4. Dirschmid K, Kiesler J. Die morpholige der leber bei der anthrakosilikose. Leber Magen Darm 1980;10:115-8. 5. Kada I, Szende B, Csikos A, Szendroi M. Extrapulmonary localization of carbon and silica particles in anthracosilicosis. Moo Lab 1984;5:404-6. 6. Pounder DJ. Malarial pigment and hepatic anthracosis. Am J Surg PathoI1983;5:501-2. 7. Van Gossum A, Pirauzx M, Fievet M, Neve P. Hepatic cholestasis mediated by subdiaphragmatic anthracosilicosis. Acta Clin Belg 1981;36:227-32.
8. Morgan WK, Baum GL. Environmental lung disease. In: Wolinskey E, ed. Text book of pulmonary disease. 3rd ed. Boston: Little, Brown and Co., 1983:754-68. 9. Kissler W, Morgenroth K, Scherbeck W. Electron microscopic investigations on dust penetration into the pulmonary interstititum in experimental pneumoconioses. Respiration 1982;43: 114-26. 10. Sharp JR, Insalaco SJ, Johnson LF. Melanosis ofthe duodenum associated with a gastric ulcer and folic acid deficiency. Gastroenterology 1980;78:366-9.
Esophageal lichen planus
performed with a four lumen pneumohydraulically perfused catheter system. The lower esophageal sphincter was normal with a pressure of 20 mm Hg and relaxed normally on swallowing. Esophageal peristaltic contractions stimulated by dry and wet swallows were weak with pressures ranging from 30 to 50 mm Hg. A Bernstein test was immediately and strongly positive, reproducing her symptoms of heartburn. The patient was dilated to 16 mm and treated with cimetidine. The heartburn resolved, but 6 months later the dysphagia returned. Repeat endoscopy revealed that the esophageal mucosa was intact and had a white lacy appearance (Fig. 2) which stripped away in whitish sheets as the endoscope was passed over it (Fig. 3). The underlying epithelium was very friable. Repeat biopsies again showed granulation tissue. The patient was left on cimetidine and taught to dilate her own strictures. She did well until 1989 when she noticed that there was an exudate on her bougie. She stopped dilating her strictures and the dysphagia returned, necessitating endoscopic dilation in January 1990. The endoscopic appearances were the same as before. A barium swallow revealed a stricture and small diverticulum in the proximal esophagus. The body of the esophagus was poorly distensible with minimal peristalsis.
Robert Y. Yoon, MSc, MD, Stephen N. Sullivan, MD, FRCPC, FRCP (Lond)
Lichen planus is a common idiopathic inflammatory disorder of the skin and mucous membranes. Mucosal lesions frequently involve the oral cavity and may occur alone or in association with skin lesions. Oral lichen planus may be asymptomatic and appear as white patches or plaque-like lesions with reticular lace-like patterns and lines on an erythematous base. Painful erosive lesions especially of the buccal mucosa, lips, tongue, and gingiva can develop. Skin lesions, when present, are most frequently pruritic, violaceous papules. The clinical diagnosis, course, and histopathological characteristics of lichen planus have been described in detaiP-S Symptomatic esophageal involvement is rare with only 11 cases reported.6-16
CASE REPORT
A 35-year-old woman presented in 1975 with a half-year history of "sore red gums." A clinical diagnosis of lichen planus was made. This diagnosis was later confirmed pathologically by biopsy of vulvar lesions which developed in 1977. Treatment with topical and systemic steroids, and even a course of plasmapheresis did little for her symptoms. In 1986, she was seen because of the development of heartburn, odynophagia, and dysphagia for solids and liquids. A barium swallow revealed diffuse narrowing of the esophagus, particularly in the upper third. At endoscopy a tight stricture was seen just beneath the cricopharyngeus muscle through which a pediatric endoscope was passed with difficulty. The mucosa of the esophagus was erythematous with a filmy tissue paper-like membrane which stripped away with minimal contact (Fig. 1). The epithelium underneath bled freely. A further stricture was found in the distal esophagus. The esophageal epithelium was biopsied and the strictures were dilated. The biopsies were reported as showing "inflamed florid granulation tissue." An esophageal manometry was From the Departments of Gastroenterology and Radiology, Victoria Hospital, London, Canada. Reprint requests: Dr. R. Y. Yoon, Department of Radiology, Victoria Hospital, 375 South Street, London, Ontario, Canada, N6A 4G5. VOLUME 36, NO.6, 1990
DISCUSSION
Contrary to previous belief, it is now evident that lichen planus frequently involves the epithelium of the esophagus usually as subtle papular lesions. l l It is, however, a very rare cause of esophageal symptoms, and there have been only 11 cases of symptomatic esophageal lichen planus described in the French and English literature.6-16 Although lichen planus affects the sexes almost equally, all the reported cases of esophageal lichen planus have been in women. Our patient is the youngest reported at 46 years old, with previous cases ranging in age from 49 to 76 years. In all cases the presenting complaint was dysphagia, although the clinical course appeared to follow two general patterns. Approximately half the patients had a history of lichen planus or oral, genital, or cutaneous lesions when they developed dysphagia. The remaining patients sought medical attention from dysphagia, but investigation frequently led to the erroneous provisional diagnosis of peptic esophagitis. In this latter 617
group, each patient subsequently developed oral lesions typical of lichen planus. Several authors described exacerbation of oral lesions following dilation of an esophageal stenosis, a Koebner-like phenomenon. However, this observation was not universal and not all patients who subsequently developed oral lichen planus had esophageal dilation. 9 • 14 Radiologic studies when performed showed proximal esophageal stenoses ranging from 1.5 to 8 cm in length. In our patient, a tight proximal stenosis was noted and the entire esophagus was poorly distensible on barium swallow. Endoscopic descriptions of the diseased areas note erosions or white plaque-like lesions and several authors report friable hemorrhagic mucosa. Our patient showed an impressive filmy, membranous layer which easily stripped away in sheets with minimal contact. The underlying mucosa was extremely friable and bled easily. A previous report describes a similar degree of mucosal desquamation. 13 Endoscopic biopsies may not confirm the diagnosis of lichen planus, but the histologic appearances are not those of reflux or fungal esophagitis. 13-15 The diagnosis of esophageal lichen planus should be considered in any middle aged or elderly woman presenting with a proximal esophageal stenosis, especially if there is a history of past or present oral or cutaneous lesions suggestive of lichen planus. The endoscopic finding of a thin white desquamating membrane and underlying friable mucosa should confirm the diagnosis, even if the biopsies are inconclusive. ACKNOWLEDGMENT
The authors are grateful for the assistance of Mrs. Marie Corke.
REFERENCES 1. Altman J, Perry HO. The variations and course oflichen planus. Arch Dermatol 1961;84:179-91. 2. Tyldesley WR. Oral lichen planus. Br J Oral Surg 1974;11:187206.
3. McClatchey KD, Silverman S, Hansen LS. Studies on oral lichen planus. III. Clinical and histologic correlations in 213 patients. J Oral Surg 1975;39:122-9. 4. Scully C, EI-Kom M: Lichen planus: review and update on pathogenesis. J Oral Surg 1985;14:431-58. 5. Fellner MJ. Lichen planus. Int J DermatoI1980;19:71-5. 6. Sayag J, Moges A, Treffot MJ, Cherif HA, Weiler M. Lichen cutane muqueux erosif et poussees iteratives d'anemie megaloblastique para-biermerienne et de Defluvium capillorum. Medit Med 1976;113:81-6. 7. AI-Shihabi BMS, Jackson JMcG. Dysphagia due to pharyngeal and oesophageal lichen planus. J Laryngol Otol 1982;96:56771. 8. Lefer LG. Lichen planus ofthe esophagus. Am J Dermatopathol 1982;4:267-9.
Figure 2. Intact esophageal epithelium prior to passage of Figure 1. Initial endoscopic appearance of esophageal lichen
endoscope but after insertion of a guide wire.
planus, with filmy white membrane and underlying friable epithelium.
Figure 3. "Stripped-away· esophageal epithelium after pas-
618
sage of endoscope but prior to dilation. GASTROINTESTINAL ENDOSCOPY
9. Guedon C, Kuffer R, Thomine E, Lerebours E, Colin R. Lichen plan stenosant de l'oesophage. Gastroenterol Clin Bioi 1982;6:1049-50. 10. Lavignolle A, Renaut JJ, Le Bodie L. Lichen plan erosif stenosant de l'oesophage. Gastroenterol Clin Bioi 1983;7:829-830. 11. Ottignon Y, Carayon P, Deschamps JP, Hirsch JP, Caille JP. Pageaut G. Lichen plan stenosant de l'oesophage: Ie reflux gastro-oesophagien ne parait pas en cause! Gastroenterol Clin Bioi 1983;7:830-1. 12. Quevrin F, Simonis-Blumenfrucht A, Andre P, Saladhin A. Lichen plan plurimuqueux et atteinte oesophagienne. Sem Hop Paris 1986;62:1255-7.
13. Sheehan-Dare RA, Cotterill JA, Simmons AV. Oesophageal lichen planus. Br J Dermatol 1986;115:729-30. 14. Bousser AM, Nilias G, Mosser C, Ramee MP, Delambre C, Gosselin M, Chevrant-Breton J. Lichen plan et stenose oesophagienne. A propos d'un cas. Ann Dermatol Venereol 1986;113:938-9. 15. Jobard-Drobacheff C, Blanc D, Quencez E, Zultak M, Paris B, Ottignon Y, Agache P. Lichen planus of the oesophagus. Clin Exp DermatoI1988;13:38-41. 16. Dickens CM, Heseltine D, Walton S, Bennett JR. The oesophagus in lichen planus: an endoscopic study. Br Med J 1990;300:84.
Inverted upper gastrointestinal endoscope in the esophagus: technique of endoscopic straightening with an auxiliary instrument
revealed complete inversion of the endoscope in the lower esophagus (Fig. 1). The patient was then sedated (10 mg of diazepam, 50 mg of Ketamin intravenously) and liberation of the endoscope by forward, backward, and circular maneuvers was attempted under fluoroscopic control without success. Even under subsequent general anesthesia, the inverted fiberscope could not be advanced more than 5 cm and did not pass the cardia. A second endoscope (Olympus PQ 20) was inserted under vision to about 1 cm from the inverted tip of the first. A flexible biopsy forceps was inserted through the biopsy channel of the auxiliary endoscope into the facing orifice of the impacted instrument and advanced for approximately 10 cm into the inverted segment (Fig. 2). With a short pull of the inverted endoscope and a simultaneous push of the second both instruments were brought in a parallel and orthograde position (Fig. 3). Due to the firm attachment of the endoscopes to each other by the flexible wire, reinversion was possible with a minimum of force and movement. Thus, a slip or lateral escape of the inverted tip during the pull back maneuver was prevented, and a tear of the surrounding tissue was avoided. With both instruments in a parallel position, first the biopsy forceps and then the now straightened endoscope were quickly withdrawn. Subsequent inspection of the esophagus revealed a narrow stricture of the gastro-esophageal junction that could not be passed. There was no damage to the esophagus except for mild erosions of the superficial epithelium in the area where the inverted endoscope was impacted. The patient recovered quickly from anesthesia and a Gastrografin@ swallow 3 days after the procedure revealed no leakage of the contrast medium from the esophagus.
H.-J. Thon, MD Markus M. Lerch, MD Siegfried Matern, MD
Since the introduction of fibergastroscopy with flexible instruments, the rate of complications has been reduced to 0.081%.1.2 Perforations of the esophagus and stomach in particular occur very rarely compared with earlier reports based on experience with rigid or semiflexible instruments. 3 • 4 One of the possible hazards of modern fiberoptic endoscopes is the accidental inversion and impaction of the instrument in the esophagus. This can occur during retrograde inspection of the fundus and cardia when the instrument is pulled back into the esophagus,5.6 or can happen during blind insertion of highly flexible endoscopes, when the instrument inverts in the oropharynx and is then advanced. 7 CASE REPORT
A 35-year-old man was admitted to commence chemotherapy for a malignant islet cell tumor of the pancreas (pancreatic polypeptide and gastrin producing) diagnosed by histology and immunocytochemistry 3 months earlier. A large mediastinal metastasis was identified on chest x-ray. An initial uncomplicated upper gastrointestinal endoscopy was done before therapy for unremitting upper abdominal pain, and revealed tumor penetration into the descending duodenum without active bleeding. While on chemotherapy, despite antacid treatment, the patient developed progressive dysphagia, and a second endoscopy was performed 4 weeks after the first. When the endoscope was inserted blindly the patient briefly gagged, and the instrument (Olympus PQ 20) could not be advanced beyond 25 cm. The endoscopic view From the Medizinische Klinik III, Klinikum der Rheinisch- Westfaelischen Technischen Hochschule, D-5100 Aachen, West Germany. Reprint requests: Markus M. Lerch, MD, Department of Surgery, Beth Israel Hospital, 330 Brookline Avenue, Boston, Massachusetts 02215. VOLUME 36, NO.6, 1990
DISCUSSION
Fiber-gastroscopy can be considered an invasive diagnostic technique with a very low rate of complications. 2 Perforations and lacerations of intestinal structures are extremely rares compared with the past when rigid or semiflexible instruments were used. The most frequent complications observed today are of cardiac origin 9 and can be influenced by the selection of appropriate sedatives and fiberscopes. Highly flexible and low diameter endoscopes (e.g., Olympus GIFP3 or PQ-20) have the potential risk of folding during 619