A Rare Case of Tubercular Cholesteatoma with TB Meningitis

Tubercular otitis media is an uncommon condition. Tuberculosis can affect any part of ear ranging from tympanic membrane to labyrinth. The symptoms of tubercular otitis media like painless otorrhea, multiple perforations, pale granulations, facial paralysis and severe SNHL though well described in literature, are not always present hence diagnosis is often missed. Diagnosis is usually made by clinical and histopathology examination of specimen obtained intra operatively. Here, we discuss clinical presentation, diagnosis and management of a case with cholesteatoma and TB meningitis.


Introduction
The incidence of tubercular otitis media ranges from 0.05% to 0.9% of all cases of chronic otitis media [1]. This is one of rarest forms of extrapulmonary tuberculosis and is often a missed diagnosis. There are no definite or pathognomic clinical signs and symptoms, making the diagnosis difficult in absence of laboratory findings [2].
Cholesteatoma in middle ear is not uncommon but presence of tubercular bacilli makes it a rare presentation. Tuberculosis should be included in the differential diagnosis of chronic middle ear infections not responding to routine management. We present an interesting case of tubercular otitis media with intracranial complication.

Case report
43 years old male presented to us with complaints of fever and left ear discharge for 20 days. He had history of similar discharge from left ear for which he un-derwent surgery 10 years back. He also gave history of right ear surgery for discharge 5 years back. This Patient was receiving treatment by physician for last 20 days with no significant improvement .Patient also had hypertension for last 4 years and Diabetes Mellitus for 5 years for which he was on regular medication. On our examination, patient was febrile with PR 98/min and BP 150/100mm of Hg. There was associated neck pain and rigidity. Kernig's sign was positive. Ophthalomologic examination showed right lateral rectus palsy. Detailed ENT examination was done which showed bilateral inadequate conchomeatoplasty. Right ear had healthy dry mastoid cavity with mobile neotympanum while left ear showed wet cavity with purulent discharge and cholesteatoma debris. Osscicles were not visualised. Facial nerve function was normal bilaterally. Pure tone audiometry showed 63.3 dB conductive deafness on right side and 65 dB mixed deafness on left side.
Considering the history that meningitis did not respond to high dose antibio- Pus culture and cholesteatoma matrix showed Staph aureus and AFB in many fields. Histopathology examination also showed presence of caseating granulomas with AFB in matrix ( Figure 2). Fluoroscent staining of cholesteatoma matrix was done, which also demonstrated AFB ( Figure 3). Hence diagnosis of cholesteatoma with TB and TB Meningitis was made. AKT was started. Patient was followed up after 15 days. Symptoms dramatically improved with marked reduction in neck rigidity and diplopia. Lateral rectus palsy recovered fully within 3 weeks post operatively.

Discussion
Tubercular otitis media is a very unusual cause of chronic otitis media, and is rarely considered in the differential diagnosis. Although the pathogenesis of

Conclusion
The main objective behind this article is to make ENT surgeon vigilant about this rare manifestation of very common disease in the Indian sub continent. Tuberculosis should be included in the differential diagnosis of chronic otitis media not responding to routine therapy or when presenting with intra cranial complication .Early diagnosis and timely initiation of anti tubercular therapy can prevent life threatening complications. and angiography can be applied to confirm diagnosis. In color ultrasound study the "yin-yang" sign indicates turbulent blood flow within the false aneurysm sac. The pulse Doppler study shows that "to and fro" pattern, which is obvious in communicating channel or neck of pseudoaneurysm, is not the sac. The "to" component is caused by enter of blood during systole and the "fro" component is seen during diastole when the blood stored in cavity is ejected in to the artery. Color Doppler ultrasound has sensitivity of 94% and specificity of 95% in different parts of the body for diagnosis of pseudoaneurysm [8].
Pseudoaneurysm in CT is demonstrated as hypoattenuating (non-contrast) or hyperattenuating (contrast-enhanced) smooth walled sac adjacent to a vessel and contrast enhanced MRI which shows high signal sac of pseudoaneurysm [1].
Uterine artery embolization is preferred treatment but ligation of affected uterine artery by surgery and hysterectomy are alternative treatments.
In this case report we present a uterine artery pseudoaneurysm diagnosed one week after Cesarean section and ruptured before endovascular therapy.

Case Presentation
A 25 years old gravid 2 woman presented with abdominal pain and vaginal bleeding 1 week after Cesarean section surgery. She was asymptomatic during 1 week after operation. Mild tenderness in left lower quadrant on abdominal examination was detected. Laboratory data showed Hb: 10.5 and WBC count 10,000 at time of admission. Blood pressure and temperature was 110/70, 37/5 respectively.
In Ultrasound study there was a round hypoecho mass measured 4 cm with echo free center in left adnexa. Color Doppler demonstrated typical "ying-yang" sign in center of mass which is characteristic for pseudoaneurysm (Figure 1).
Pulse Doppler at the neck of mass showed "to and fro" pattern ( Figure 2). Following ultrasound study CT angiography of pelvis and MRI was requested to confirm diagnosis of pseudoaneurysm.
A. R. Azandaryani et al.  CT angiography showed a collection of contrast adjacent to left internal iliac artery suggestive of pseudoaneurysm ( Figure 3).
Contrast enhanced MRI demonstrated enhanced center of psudoaneurysm and peripheral hematoma (Figure 4).  Patient was admitted to treat by endovascular intervention. Several hours after admission blood pressure decreased and abdominal pain and vaginal bleeding became more sever. Ultrasound study showed evidence of free fluid in abdominopelvic cavity which was suggestive of pseudoaneurysm rupture. Although hysterectomy is the last choice in treatment of uterine artery pseudoaneurysm, due to rupture of pseudoaneurysm and because ligation of internal iliac artery was not effective, hysterectomy was done to control bleeding. Patient was dis-

Discussion
Pseudoaneurysm in pelvic vessels is a rare complication of pelvic surgery. Cesarean section is the most common cause but this complication has also been reported in association with abortion, repeated curettage, myomectomy, hysterectomy and even uncomplicated vaginal delivery [4].
Ultrasound, CT angiography, MRI and angiography are modalities applied to diagnosis of this complication [1].
Pseudoaneurysm of uterine artery is a rare cause of delayed postpartum hemorrhage. Delayed postpartum hemorrhage is defined as bleeding from 24 hours after delivery up to 6 weeks postpartum and most commonly occurring between 8 and 14 days postpartum. Retained products of conception, subinvolution of the placental bed and endometritis are more Common causes of delayed postpartum hemorrhage [9].
Treatment includes ligation of uterine artery by surgery or endovascular embolization but endovascular embolization of involved artery is preferred treatment. Hysterectomy is the last choice if uterine artery ligation by surgery or embolization is not effective to control bleeding [4]. The first case of selective arterial embolization that used successfully to treat uncontrollable postpartum bleeding was reported by Brown et al. in 1979 [10]. Angiographic embolization has the advantages of decreased morbidity, ability to localize the bleeding site and provide a more distal occlusion than surgical ligation, and preservation of future fertility compared to hysterectomy [9].

Conclusion
Uterine artery pseudoaneurysm should be considered in the differential diagnosis of delayed postpartum hemorrhage. It occurs most commonly after Cesarean section but is also associated with abortion, repeated curettage, myomectomy, hysterectomy and vaginal delivery. Ultrasound, CT angiography and MRI are modalities applied to diagnosis of this complication. Uterine artery embolization is preferred treatment but ligation of uterine artery by surgery and hysterectomy are alternative treatments.