodes of coughing or hyperemesis. (1,14) In cases of spontaneous pneumomediastinum during labor, it is believed that alveolar rupture secondary to a rapid increase in air pressure in the alveoli leads to pulmonary interstitial emphysema. The free air subsequently accumulates and moves centrally along the bronchovascular tissue sheath toward the mediastinum, and it may then dissect through the facial planes into more subcutaneous tissues. (15)

Patients report a variety of symptoms, including a change of voice, (16) dyspnea, cough, sore throat, pain on swallowing, dysphagia, hemoptysis, palpitations, and chest pain. Leukocytosis (17) and fever without evidence of infection have also been noted with some frequency. Symptoms are often not noticed until after delivery. (5,12) Four maternal deaths and 13 stillbirths have been reported, all of which occun'ed before 1925. (11)

The diagnosis of cervical emphysema is not difficult. The crepitation palpable in the neck is practically pathognomonic, and the appearance of cervical emphysema during labor is the hallmark of pneumomediastinum. (18) The definitive diagnosis is made radiographically. (19)

Spontaneous pneumomediastinum and cervical emphysema are usually self-limiting. Therefore, observation and symptomatic treatment are all that is required in most cases. Positive-pressure anesthesia should be avoided, (2,10,20) and further straining should be prevented. Recovery generally takes 3 to 14 days. (21) Only a few patients have required a mediastinotomy. (7,22)

In view of the head and neck symptoms associated with pneumomediastinum and cervical emphysema during labor, otolaryngologists may be consulted. We should therefore be aware of these conditions in order to recognize and treat them and, by doing so, prevent severe complications.

References

(1.) Karson EM, Saltzman D. Davis MR. Pneumomediastinum in pregnancy: Two ease reports and a review of the literature, pathophysiology, and management. Obstet Gynecol 1984;64(Suppl): 39S-43S.

(2.) Jayran-Nejad Y, Subcutaneous emphysema in labour, Anaesthesia 1993;48:139-40.

(3.) Gemer O. Popescu M, Lebowits O, Segal S. Pneumomediastinum in labor. Arch Gynecol Obstet 1994;255:47 9.

(4.) Kosmak GW. Subcutaneous emphysema following labor. Surg Gynecol Obstet 1925:40:434.

(5.) Spellacy WN, Prem KA. Subcutaneous emphysema and pregnancy: Report of 3 cases. Obstet Gynecol 1963;22:521-3.

(6.) Crean PA, Stronge JM, FitzGerald MX. Spontaneous pneumomediastinum in pregnancy. Case report. Br J Obstet Gynaecol 1981;88:952-4.

(7.) Kobak AJ, Abrams RH. Pregnancy complicated by massive subcutaneous emphysema of mediastinal origin (Hamman's syndrome). Am J Obstet Gynecol 1949;57:789 92.

(8.) Lain CA. Mediastinal and subcutaneous emphysema during labor. Report of a ease. Obstet Gyneeol 1967;29:378-81.

(9.) Gordon CA. Respiratory emphysema in labor. Am J Obstet Gynecol 1927;14:633-46.

(10.) Hamman L. Mediastinal emphysema. JAMA 1945;128:1-6.

(11.) Reeder SR. Subcutaneous emphysema, pneumomediastinum, and pneumothorax in labor and delivery. Am J Obstet Gynecol 1986;154:487-9.

(12.) Knox GS. Spontaneous subcutaneous emphysema during labor. Am J Roentgenol 1963;89:1087-90.

(13.) Furst NJ, Lawrence LR. A case of bilateral pnenmothorax, associated with pneumomediastinum, atelectasis, pulmonary edema and subcutaneous emphysema, occurring during labor: Mediastinal air block. Am J Roentgenol 1949;62:798-806.

(14.) Gorbach JS, Counselman FL, Mendelson MI 1. Spontaneous pneumomediastinum secondary to hyperemesis gravidarum. J Emerg Med 1997;15:639-43.

(15.) Macklin MT. Macklin CC. Malignant interstitial emphysema of the lungs and mediastinum as an important occult complication in many respiratory diseases and other conditions: An interpretation of the clinical literature in light of laboratory experiment. Medicine 1944;23:281-358.

(16.) Sleeman D. Turner R. Spontaneous pneumomediastinum with alteration in voice. J Laryngul Otol 1989;103:1222-3.

(17.) Singh ON, Didolkar S. Maternal-fetal pneumomediastinum complicating labor: Report of a case and review of the literature. J Reprod Med 1980;25:329-32.

(18.) Dudley DK, Patten DE. Intrapartum pneumomediastinum associated with subcutaneous emphysema. CMAJ 1988;139:641-2.

(19.) Lillard RL, Allen RP. The extrapleural air sign in pneumomediastinum. Radiology 1965;85:1093-8.

(20.) Aisner M, Franco JE. Mediastinal emphysema. N Engl J Med 1949;241:818 25.

(21.) Brandfass RT, Martinez DM. Mediastinal and subcutaneous emphysema in labor. South Med J 1976;69:1554-5.

(22.) Hovick JH, West OT. Mediastinal and subcutaneous emphysema in early pregnancy. Obstet Gynecol 1954;4:606-10.

From the Department of Otolaryngology (Dr. Nakagawa) and the Department of Obstetrics and Gynecology (Dr. Yamauchi). Seibo International Catholic Hospital, Tokyo: the Tokyo Voice Center, International University of Health and Welfare (Dr. Kusuyama and Dr. Fukuda); and the Department of Otolaryngology, Keio University School of Medicine, Tokyo (Dr. Ogawa).

Reprint requests: Hideki Nakagawa, MD, Department of Otolaryngology, Seibo International Catholic Hospital, 2-5-1 Nakaochiai, Shinjuku-ku, Tokyo 161-8521, Japan. Phone: 81-3-3951-1111; fax: 81-3-5982-3077: e-mail: hnakagawa@seibokai.or.jp
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Title Annotation:Original Article
Author:Ogawa, Kaoru
Publication:Ear, Nose and Throat Journal
Geographic Code:9JAPA
Date:Dec 1, 2003
Words:1705
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