Chronic Infection of the Lingula withBronchial Lymphadenopathy

I. MANDELBAUM, M.D., J. S. BATTERSBY, M.D.

From the Department of Surgery and Heart Research Center, Indiana University School of Medicine

EXTRINSIC compression of the right mid-dle lobe bronchus by enlarged lymph nodesassociated with chronic alterations of thepulmonary parenchyma has been termedthe "middle lobe syndrome."4 Recent ex-periences at this institution have confirmedthe observation that a similar pathologicprocess may involve the lingular segmentof the left upper lobe, an observationothers have made in a few instances. It isthe purpose of this paper to present fivepatients who were operated upon for dis-ease of the lingula associated with ex-trinsic compression of the segmentalbronchus, and to review the pertinent lit-erature.

Case ReportsCase 1. The first patient, a 14-year-old girl,

entered the hospital in December of 1951 becauseof chronic cough productive of purulent sputum.She had poliomyelitis in 1950, and requiredtracheostomy then for aspiration of excessivetracheobronchial secretions. Physical examinationwas negative except for crepitant rales in the leftanterior chest. The PPD skin test was negative,and x-rays showed increased bronchovascularmarkings in the left paracardiac area of the lung.Bronchograms revealed bronchiectasis of the lin-gula of the left upper lobe (Fig. 1).

At operation the lingula was completely ate-lectatic. There were large lymph nodes surround-ing and compressing the segmental bronchus.Lingulectomy was performed. The operative andpostoperative courses were uncomplicated. Patho-

logic study of the specimen disclosed severebronchiectasis and organized pneumonia.

Case 2. This 40-year-old woman was admittedto the hospital on May 14, 1955 because of leftchest pain, weakness and shortness of breath oftwo-month duration. She had three episodes offever and left chest pain in the six months pre-ceding her admission with some improvementfollowing antibiotic therapy. Hemoptysis had notoccurred. She smoked one to three cigarettes dailyfor six years. Physical examination revealed anarea of dullness and decreased tactile fermitus inthe left axillary area. On skin test, the firststrength PPD was negative and the second strengthwas strongly positive. Chest x-ray film disclosedsegmental atelectasis of the lingula with em-physema of the left lower lobe (Fig. 2). Atbronchoscopy redness and thickening of the leftupper lobe bronchus was noted. There was asmall amount of purulent material at the orificeof the lingular bronchus. It was impossible to passan aspiration tip into the lingular bronchialorifice. Sputa for acid-fast bacteria and neoplasticcells were negative.

Operation was performed on May 19. Thelingula was partially atelectatic and adherent tothe pericardium. There was an enlarged lymphnode in the acute angle between the lingular seg-mental bronchus and bronchus of the lower lobe.A broncholith in the lingular bronchus was re-moved with the segment. The patient made agood recovery, and was asymptomatic when seentwo years after operation. Pathologic study of thelingula revealed bronchiectasis with focal ate-lectasis and fibrosis.

Case 3. The third patient was a 31-year-oldwoman who entered the hospital on February 25,1957 because of recurrent episodes of pneumoniaeach winter for five years. She had a chroniccough with mucopurulent sputum. There was nohistory of hemoptysis or contact with tuberculosis.She smoked half to a full pack of cigarettes each

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* Submitted for publication November 29,1962.

This work was aided in part by a grant fromthe Indiana Heart Association.

Volume 158Number 6

FIG. 1. (left) Leftlateral bronchogram dem-onstrates bronchiectasisin Case 1. FIG. 2. (right)Left lateral roentgeno-gram of the chest in thesecond patient with ate-lectasis of the lingula.

CHRONIC INFECTION OF THE LINGULA

day for about 12 years. Physical examination was

normal. X-ray films of the chest, including plano-grams, revealed a cystic area in the lingular seg-

ment of the left upper lobe and bronchogramsfailed to demonstrate any filling of the lingula(Fig. 3). Histoplasmin skin test was positive andthe first strength PPD was negative. Bronchoscopydisclosed thick mucopurulent material near theleft upper lobe orifice. Acid-fast studies of thesputum were negative. Operation was performedon March 4. Dense adhesions were divided be-tween the lingula and pericardium and diaphragm.A cystic lesion at the tip of the lingula measuredabout one centimeter in diameter, and decreasedcrepitation was observed throughout the entirelingula. During lingulectomy, several large lymphnodes were found to encompass the segmentalbronchus at its origin, and were felt to impingeupon it. The operative and postoperative courses

FIG. 3. (left) Absence

of complete filling of the

lingula bronchus in Case3. FIG. 4. (right) Lin-gular bronchiectasis inCase 4 with postopera-tive pleural thickening in

the right chest followingright middle lobectomythree years previously.

Iwere smooth. Examination of the surgical speci-men revealed bronchiectasis with evidence ofacute inflammation in several bronchioles. Focalareas of atelectasis were present.

Case 4. A 54-year-old woman was admittedto the hospital on July 10, 1959 because ofchronic cough productive of purulent sputum.She had had many episodes of pneumonitis andchest pain, usually in the left side, for 25 years.In 1956, resection of the right middle lobe wasperformed because of atelectasis and bronchiec-tasis. At that time, a hard, partially calcified lymphnode was found adherent to the right middle lobebronchus and was thought to have caused bron-chial compression. She was asymptomatic for twoyears, and then was re-admitted because of re-currence of cough and purulent sputum. Physicalexamination was unremarkable. Chest x-ray films

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1068 MANDELBAUM Al'

FIG. 5. Left lateral bronchogram of Case 5showing bronchiectasis of the lingula.

revealed accentuation of bronchovascular mark-ings in the left base, and bronchograms disclosedbronchiectasis of the lingula (Fig. 4). A great dealof mucopurulent material was seen in the left mainbronchus during bronchoscopic examination.Smears and culture of the sputum for acid-fastbacteria were negative. At operation, on July 20,the lingula was small and atelectatic. There were

adhesions between this segment and the peri-cardium, and several large lymph nodes sur-

rounded the lingular bronchus. These were re-

moved with the segment. The patient had an un-

eventful postoperative course, and has had no

further pulmonary infection to date. Pathologicstudy of the lingula showed bronchiectasis andorganized pneumonia.

Case 5. The patient was a 29-year-old woman

who was admitted to the hospital on March 20,1961 because of chronic cough and left anteriorchest pain. She had had recurrent episodes ofpneumonitis since six years of age and, for thepast two months a cough productive of about one

cup of greenish-yellow sputum per day. Thesputum was occasionally streaked with blood.There was no history of contact with tuberculosis.She had smoked one pack of cigarettes a day forten years. Vital signs and physical examinationwere normal except for expiratory wheezes in the

3DBATTERSBY Annals of SurgeryDecember 1963

left anterior chest which diminished with cough-ing. Chest x-ray films disclosed strand-like den-sities of infiltration in the lingula of the leftupper lobe, and bronchograms (Fig. 5) revealedmarked bronchiectatic changes in this segmentwith extrinsic deformity of the lingular bronchus.Bronchoscopy showed thick secretions in the leftmain stem bronchus. Smears and cultures for acid-fast bacteria were negative, and a Papanicolaousmear was negative for neoplastic cells.

Antibiotics and expectorants were administeredand postural drainage was instituted. On April 3,a left posterolateral thoracotomy was performed.The lingula was firm with decreased aeration, andadhesions were divided between it and the peri-cardium. During lingulectomy, enlarged firm lymphnodes surrounded, and were adherent to thelingular bronchus at its origin. The postoperativecourse was uneventful, and three months later thepatient was asymptomatic. Pathologic examina-tions of the surgical specimen showed bronchiec-tasis and focal squamous metaplasia of the bron-chial epithelium. There was atelectasis and fibrosisof the pulmonary parenchyma.

DiscussionIn 1937, Brock called attention to chronic

pulmonary infection related to compressionof segments of the bronchial tree by en-larged tuberculous lymph nodes.' Nineyears later, two patients were reported withsimilar pathologic alterations of the rightmiddle lobe associated with nontuberculouslymph node infection.7 In 1948, Graham,Burford and Mayer reported 12 additionalcases with the "middle lobe syndrome."4This descriptive term grouped togethercases of chronic pneumonitis, atelectasisand bronchiectasis of the right middle lobeassociated with compression of the lobarbronchus by enlarged lymph nodes. It wastheir belief that repeated bronchial infec-tion, and lymph node enlargement favoredstasis of bronchial secretions with progres-sive pulmonary pathological change. Sub-sequent reports have confirmed this be-lief.3 5,6 Since the pathologic picture isnot pathognomonic of any specific disease,it is not altogether surprising that a similarclinical picture may occur in the lingulaof the left upper lobe.

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Five female patients are presented withchronic pulmonary infection, atelectasis,and bronchiectasis with peribronchiallymphadenopathy compressing the lingularbronchus. They ranged in age from 14 to54 years. Chronic cough productive ofmucopurulent sputum was the most com-mon presenting symptom, and was notedin four patients. Recurrent pneumonitis hadoccurred in three, shortness of breath, andhemoptysis in one each, and chest pain intwo. These symptoms parallel those re-ported in patients with middle lobe syn-drome.3-6 Physical examination revealed anexpiratory wheeze in one patient, increaseddullness to percussion in another andcrepitant rales in a third patient, while intwo patients no significant physical findingwas discovered. Chest x-ray films disclosedincreased bronchovascular markings in thelingula in three patients, a cystic area inthe lingula in one, and complete atelectasisof the lingula in the other. Bronchogramswere performed in four patients and showedbronchiectasis in three and absence of lin-gular bronchial filling in one. It was omittedin the patient with collapse of the lingula.Bronchoscopy was carried out in four pa-tients to rule out an endobronchial lesion,but was omitted in a 14-year-old girl withevidence of bronchiectasis on broncho-graphic study. In two patients, thickeningand reddening of the bronchial orifice ofthe lingula with retained mucopurulentsecretion was noted. In one, there was ob-struction of the segmental bronchus by abroncholith. In two other patients, bron-choscopy was essentially negative exceptfor mucopurulent secretion in the left mainstem bronchus. Histoplasmin and tuber-culin skin tests revealed a negative firststrength PPD and positive histoplasmin inone patient, a negative PPD in another, anda negative first strength, and positive sec-ond strength PPD in a third patient. Skintests were not performed in two patients.Acid-fast smear and cultures of the sputain all patients were negative.

N OF THE LINGULA 1069

Resection and pathologic study of theresected lingula disclosed partial, or com-plete atelectasis of the lingula, bronchiec-tasis, and enlarged lymph nodes surround-ing the lingular bronchus at its origin.An additional finding in one patient was abroncholith which had eroded into thebronchial lumen. Observations during op-eration confirmed the roentgenologic im-pression that the disease was limited to thelingula. The left lower lobe, which is fre-quently the site of bronchiectasis, was nor-mal. One patient had right middle lobec-tomy three years before lingulectomy be-cause of chronic infection associated withbronchial lymphadenopathy. Lingulectomywas carried out in all five patients withoutcomplication, and with disappearance ofthe presenting symptoms. The period offollow up observation has varied from oneto seven years.

During the past ten years, when five pa-tients were treated here for disease of thelingula, 20 middle lobectomies were per-formed for the middle lobe syndrome.Paulson and Shaw noted chronic lingulardisease in four patients and right middlelobe infection in 32.6 Harper and his as-sociates reported 26 patients with middlelobe syndrome, while nine had left upper,or lower lobe involvement.5 Rarely, seg-ments other than the middle lobe or thelingula may be affected, and Beck de-scribed one patient with left upper lobecollapse related to an enlarged anthracoticlymph node.'

Study of the operative specimens inchronic disease of the lingula associatedwith peribronchial lymphadenopathy hasrevealed the pathologic changes to be thoseof severe nonspecific chronic infection.Widespread atelectasis, fibrosis, and ad-vanced bronchiectasis were found in allspecimens. These findings would seem toemphasize the futility of prolonged medicaltherapy and the needs for surgical extirpa-tion of the diseased segment in these pa-tients.

MANDELBAUM AND BATTERSBY Annals of Surgery1070 December 1963

Summary

Five patients with chronic infection ofthe lingula associated with bronchiallymphadenopathy are reported. The simi-larity between these patients and thosewith the right middle lobe syndrome isstressed. Surgical extirpation is the treat-ment of choice, and was performed in thepresent series with good result.

References1. Beck, H. R.: Atelektase Durch Anthrakotischen

Lymphknoten. Fortschr. Roentgenstrahlen,71:935, 1949.

2. Brock, R. C., R. J. Cann and J. R. Dickinson:Tuberculous Mediastinal Lymphadenitis inChildhood; Secondary Effects on Lungs. Guy'sHosp. Rep., 87:295, 1937.

3. Fretheim, B.: The So-Called Middle LobeSyndrome. Thorax, 1:156, 1952.

4. Graham, E. A., T. H. Burford and J. H.Mayer: Middle Lobe Syndrome. Postgrad.Med., 4:29, 1948.

5. Harper, F. R., W. B. Condon and W. H. Wier-man: Middle Lobe Syndrome. Arch. Surg.,61:696, 1950.

6. Paulson, D. L. and R. R. Shaw: Chronic Ate-lectasis and Pneumonitis of the Middle Lobe.J. Thorac. Surg., 18:747, 1949.

7. Zdansky, E.: Der Mitellappen als PunctumMinoris Resistentiae der Lunge. Wien. Klin.Wchnschr., 58:197, 1946.