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Esophageal tuberculosis: a rare but not to be forgotten entity
Letters to the Editor
temically toxic. The neurologic examination was remarkable for diminished sensation over the left lower extremity distal to the knee. Although the strength of the left hip and knee flexors was normal, movement of the ankle and toes was absent. Additionally, both the left knee and ankle reflexes were hypoactive compared with the right, and the plantar response was absent. Examination of the right lower extremity was unremarkable. The patient’s genitalia were erythematous and grossly edematous, with patchy necrosis. Multiple areas of pressure necrosis were present on both buttocks. The relevant laboratory investigations showed a leukocytosis of 19.2 ⫻ 109/L and an elevated creatinine phosphokinase (9,365 U/L) and serum creatinine (235 umol/L). The remaining investigations were unremarkable. Despite adequate fluid resuscitation for rhabdomyolysis, the patient became anuric, requiring hemodialysis for acute renal failure. He also developed gangrene of the perineum, requiring debridement, bilateral orchiectomy, and partial perinectomy. He subsequently died in hospital from uncontrollable sepsis. This case illustrates the hazards of prolonged immobilization, specifically sitting. The following sequence of events may account for his clinical deterioration. The left sciatic nerve palsy may have resulted from either direct compression of the nerve coursing through the sciatic notch or a gluteal compartment syndrome, or both. The nerve compression may have been worsened by prolonged hip flexion, from sitting on the toilet seat, as this tightens the sciatic nerve in its notch (1). The gluteal compartment syndrome likely developed from compression of the gluteal vessels and nerves in the unyielding fascial compartment, leading to ischemic necrosis (2). The prolonged immobilization on the toilet seat also led to a perineal compression syndrome, developing
as a consequence of ischemic necrosis from compression of the perineal and internal pudendal vessels. The ensuing rhabdomyolysis combined with sepsis resulted in acute renal failure (2). Sciatic neuropathy has also been previously described after hip replacements (3), prolonged episodes of sitting on hard surfaces (4), and in two other cases involving seat entrapment (5). This case of the “toilet seat syndrome” was characterized by acute renal failure, perineal gangrene, and sciatic neuropathy as a consequence of prolonged immobilization while sitting on a toilet. Rajive Jassal, BSc(Med) Davinder S. Jassal, MD John M. Embil, MD Faculty of Medicine and Sections of General Internal Medicine and Infectious Diseases Department of Medicine University of Manitoba Winnipeg, Manitoba, Canada 1. Yuen EC, So Y. Sciatic neuropathy. Neuro Clin. 1999;17:617– 631. 2. Hynes JE, Jackson A. Atraumatic gluteal compartment syndrome. Postgrad Med J. 1994;70:210 –212. 3. Stewart JD, Angus E, Gedron D. Sciatic neuropathies. BMJ. 1983;287:1108 –1109. 4. Tyrell PJ, Feher MD, Rossor MN. Sciatic nerve damage due to toilet seat entrapment: another Saturday night palsy. Neurol Neurosurg Psych. 1989;52:113–115. 5. Deverell WF, Ferguson JH. An unusual case of sciatic nerve paralysis. JAMA. 1968;205: 699 –700.
ESOPHAGEAL TUBERCULOSIS: A RARE BUT NOT TO BE FORGOTTEN ENTITY To the Editor: Tuberculosis of the esophagus is an unusual presentation of this disease, having been estimated to occur in 0.15% of people who die with tuberculosis (1). Esophageal disease is generally secondary to mycobacterial disApril 1, 2001
ease elsewhere, although a few cases of possible primary tuberculous esophagitis have been described (2,3). We describe a case of probable reactivation of tuberculosis in the mediastinal lymph nodes and extension into the esophagus. A 48-year-old Laotian man who had immigrated to the United States 18 years earlier presented with odynophagia, which he had had for 1 week and described as a sharp midsternal pain when he swallowed liquids and solids. He was otherwise in good health and reported no other symptoms. He was a previous heavy smoker and consumed at least 6 beers daily. His mother had died of tuberculosis 1 year before he immigrated to the United States. The physical examination was unremarkable; no lymphadenopathy was found and the patient’s lungs were clear. A bariumswallow study revealed a small mucosal lesion in his upper esophagus. Endoscopy characterized the lesion as having heaped up borders and little associated inflammation (Figure 1). Biopsies revealed noncaseating granulomas. No acid-fast organisms were observed on stains, and cultures were negative. A purified protein derivative skin test was positive. Serologic testing for human immunodeficiency virus (HIV) was negative. A roentgenogram of the chest showed no abnormalities. However, a computed tomogram of the thorax showed mediastinal lymphadenopathy adjacent to and abutting the esophagus (Figure 2). Mediastinoscopy was performed, and biopsies of the mediastinal nodes revealed multiple granulomas with rare central necrosis and numerous giant cells. Scant acid-fast bacilli were observed on auramine rhodamine stain and cultures grew Mycobacterium tuberculosis. The patient underwent therapy for active tuberculosis with isoniazid, pyrazinamide, rifampin, ethambutol, and pyridoxine. After 2 months of therapy, he is doing well, with no symptoms of odynophagia.
THE AMERICAN JOURNAL OF MEDICINE威
Volume 110 415
Letters to the Editor
Figure 1. Upper endoscopy showed an ulcerative lesion (arrow) in the esophagus at 30 cm.
Figure 2. Computed tomogram of the thorax revealed mediastinal lymphadenopathy adjacent to and abutting the esophagus.
Secondary esophageal tuberculosis is a very rare but well-documented entity. In 1913 Lockard (1) described involvement of the esophagus in 25 of 16,489 patients who died of tuberculosis. The most common mechanism for secondary involvement of the esophagus is reactivation in mediastinal lymph nodes and erosion into the esophagus, as occurred in our patient, but it can also occur from local extension from pharyngeal or laryngeal disease, broncho-esophageal fistulas, 416
April 1, 2001
infected aortic aneurysms or infected bone, retrograde flow of lymphatic drainage from infected paratracheal, subcarinal or peribronchial lymph nodes, and hematogenous spread (1,3–5). Esophageal tuberculosis most commonly presents with odynophagia or dysphagia and constitutional symptoms, although patients can be asymptomatic (1– 4,6,7). Lesions found by endoscopy typically appear as mucosal irregularities or ulcers in
THE AMERICAN JOURNAL OF MEDICINE威 Volume 110
the middle third of the esophagus and can resemble malignancy (5–10). Strictures and fistulas are common (1,9). Even patients with advanced disease who have tuberculous esophagitis do well on antibiotic therapy and may experience complete resolution of disease (2– 4,6 – 8). Particularly in persons with risk factors for tuberculosis, esophageal involvement with tuberculosis should be considered in the differential diagnosis for any unexplained esophageal ulcer or mucosal abnormality seen on x-ray film or endoscopy. The outcome is generally good and antibiotic therapy alone is generally successful. Sigurdur T. Sigurdarson, MD F. Jeffrey Field, MD Larry S. Schlesinger, MD Departments of Internal Medicine and Microbiology University of Iowa Hospitals and Clinics, Iowa City, Iowa 1. Lockard LB. Esophageal tuberculosis: a critical review. Laryngoscope. 1913;23:561– 584. 2. Upadhyay AP, Bhatia RS, Anbarasu A, et al. Esophageal tuberculosis with intramural pseudodiverticulosis. J Clin Gastroenterol. 1995;22:38 – 40. 3. Rosario MT, Raso CL, Comer GM. Esophageal tuberculosis. Dig Dis Sci. 1989;34: 1281–1284. 4. Gordon AH, Marshall JB. Esophageal tuberculosis: definitive diagnosis by endoscopy. Am J Gastroenterol. 1990;85:174 – 177. 5. McNamara M, Williams CE, Brown TS, et al. Tuberculosis affecting the oesophagus. Clin Radiol. 1987;38:419 – 422. 6. Laajam MA. Primary tuberculosis of the esophagus: pseudotumoral presentation. Am J Gastroenterol. 1984;79:839 – 841. 7. Damtew B, Frengley D, Wolinsky E, et al. Esophageal tuberculosis. Mimicry of gastrointestinal malignancy. Rev Infect Dis. 1987;9:140 –146. 8. Seivewright N, Feehally J, Wicks ACB. Primary tuberculosis of the esophagus. Am J Gastroenterol. 1984;79:842– 843. 9. Annamalai AL, Shreekumar S. Tuberculosis of the esophagus. Am J Gastroenterol. 1972;57:166 –168. 10. Brullet E, Font B, Rey M, et al. Esophageal tuberculosis. Early diagnosis by endoscopy. Endoscopy. 1993;25:485.
temically toxic. The neurologic examination was remarkable for diminished sensation over the left lower extremity distal to the knee. Although the strength of the left hip and knee flexors was normal, movement of the ankle and toes was absent. Additionally, both the left knee and ankle reflexes were hypoactive compared with the right, and the plantar response was absent. Examination of the right lower extremity was unremarkable. The patient’s genitalia were erythematous and grossly edematous, with patchy necrosis. Multiple areas of pressure necrosis were present on both buttocks. The relevant laboratory investigations showed a leukocytosis of 19.2 ⫻ 109/L and an elevated creatinine phosphokinase (9,365 U/L) and serum creatinine (235 umol/L). The remaining investigations were unremarkable. Despite adequate fluid resuscitation for rhabdomyolysis, the patient became anuric, requiring hemodialysis for acute renal failure. He also developed gangrene of the perineum, requiring debridement, bilateral orchiectomy, and partial perinectomy. He subsequently died in hospital from uncontrollable sepsis. This case illustrates the hazards of prolonged immobilization, specifically sitting. The following sequence of events may account for his clinical deterioration. The left sciatic nerve palsy may have resulted from either direct compression of the nerve coursing through the sciatic notch or a gluteal compartment syndrome, or both. The nerve compression may have been worsened by prolonged hip flexion, from sitting on the toilet seat, as this tightens the sciatic nerve in its notch (1). The gluteal compartment syndrome likely developed from compression of the gluteal vessels and nerves in the unyielding fascial compartment, leading to ischemic necrosis (2). The prolonged immobilization on the toilet seat also led to a perineal compression syndrome, developing
as a consequence of ischemic necrosis from compression of the perineal and internal pudendal vessels. The ensuing rhabdomyolysis combined with sepsis resulted in acute renal failure (2). Sciatic neuropathy has also been previously described after hip replacements (3), prolonged episodes of sitting on hard surfaces (4), and in two other cases involving seat entrapment (5). This case of the “toilet seat syndrome” was characterized by acute renal failure, perineal gangrene, and sciatic neuropathy as a consequence of prolonged immobilization while sitting on a toilet. Rajive Jassal, BSc(Med) Davinder S. Jassal, MD John M. Embil, MD Faculty of Medicine and Sections of General Internal Medicine and Infectious Diseases Department of Medicine University of Manitoba Winnipeg, Manitoba, Canada 1. Yuen EC, So Y. Sciatic neuropathy. Neuro Clin. 1999;17:617– 631. 2. Hynes JE, Jackson A. Atraumatic gluteal compartment syndrome. Postgrad Med J. 1994;70:210 –212. 3. Stewart JD, Angus E, Gedron D. Sciatic neuropathies. BMJ. 1983;287:1108 –1109. 4. Tyrell PJ, Feher MD, Rossor MN. Sciatic nerve damage due to toilet seat entrapment: another Saturday night palsy. Neurol Neurosurg Psych. 1989;52:113–115. 5. Deverell WF, Ferguson JH. An unusual case of sciatic nerve paralysis. JAMA. 1968;205: 699 –700.
ESOPHAGEAL TUBERCULOSIS: A RARE BUT NOT TO BE FORGOTTEN ENTITY To the Editor: Tuberculosis of the esophagus is an unusual presentation of this disease, having been estimated to occur in 0.15% of people who die with tuberculosis (1). Esophageal disease is generally secondary to mycobacterial disApril 1, 2001
ease elsewhere, although a few cases of possible primary tuberculous esophagitis have been described (2,3). We describe a case of probable reactivation of tuberculosis in the mediastinal lymph nodes and extension into the esophagus. A 48-year-old Laotian man who had immigrated to the United States 18 years earlier presented with odynophagia, which he had had for 1 week and described as a sharp midsternal pain when he swallowed liquids and solids. He was otherwise in good health and reported no other symptoms. He was a previous heavy smoker and consumed at least 6 beers daily. His mother had died of tuberculosis 1 year before he immigrated to the United States. The physical examination was unremarkable; no lymphadenopathy was found and the patient’s lungs were clear. A bariumswallow study revealed a small mucosal lesion in his upper esophagus. Endoscopy characterized the lesion as having heaped up borders and little associated inflammation (Figure 1). Biopsies revealed noncaseating granulomas. No acid-fast organisms were observed on stains, and cultures were negative. A purified protein derivative skin test was positive. Serologic testing for human immunodeficiency virus (HIV) was negative. A roentgenogram of the chest showed no abnormalities. However, a computed tomogram of the thorax showed mediastinal lymphadenopathy adjacent to and abutting the esophagus (Figure 2). Mediastinoscopy was performed, and biopsies of the mediastinal nodes revealed multiple granulomas with rare central necrosis and numerous giant cells. Scant acid-fast bacilli were observed on auramine rhodamine stain and cultures grew Mycobacterium tuberculosis. The patient underwent therapy for active tuberculosis with isoniazid, pyrazinamide, rifampin, ethambutol, and pyridoxine. After 2 months of therapy, he is doing well, with no symptoms of odynophagia.
THE AMERICAN JOURNAL OF MEDICINE威
Volume 110 415
Letters to the Editor
Figure 1. Upper endoscopy showed an ulcerative lesion (arrow) in the esophagus at 30 cm.
Figure 2. Computed tomogram of the thorax revealed mediastinal lymphadenopathy adjacent to and abutting the esophagus.
Secondary esophageal tuberculosis is a very rare but well-documented entity. In 1913 Lockard (1) described involvement of the esophagus in 25 of 16,489 patients who died of tuberculosis. The most common mechanism for secondary involvement of the esophagus is reactivation in mediastinal lymph nodes and erosion into the esophagus, as occurred in our patient, but it can also occur from local extension from pharyngeal or laryngeal disease, broncho-esophageal fistulas, 416
April 1, 2001
infected aortic aneurysms or infected bone, retrograde flow of lymphatic drainage from infected paratracheal, subcarinal or peribronchial lymph nodes, and hematogenous spread (1,3–5). Esophageal tuberculosis most commonly presents with odynophagia or dysphagia and constitutional symptoms, although patients can be asymptomatic (1– 4,6,7). Lesions found by endoscopy typically appear as mucosal irregularities or ulcers in
THE AMERICAN JOURNAL OF MEDICINE威 Volume 110
the middle third of the esophagus and can resemble malignancy (5–10). Strictures and fistulas are common (1,9). Even patients with advanced disease who have tuberculous esophagitis do well on antibiotic therapy and may experience complete resolution of disease (2– 4,6 – 8). Particularly in persons with risk factors for tuberculosis, esophageal involvement with tuberculosis should be considered in the differential diagnosis for any unexplained esophageal ulcer or mucosal abnormality seen on x-ray film or endoscopy. The outcome is generally good and antibiotic therapy alone is generally successful. Sigurdur T. Sigurdarson, MD F. Jeffrey Field, MD Larry S. Schlesinger, MD Departments of Internal Medicine and Microbiology University of Iowa Hospitals and Clinics, Iowa City, Iowa 1. Lockard LB. Esophageal tuberculosis: a critical review. Laryngoscope. 1913;23:561– 584. 2. Upadhyay AP, Bhatia RS, Anbarasu A, et al. Esophageal tuberculosis with intramural pseudodiverticulosis. J Clin Gastroenterol. 1995;22:38 – 40. 3. Rosario MT, Raso CL, Comer GM. Esophageal tuberculosis. Dig Dis Sci. 1989;34: 1281–1284. 4. Gordon AH, Marshall JB. Esophageal tuberculosis: definitive diagnosis by endoscopy. Am J Gastroenterol. 1990;85:174 – 177. 5. McNamara M, Williams CE, Brown TS, et al. Tuberculosis affecting the oesophagus. Clin Radiol. 1987;38:419 – 422. 6. Laajam MA. Primary tuberculosis of the esophagus: pseudotumoral presentation. Am J Gastroenterol. 1984;79:839 – 841. 7. Damtew B, Frengley D, Wolinsky E, et al. Esophageal tuberculosis. Mimicry of gastrointestinal malignancy. Rev Infect Dis. 1987;9:140 –146. 8. Seivewright N, Feehally J, Wicks ACB. Primary tuberculosis of the esophagus. Am J Gastroenterol. 1984;79:842– 843. 9. Annamalai AL, Shreekumar S. Tuberculosis of the esophagus. Am J Gastroenterol. 1972;57:166 –168. 10. Brullet E, Font B, Rey M, et al. Esophageal tuberculosis. Early diagnosis by endoscopy. Endoscopy. 1993;25:485.