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Rhinoscleroma in an Octogenarian Woman
CASE REPORT
Rhinoscleroma in an Octogenarian Woman A. Leibovitz, MD, I. Gil, MD, G. Plotnikov, MD, D. Stempler, MD, S. Zahavi, MD, and B. Habot, MD Rhinoscleroma, a granulomatous infection caused by Klebsiella rhinoscleromatis and usually afflicting young adults, was diagnosed in an 81-year-old woman. Treatment was started with orally administered antibiotics but was soon halted because of side effects. She was treated instead by local spread of a 3% rifampin ointment. After 6 weeks, the lesion cleared up and she improved remarkably. There was no recurrence during 18 months of follow-up. The cur-
rent report is the second published case of rhinoscleroma in an elderly person and the first report on its cure by local treatment with a 3% rifampin ointment. This favorable experience contributes to the limited knowledge on rhinoscleroma in the elderly. (J Am Med Dir Assoc 2002; 3: 71–72) Keywords: Rhinoscleroma; elderly; rifampin.
Rhinoscleroma is a granulomatous infection of the upper airways caused by Klebsiella rhinoscleromatis. It is prevalent in endemic areas (Middle East, East Europe, India, and Central America) and affects mainly young adults.1 Immigration patterns could lead to increasing frequency all over the world.2 We report here for the first time in the English language medical literature a case of rhinoscleroma in an elderly patient. CASE DESCRIPTION An 81-year-old women resident of a nursing home progressively deteriorated over several months. She was anorectic, apathetic, uncooperative, and her weight was 37 kg. A cystic lesion 3 cm in diameter with mild oozing and crust was noticed at the opening of her right nostril. A shave biopsy was performed but was inconclusive. Her condition continued to deteriorate, and the lesion enlarged to 4 cm in diameter, nearly obstructing the right nostril (FIG. S.). A repeated biopsy—this time a 4 mm punch biopsy— revealed rhinoscleroma, with Mikulicz cells and a number of Russell bodies. A full ear, nose, and throat examination did not detect extension of the disease. Treatment with oxytetracycline 100 mg/day was started, but in the following days the patient developed thrombocytopenia (platelet count dropped from 170,000 to 44,000 per Long-Term Care Department (A. L. and G.P.), and Acute Geriatric Department (I. G. and B. H.), Shmuel Harofe Hospital, Geriatric Medical Center. Affiliated with the Sackler Faculty of Medicine, Tel-Aviv University, Dermatologic Unit (D. S.), and Pathologic Department (S. Z), Asaf Harofe Medical Center, Zerifin. Address correspondence to: Arthur Leibovitz, Shmuel Harofe Hospital, POB 2, Beer-Yacov, Israel.
Copyright ©2002 American Medical Directors Association CASE REPORT
Fig. S. Before treatment with rifampin, the lesion had enlarged to 4 cm in diameter, nearly obstructing the right nostril.
cubic milliliter) and this drug was halted. Ciprofloxacin was given instead, but it induced nausea, and the patient refused it repeatedly. Looking for an alternative, we came to the report of Gamea and El-Tatawi who treated rhinoscleroma locally with a 2% rifampin ointment.3 Because they mentioned insufficient cure in some cases, we decided to use a 3% rifampin ointment, which was applied on the lesion twice a day. This ointment was prepared by our pharmacist by mixing 3 g rifampin (the content of 10 capsules of Rimactane 300 mg (Biochemie GmbH, Kundl, Austria) with 87 g soft white paraffin and 10 g liquid paraffin. After 2 weeks of this treatment, an improvement was noticed. The patient became alert, cooperative, and started to eat willingly. By the fourth week the lesion shrank to 1 cm, the right nostril was unobstructed, and she gained 2 kg in her Leibovitz et al. 71
Fig. T. By the sixth week, the lesion had disappeared, leaving only a small area of mild hypopigmentation.
weight. The lesion disappeared after the sixth week, leaving only a small area of mild hypopigmentation (FIG. T). Treatment was continued for another 2 weeks and stopped thereafter. After 18 months, there was no sign of recurrence. Her weight was then 44 kg. She lived in the nursing home, still wheelchair-bound but happily enjoying occupational activities and the visits of her family. DISCUSSION This is the first case of rhinoscleroma in an elderly patient in the English language medical literature, and only the second case reported at all. The first case of rhinoscleroma in an elderly patient hitherto published was that of an old
72 Leibovitz et al.
Danish woman4 treated with both oral ciprofloxacin and surgery but with no complete resolution. Our patient presented with anorexia, weight loss, malnutrition and depressive features. All these symptoms cleared up following the healing of the rhinoscleroma. It suggests a state of geriatric failure to thrive.5 The main point to be emphasized regarding this case is our success with the local treatment by a 3% rifampin ointment. The only report on rifampin ointment to treat rhinoscleroma is that of Gamea.3 He used a 2% preparation for 6 weeks and mentioned that a longer treatment period is needed for complete healing. The rapid recovery in our case and the lack of recurrence after 18 months suggest that a 3% preparation may be more efficient. It is also noteworthy that the alternative of a local treatment should be preferred in elderly patients who may have adverse effects to orally administrated drugs as did the patient in our case. Our favorable experience with this case contributes to the limited knowledge on rhinoscleroma in the elderly. REFERENCES 1. Stiernberg CM, Clark WD. Rhinoscleroma–A diagnostic challenge. Laryngoscope, 1983;866 – 870. 2. Andraca R, Edson RS, Kern EB. Rhinoscleroma. A Growing concern in the United States. Mayo Clin Proc 1993;68:1151–1157. 3. Gamea AM, El-Tatawi FAY. The effect of rifampicin on rhinoscleroma: An electron microscopic study. J Laryngol Otol 1990;104:772–777. 4. Stougaard MJ, Vimpel T, Petersen K. Rhinoscleroma in an elderly Danish woman. Ugeskr Laeger, 1996;158:5041–5043. 5. Lonergan ET, ed. Extending Life, Enhancing life. A National Research Agenda on Ageing. Washington DC. National Academy Pr. 60, 1991.
JAMDA – March/April 2002
Rhinoscleroma in an Octogenarian Woman A. Leibovitz, MD, I. Gil, MD, G. Plotnikov, MD, D. Stempler, MD, S. Zahavi, MD, and B. Habot, MD Rhinoscleroma, a granulomatous infection caused by Klebsiella rhinoscleromatis and usually afflicting young adults, was diagnosed in an 81-year-old woman. Treatment was started with orally administered antibiotics but was soon halted because of side effects. She was treated instead by local spread of a 3% rifampin ointment. After 6 weeks, the lesion cleared up and she improved remarkably. There was no recurrence during 18 months of follow-up. The cur-
rent report is the second published case of rhinoscleroma in an elderly person and the first report on its cure by local treatment with a 3% rifampin ointment. This favorable experience contributes to the limited knowledge on rhinoscleroma in the elderly. (J Am Med Dir Assoc 2002; 3: 71–72) Keywords: Rhinoscleroma; elderly; rifampin.
Rhinoscleroma is a granulomatous infection of the upper airways caused by Klebsiella rhinoscleromatis. It is prevalent in endemic areas (Middle East, East Europe, India, and Central America) and affects mainly young adults.1 Immigration patterns could lead to increasing frequency all over the world.2 We report here for the first time in the English language medical literature a case of rhinoscleroma in an elderly patient. CASE DESCRIPTION An 81-year-old women resident of a nursing home progressively deteriorated over several months. She was anorectic, apathetic, uncooperative, and her weight was 37 kg. A cystic lesion 3 cm in diameter with mild oozing and crust was noticed at the opening of her right nostril. A shave biopsy was performed but was inconclusive. Her condition continued to deteriorate, and the lesion enlarged to 4 cm in diameter, nearly obstructing the right nostril (FIG. S.). A repeated biopsy—this time a 4 mm punch biopsy— revealed rhinoscleroma, with Mikulicz cells and a number of Russell bodies. A full ear, nose, and throat examination did not detect extension of the disease. Treatment with oxytetracycline 100 mg/day was started, but in the following days the patient developed thrombocytopenia (platelet count dropped from 170,000 to 44,000 per Long-Term Care Department (A. L. and G.P.), and Acute Geriatric Department (I. G. and B. H.), Shmuel Harofe Hospital, Geriatric Medical Center. Affiliated with the Sackler Faculty of Medicine, Tel-Aviv University, Dermatologic Unit (D. S.), and Pathologic Department (S. Z), Asaf Harofe Medical Center, Zerifin. Address correspondence to: Arthur Leibovitz, Shmuel Harofe Hospital, POB 2, Beer-Yacov, Israel.
Copyright ©2002 American Medical Directors Association CASE REPORT
Fig. S. Before treatment with rifampin, the lesion had enlarged to 4 cm in diameter, nearly obstructing the right nostril.
cubic milliliter) and this drug was halted. Ciprofloxacin was given instead, but it induced nausea, and the patient refused it repeatedly. Looking for an alternative, we came to the report of Gamea and El-Tatawi who treated rhinoscleroma locally with a 2% rifampin ointment.3 Because they mentioned insufficient cure in some cases, we decided to use a 3% rifampin ointment, which was applied on the lesion twice a day. This ointment was prepared by our pharmacist by mixing 3 g rifampin (the content of 10 capsules of Rimactane 300 mg (Biochemie GmbH, Kundl, Austria) with 87 g soft white paraffin and 10 g liquid paraffin. After 2 weeks of this treatment, an improvement was noticed. The patient became alert, cooperative, and started to eat willingly. By the fourth week the lesion shrank to 1 cm, the right nostril was unobstructed, and she gained 2 kg in her Leibovitz et al. 71
Fig. T. By the sixth week, the lesion had disappeared, leaving only a small area of mild hypopigmentation.
weight. The lesion disappeared after the sixth week, leaving only a small area of mild hypopigmentation (FIG. T). Treatment was continued for another 2 weeks and stopped thereafter. After 18 months, there was no sign of recurrence. Her weight was then 44 kg. She lived in the nursing home, still wheelchair-bound but happily enjoying occupational activities and the visits of her family. DISCUSSION This is the first case of rhinoscleroma in an elderly patient in the English language medical literature, and only the second case reported at all. The first case of rhinoscleroma in an elderly patient hitherto published was that of an old
72 Leibovitz et al.
Danish woman4 treated with both oral ciprofloxacin and surgery but with no complete resolution. Our patient presented with anorexia, weight loss, malnutrition and depressive features. All these symptoms cleared up following the healing of the rhinoscleroma. It suggests a state of geriatric failure to thrive.5 The main point to be emphasized regarding this case is our success with the local treatment by a 3% rifampin ointment. The only report on rifampin ointment to treat rhinoscleroma is that of Gamea.3 He used a 2% preparation for 6 weeks and mentioned that a longer treatment period is needed for complete healing. The rapid recovery in our case and the lack of recurrence after 18 months suggest that a 3% preparation may be more efficient. It is also noteworthy that the alternative of a local treatment should be preferred in elderly patients who may have adverse effects to orally administrated drugs as did the patient in our case. Our favorable experience with this case contributes to the limited knowledge on rhinoscleroma in the elderly. REFERENCES 1. Stiernberg CM, Clark WD. Rhinoscleroma–A diagnostic challenge. Laryngoscope, 1983;866 – 870. 2. Andraca R, Edson RS, Kern EB. Rhinoscleroma. A Growing concern in the United States. Mayo Clin Proc 1993;68:1151–1157. 3. Gamea AM, El-Tatawi FAY. The effect of rifampicin on rhinoscleroma: An electron microscopic study. J Laryngol Otol 1990;104:772–777. 4. Stougaard MJ, Vimpel T, Petersen K. Rhinoscleroma in an elderly Danish woman. Ugeskr Laeger, 1996;158:5041–5043. 5. Lonergan ET, ed. Extending Life, Enhancing life. A National Research Agenda on Ageing. Washington DC. National Academy Pr. 60, 1991.
JAMDA – March/April 2002