Academia.eduAcademia.edu

Scrofuloderma (tuberculosis colliquativa cutis)

1996, British Journal of Dermatology

A 16-year-old girl presented with long-standing purulent sinuses to the skin overlying the lacrimal ducts, which had evolved into ulcers and resulted in hypertrophic scarring. The ulcers were serpiginous, with an uneven soft floor, and undermined edges. Histological examination revealed non-caseating tuberculoid granulomas in the dermis. Cultures for Mijcobaeterium tubereuhsis were positive. Chest X-ray did not show pulmonary involvement. Antituberculous therapy with isoniazid 300mg/day and rifampicin 6()()mg/day achieved a great improvement within 5 weeks of being started. A further improvement was seen at 4 months and complete resolution had occurred by 4 months. To the best of our knowledge, there are no other documented cases of scrofuloderma over the lacrimal system. The delay in the diagnosis emphasizes the importance of maintaining a high level of awareness.

British Journal of Dermatology 1996; 134: 350-352. Scrofuloderma (tuberculosis coUiquativa cutis) E.TUR. S.BRENNER AND Y.MEIRON* Departments of Dermatology and 'Ophthalmoloay. Tel Aviv Sourasky Medical Center. Tel Aviv University. Sackler School of Medicine. Tel Aviv. Israel Accepted for publication 15 March 1995 Summary A 16-year-old girl presented with long-standing purulent sinuses to the skin overlying the lacrimal ducts, which had evolved into ulcers and resulted in hypertrophic scarring. The ulcers were serpiginous, with an uneven soft floor, and undermined edges. Histological examination revealed non-caseating tuberculoid granulomas in the dermis. Cultures for Mijcobaeterium tubereuhsis were positive. Chest X-ray did not show pulmonary involvement. Antituberculous therapy with isoniazid 300mg/day and rifampicin 6()()mg/day achieved a great improvement within 5 weeks of being started. A further improvement was seen at 4 months and complete resolution had occurred by 4 months. To the best of our knowledge, there are no other documented cases of scrofuloderma over the lacrimal system. The delay in the diagnosis emphasizes the importance of maintaining a high level of awareness. Tuberculosis is still a significant health problem, and not only in developing countries. In the U.S.A. the numbers of cases are growing, mainly in immigrants and Native Americans.' Cutaneous tuberculosis, however, is uncommon." This report describes scrofuloderma over the lacrimal ducts in a 16-year-old girl with no other tuberculous foci. To the best of our knowledge, there are no other documented cases of scrofuloderma over the lacrimal system. Case report A 16-year-oId healthy girl presented to the dermatology clinic with a 2-year history of recurrent infections of the right lacrimal duct, with sinuses to the skin, following four operations for lacrimal duct obstruction (dacryocystorhynostomy). Her first operation was performed by a local doctor in Gaza, and there was no documentation available. During the second operation the right lacrimal system was probed and intubated and a purulent skin sinus was excised. The skin around the sinus was inflamed (Fig. 1), and bistological exiuiiination at this stage showed a mixed dermal inflammatory infiltrate composed of neutrophils and mononuclear cells. An improvement was noted following several weeks of topical antibiotic treatment, leaving a hypertrophic scar, for which treatment with steroid ointments was administered. An enlarging purulent ulcer developed under her Correspondence: Dr E.Tur, Department of Dermatology, Ichilov Medical Center, 6 Wcizman St. Tel Aviv, Israel 642 J9. 350 right eye. and a smaller one appeared under her left eye. The ulcers were serpiginous. with an uneven soft floor, and undermined edges. A biopsy specimen revealed non-caseating tuberculoid granulomas in the dermis (Fig. 2). No acid-fast bacilli were seen. Cultures for Mycohacterium tuberculosis were positive. Streptococcus pneumoniae and Candida were also cultured. Chest X-rays of the patient and of her immediate family did not show any pulmonary involvement. Antituberculous therapy, with isoniazid 3()()mg/day and rifampicin 60()mg/day. achieved a considerable improvement within 5 weeks of being started, with further resolution at 4 months and complete healing Figure t. A sinus is seen under right eye, with associated redness and scaling. The border of the sinus is elevated and serpiginous. 1996 British Association of Dermatologists SCROFULODERMA (TUBERCULOSIS COLUQUATIVA CUTIS) Figure 2. A pbolomicrograph showing non-casealing tuherculok! granulomas in tbe dermis (haematoxylin and eosin x 100). «' 351 ^ • .T- -•. * al 9 months. The great discomfort caused by tearshedding, which was still present after 4 months treatment, disappeared after 9 months, when the nasolacrimal ducts were patent. Her full blood count, and liver and renal function tests remained unaltered. Discussion Scrofuloderma results from the involvement and breakdown of the skin overlying a tuberculous focus. In most instances, the cervical lymph nodes are involved.^ Other cases are due to disease in lymph glands in other regions, or to bone involvement. As seen in our patient, excessive granulation tissue may occur, especially in cases secondary to tuberculosis of the bones. Our patient may have acquired tuberculosis from exogenous inoculation,""' dormant bacilli reactivated by trauma and non-specific inflammation induced by the operation,^'* or from lymphatic or haematogenous spread from an unknown primary focus. Possible immunosuppression by treatment with steroids might have aggravated the condition. Although there are no other reports of scrofuloderma over an infected lacrimal gland or duct, the probability of a preceding tuberculous infection in the lacrimal gland in our patient is high, because an obstruction of the lacrimal system at the age of 14 is unusual. Fistulation of a cold abscess probably resulted in the clinical presentation of scrofuloderma. Tuberculosis of the lacrimal gland was first described in 1881 by Abadie. and is extremely rare.'' Less than 50 cases have been reported. In over 20 years, only 14 cases of tuberculosis of the eye were recorded in India, where the prevalence of tuberculosis, including extrapulmonary disease, is high.'' Out of these 14 cases, only two had involvement of the lacrimal gland. Unlike our patient, they both had additional tuberculous foci elsewhere. The dissemination in these cases is usually haematogenous.'^''* Two other cases were described by Mortada from Cairo, They were of the localized isolated form which usually shows non-caseating granulomas.** Some of the reported cases were unilateral,''"^ whereas others were bilateral.^" ^ ^ In our patient, the disease started unilaterally and spread to involve the other side as well. Surgical treatment is usually required, in addition to chemotherapy, but sometimes chemotherapy alone may be sufficient.*"'^ as was the case here. Our patient successfully responded to short-term chemotherapy, as expected in scrofuloderma. This patient serves as a reminder that chronic facial sores may be due to tuberculosis and that the lacrimal gland may be a focus for tuberculous infection. The delay in the diagnosis emphasizes the importance of maintaining a high level of awareness for tuberculosis. Acknowledgment We are grateful to Prof. E.J.Feuerman for his helpful comments. References 1 Wortman PD. Pulmonary and cutaneous tuberculosis. / Am Acad Dermatol 1 9 9 2 : 2 7 : 4 5 9 - 6 0 . g) 1996 British Association of Dermatologists, British journal of Fkrmatology. \i4. J5O-i52 352 E.TUR etal 2 Sehgal VN, Wagh SA. Cutaneous tuberculosis. Current concepts. Ml Dermatol 1990; 29: 237-52. 3 Sehgal VN. Srivastava G, Khurana VK et al. An appraisal of epidemiologic, clinical, hacteriologic, histopathologic, and immunologic parameters in cutaneous tuberculosis. Int J Dermatol 1987:26: 521-6. 4 Kakakhel KV, Fritsch P. Cutaneous tuberculosis, Int } Dermatol 1989: 28: 355-62. 5 Chien |TT. Wiggins ML. Self-inoculation with M. tuberculosia and P. aeruginosa by a diabetic woman. Am Rev Tuberc 19 54: 69: 8 1 8 23. 6 Madhukar K. Bhide M, Prasad CE, Venkatramayya. Tuberculosis of the lacrimal gland,/Trap McffHi/ff 1991:94: 150-1. 7 Sen DK. Tuberculosis of the orbit and laerimal gland: a clinical study o f H cases. / Pediatr Ophthalmol Strabismus 1980; 17; 2 J2-8, 8 Mortada A, Tubercuioma of the orbit and laerimal gland, Br } Ophthalmol 1971: 55: 5fi5-7, 9 Baghdassarian SA, Zakharia H. Asdourian KK. Report of a case of hilateral caseous tuberculous dacryoadenltls. Am J Ophthalmol 1972: 74: 744-6, 10 Csiillog F, Gat L, Kelemen |T: On tuberculosis of the conjunctiva and the lacrimal sac. Report of a female patient wilh bilateral involvement, Klin Mbl Augenheilk 1966: 148: 79-86. 11 Offret G. Renard G. Pasticier. Dhermy P: A case of bilateral tuberculosis daryoadenitis. Bull Soc Ophthalmol /•> 1974: 74: 557-67. 1996 British Association of Dermatologists, British Journal of Dermatology. 134. 350-352