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Case reports

Vol. 43, 2007



bullet 4'2007

Simple (nonparasitary) hepatic cyst. Literature review and a case presentation  – 43, 2007, No 4,  80-84.
F. Mitov, L. Nachev, At. Kalchev, Il. Bodurov and V. Molov
Clinic of Surgery, Military Hospital -- Plovdiv
Summary: After in detail literature review of the matter, incidence, dimensions, preferred sex and age, and complications in nonparasitary hepatic cysts, authors described a case of a giant cyst of this type. The formation was localized in the right subdiaphragmatic hepatic region with dimensions of 192.0/110.0 mm. Diagnosis was based on thorax X-ray and abdominal CT scan. The formation was approached through a right toracophrenolaparotomy. A total cyst and pericystectomy and a plasty of the diaphragm were performed. Six months later, the patient recovery was completed without signs of disease recurrence.
Key words: hepatic cyst, nonparasitary, simple.
Address for correspondence: F. Mitov, M. D., Clinic of Surgery, Military Hospital, 81, Hr. Botev Blvd., Bg -- 4000 Plovdiv


Clear cell odontogenic carcinoma -- diagnostic problems – 43, 2007, No 4, 85-87.
E. Poryazova(1), Z. Zapryanov(1), G. Ivanov(1), Hr. Petrov(2) and St. Peycheva(3)
(1)Department of Clinical Pathology, (2)Clinic of Otorhinolaryngology, (3)Clinic of Oral and Maxillofacial Surgery, UMHAT “Sv. Georgi” -- Plovdiv
Summary: Malignant odontogenic tumors of the jaws are rare. The clear cell odontogenic carcinoma (CCOC) is a locally infiltrative neoplasm with a marked tendency for local recurrence and regional lymph node metastases. Histologic and immunohistochemical examination of the tumor cells was carried out. Differential diagnostic problems are discussed. We describe a case of CCOC in a 32-year-old female who presented with a swelling and destruction of the left body of the mandible, in premolar area of the jaw. The histological features of the tumor presented nests and sheets of large clear cells, which were separated by mature, fibrous septae. The tumor was not encapsulated. The clear cells contained glycogen. Immunohistochemically the tumor cells were positive for cytokeratin and S-100 protein, but were negative forvimentin, desmin and actin. Tumors with consistent clear cell component in the head and neck region can impose serious problems with respect to differential diagnosis. They can originate from various sources, including salivary gland tumors, metastatic renal cell carcinoma, melanomas and other odontogenic tumors, such as ameloblastoma and calcifying epithelial odontogenic tumor.
Key words: malignant odontogenic tumor, clear cell
Address for correspondence: Elena Poryazova, M. D., Department of Clinical Pathology, UMBAL “Sv. Georgi”, 15А, V. Aprilov Blvd., Bg -- 4002 Plovdiv, e-mail: eporiazova@abv.bg


Stevens--Johnson syndrome: still a great pathogenetic mystery and a common dermatological challenge  – 43, 2007, No 4, 88-91.
G. Tchernev(1), M. D. Apostolova(1), Y. Georgieva(2), A. Kotzev(3) and A. Zhelyaskov(4)
(1)Department of Molecular and Cell Biology, Bulgarian Academy of Sciences -- Sofia, (2)Free Universitaet -- Berlin, (3)Department of Internal Diseases and Gastroenterology, University Hospital Alexandrovska -- Sofia, (4)Department of Dermatology and Venereology, University Hospital Alexandrovska -- Sofia
Summary: We present a rare case of a female patient suffering from the Stevens--Johnson syndrome, generated after the introduction of a combined antibiotic therapy for bronchopneumonia.  Simultaneously, a systematic therapy with ACE (angiotensin converting enzyme) inhibitor has been introduced, in order to optimize the antihypertensive therapy, as well as antiviral therapy with aciclovir as herpes of the lips was suspected. Immediately after, symmetrical skin lesions appeared, with bizonal configuration trending to confluence, accompanied by hemorrhagic blister formation with palmar and plantar localization. Although it may be supposed that EM (erythema exudativum multiforme) could be an immunologic reaction of tardy type with respect to some viral or medical antigens, the exact mechanisms and their sequence are not completely clear. Frequently, they are of complex nature and include the presence of certain infectious bacterial and/or viral agents, as well as newly prescribed medication.  The specific dermatological tests such as epicutaneous, prick-, scratch- and oral provocative tests made after a successful therapy, in most of the cases are negative.
Key words: erythema multiformе, allergy, muco-cutaneous reaction, bizonal configuration
Address for correspondence: Georgi Tchernev, M. D., Department of Dermatology, Venereology, Allergology and Pneumology, Ostseeclinic Kuehlungsborn, Waldstrasse 51, 18225 Kuehlungsborn, Germany, GSM +49 1622643815, e-mail: georgi_tchernev@yahoo.de



Use of Sildenafil in pulmonary arterial hypertension associated with scleroderma (Presentation of a case)   – 43, 2007, No 4, 92-94.
T. Isidorova and M. Panchovska
(1)Clinic of Rheumathology, Specialized Hospital of Orthopedics and Traumatology, Vogelsang/Gomern at Magdeburg University, Germany, (2)Therapeutic Clinic, Military Medical Academy -- Plovdiv
Summary: Pulmonary arterial hypertension is one of the serious complications of progressive scleroderma. According to literature data, from 8 to 10% of the scleroderma patients develop pulmonary arterial hypertension. About 50% of the patients exhibit clinical manifestations corresponding to physical capacity restriction of NYHA functional class III and IV. Treatment of pulmonary hypertension with calcium antagonists, prostacyclin analogs and endothelin-1 receptor antagonists has no effect in most of the cases. The use of Sildenafil (the first oral phosphodiesterase-5 inhibitor) significantly improves the signs of increased pulmonary pressure, the hemodynamic indices and the symptoms of right heart failure. In the paper, the results of Sildenafil treatment of a patient with pulmonary arterial hypertension associated with scleroderma are presented.
Key words: progressive systemic sclerosis, pulmonary arterial hypertension, Sildenafil
Address for correspondence: Assoc. Prof. Maria Panchovska, Therapeutic Clinic, Military Medical Academy, 81, Hr. Botev Blvd., Bg -- 4000 Plovdiv, e-mail: panchovska@abv.bg




bullet 3'2007

Lymphogranuloma venereum of the penis mimicking clinically gangrenous pyoderma and presenting histologically as macrophagocytic-histiocytic balanitis – 43, 2007, No 3, 90-94.   
G. Tchernev
Department of Dermatology and Immunology, Municipal Hospital Dessau, Academic Educational Hospital of the Martin Luther University -- Halle-Wittenberg, Germany
Summary: Lymphogranuloma venereum (LGV) is a rare, sexually transmitted disease, caused by Chlamydia trachomatis, serotypes L1, L2 and L3. The lesions are mostly anogenitally located. We are describing a case with a papula, turning into dolorous ulceration in the genital area. The histological analysis of the lesion showed strongly expressed lymphocytic, as well as macrophagocytic-histiocytic infiltrates with some eosinophilic granulocytes. The immunohistochemistry showed chronic fibrous macrophagocytic-histiocytic inflammation reaction. The data about histological finding of venereal lymphogranuloma in the initial stage of the disease are very poor in the literature. It is not clear if the macrophagocyte/histiocyte reaction proved by us can be used as a standard reaction in the diagnostics of a sexually transmitted disease at the so called ulcerative stage of LGV. Some overlap of the symptoms with ulceration induced by immunologic way within the rheumatoid arthritis is also possible. The full remission after the applied antibiotic therapy supports the diagnosis and the specific histological finding.
Key words: ulceration, lymphogranuloma venereum, Chlamydia trachomatis, doxycyclin
Аddress for correspondence:
Georgy Tchernev, M. D., Department of Dermatology and Immunology, Municipal Hospital, Auenweg 38, 06847 Dessau, tel. 0049 1622643815 (GSM), e-mail: georgi_tchernev@yahoo.de


Nontraumatic (spontaneous, idiopathic) subcapsular rupture of intact spleen – 43, 2007, No 3, 95-98. 
F. Mitov, L. Nachev, At. Kalchev, Chr. Georgiev, Il. Bodurov and V. Molov
Surgery Clinic, Clinic of Internal Diseases, Military Hospital -- Plovdiv
Summary: After a review of the literature, authors describe a case of a 69-year-old man with atraumatic (spontaneous) subcapsular rupture of intact spleen. The patient was treated for intercostal neuralgia for a period of 40 days. Because of the worsening of the health condition, a consultation with internist was performed. The primary diagnosis was “left pleuropneumonia”. The consulting surgeon suggestion was “splenic rupture” and this was proved during the operation and histological examination. Authors consider the case corresponding to M. Orloff and G. Peskin criteria. This case is the first one described in Bulgaria, presenting a rare pathology with very difficult diagnosis.
Key words: splenic rupture, spontaneous, idiopathic, nontraumatic
Address for correspondence:
Prof. F. Mitov, M. D., Surgery Clinic, Military Hospital, 21, Peshtersko Shose Blvd., Bg -- 4002 Plovdiv


Tuberculous meningitis with atypical course -- case study – 43, 2007, No 3, 99-102. 
K. Kostov, I. Petrov, R. Ikonomov and K. Calovski
Neurology Clinic, Medical Institute, Ministry of Interior -- Sofia
Summary: Tuberculous meningitis is a comparatively rare disease in developed countries. With its varying clinical signs and difficult diagnosis, it is still a diagnostic and therapeutic challenge to the clinicist. The literature sources evidence only a few cases of tuberculosis infection combined with another pyogenic flora. The paper presents a case with atypical course of tuberculous meningitis combined with possible pyogenic meningitis in a male patient, aged 35. The onset of the disease was acute with septic temperature curve, leukocytosis, meningeal syndrome, proteinorachy and mainly mononuclear pleocytosis. Pseudomonas aeruginosa were isolated from the liquor. The aggravation of the patient’s status despite administered antibiotic therapy, and the increasing liquor protein and mononuclear pleocytosis at sterile inoculations were the reason to start tuberculostatic therapy in spite of the atypical clinical signs, normal levels of ADA and negative liquor and serum PCR. Persisting liquor pleocytosis and proteinorachy were found up to the 3rd month after beginning the therapy although there was outlined clinical improvement. The diagnosis was confirmed by positivation of inoculations into Lowenstein-Jensen medium on the 52nd day. The presented case confirms the fact that in spite of the advanced current immunological and molecular methods the early diagnosis of tuberculous meningitis is mainly clinical and liquorological.
Key words: tuberculous meningitis, diagnosis, treatment
Address for correspondence:
K. Kostov, MD, PhD, Clinic of Neurology, Medical Institute of Ministry of Interior, 79 “Scobelev” Blvd., Bg -- 1606 Sofia, tel. +359 2 9821576, Fax: +359 2 9531235, e-mail: drkostov@abv.bg





bullet 2'2007

A case of septic anthrax -- diagnostic problem and perspectives in the contemporary microbiological diagnosis   –  43, 2007, No 2, 94-98.
I. Valcheva-Komitska(1), L. Dimitrova(1), Y. Borisova(1), R. Nenova(2), I. N. Ivanov(2) and P. Kircheva(3)
(1)Multiprofile Hospital for Active Treatment -- Dobrich
(2)National Center of Infectious and Parasitic Diseases -- Sofia
(3)Regional Inspectorate for Protection and Control of Public Health -- Dobrich

Summary: This study describes a rare case of septic anthrax that took fudroyant course and caused difficulties in the establishment of clinical diagnosis in a female patient hospitalized with acute inferior myocardial infarction. After four days in the hospital, the patient died with severe sepsis and multiorgan failure. There were anamnestic and epidemiological data for contact with domestic animals, however the entry and mechanism of infection remained obscure. The initial differentialdiagnostic investigations supposed probable hemorrhagic fever as a cause of the severe presentations. The diagnosis septic anthrax was considered post mortem when the initial direct microscopic findings of the positive blood cultures demonstrating bacilli were taken into account. The anthrax diagnosis was microbiologically established at the hospital laboratory together with the laboratory of the Regional Inspectorate for Protection and Control of Public Health in Dobrich. The isolated strain was confirmed by classical and contemporary microbiological methods at the National Reference Laboratory for High-Risk Bacterial Infections under National Center for Infectious and Parasitic Diseases.
Key words: anthrax, sepsis, diagnosis, bioterrorism
Address for correspondence:
Iva Valcheva-Komitska, M. D., Microbiological Laboratory, Multiprofile Hospital for Active Treatment, 23 ”Panayot Hitov” Str., Bg -- 9300 Dobrich, e-mail: ivavk@gbg.bg



Pseudohypoparathyroidism or pseudopseudohypoparathyroidism   –  43, 2007, No 2, 99-104.
in a family with Albright hereditary osteodystrophy
V. Boyadzhiev(1), V. Iotova(1), K. Hristozov(2) and M. Boyadzhieva(2)
(1)Department of Pediatrics and Medical Genetics
(2)Clinic of Endocrinology and Metabolism
Medical University -- Varna

Summary: We present a family -- a mother (34-year-old) and her two children -- a boy (14-year-old) and a girl (10-year-old) with typical Albright hereditary osteodystrophy features. They were investigated for impaired Ca-P metabolism and multihormonal resistance. The results showed the most commonly observed combination of the two major subforms -- pseudohypoparathyroidism and pseudopseudohypoparathyroidism, represented in one and the same family. An overview of the current knowledge about the genetic background and the ways of inheritance is made. The appropriate diagnostic approach and the need for treatment are discussed.
Key words: pseudohypoparathyroidism, GNAS-1, treatment
Address for correspondence:
V. Boyadzhiev, M. D., Department of Pediatrics and Medical Genetics, Medical University, 55, M. Drinov Str., Bg -- 9000 Varna



bullet 1'2007

A case of large ovarial mucinous cystadenoma – 43, 2007, No 1, 90-92.
E. Kovatchev and Ya. Kornovski
Department of Obstetrics and Gynecology, Medical University -- Varna

The aim of our study was to demonstrate the nontypical clinical progression of some ovarial tumors and to have a focused diagnostic approach on them in cases of unspecified abdominal discomfort and symptoms from gastrointestinal tract. We report a case of a 45-year-old patient with mucinous cystadenoma. Ultrasound imaging revealed a huge cystic ovarian mass without ascites. Laparotomy, right oophorectomy and omentectomy were performed for the ovarian tumor. Histology revealed a large mucinous cystadenoma.
Key words: large mucinous cystadenoma, ovarial tumor
Address for correspondence:
Е. Kovatchev, Department of Obstetrics and Gynecology, Medical University, 55, M. Drinov Blvd., Bg -- 9000 Varna А


Clinical case of apical hypertrophic cardiomyopathy  – 43, 2007, No 1, 93-96.
K. Koleva, N. Zlatareva and A. Goudev

Department of Cardiology, University Hospital “Tsaritsa Ioanna” -- Sofia

Apical hypertrophic cardiomyopathy (AHCMP) is a subtype of the hypertrophic cardiomyopathy (HCMP). HCMP was described 100 years ago and is a complex and relatively common genetic heart disease (at about 1:500 among the adult population). In 1979, a variant of the HCMP -- the AHCMP, was described by Sakamoto. The hypertrophy affects mainly the left ventricle and has a typical “spadelike”?shape of the cavity of the LV. In about 50% of the cases, there are inheritable features. It is an autosomal dominant disease but rarely cases there are sporadic cases. It is equally presented in both men and women. HCMP is a relatively common cause of sudden cardiac death in the young, but may cause different damages in all generations. In some of the more rare cases, the hypertrophy is localized in the apical region of the left ventricle and the disease is called AHCMP. The studied subjects were Japanese men so the disease is called “Japanese” type HCMP. It is common in Asia and presents 1/4 of the HCMP in Japanese population. The disease is relatively rare in other parts of the world. It is not typical for the European population, and for Bulgarian one too.
Key words: hypertrophic cardiomyopathy, apical hypertrophy
Address for correspondence:
Kalina Koleva, M. D., Department of Cardiology, University Hospital “Tsaritsa Ioanna”, 8, Bialo more Str., Bg -- 1527 Sofia, tel. +359 2 9432-556, GSM 0886314473, e-mail: med_help@yahoo.com


 Two clinical observations of histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto’s disease) – 43, 2007, No 1, 97-100.
I. Zhutev(1), E. Nakov(2), D. Dimov(1), K. Stanimirova(1) and D. Ivanova(1)
(1)Rheumatological Department
(2)Pathomorphological Laboratory
Military Medical Academy -- Sofia

The authors describe two own clinical observations of the rare disease histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto’s disease). The illness has an extensive differential diagnosis which can be resolved by a histological examination of a biopted lymph node. The authors keep to an activer therapeutic approach considered with the clinical form of the disease using corticosteroidis in a more severe course with extranodal involvement.
Key words: Kikuchi-Fujimoto’s disease, diagnostics, treatment
Address for correspondence:
Prof. Detelin Dimov, Military Medical Academy, 3, Sv. Gеorgi Sofiyski Str., Bg --  1606 Sofia,
tel. +359 2 922 57 35


Fuchs’ syndrome simulating cicatricial (mucous membrane) pemphigoid – 43, 2007, No 1, 101-104.
G. Tchernev
Department of Dermatology and Immunology, Municipal Hospital -- Dessau, Academic Educational Hospital of Martin Luther University Halle-Wittenberg, Germany

Summary: Fuchs’ syndrome could be viewed as a side allergic (probably drug induced) reaction of vague origin caused most frequently by taking antibiotics like macrolides, sulfonamides, penicillin and derivatives, and also non-steroid anti-inflammatory drugs like aspirin in the course of an acute viral inflammation or infection due to a bacterial agent. Characteristic clinical symptom in the affected patients is the presence of erosions which are strictly confined within the area of the mucous membranes of the oral cavity and periocularly. Lacking are the bizonally configured cockades, which are typical of erythema exudativum multiforme. The skin of the patients remains intact in more than 90% of the cases. The pathogenesis of the described phenomenon as well as the reason for the “atypical localization” are not known. We present a rare case of a patient with Fuchs’ syndrome which we define as a subvariant of erythema exudativum multiforme. Timely diagnostics and therapy of the syndrome help avoid complications that most often affect the eye in the form of keratitis, uveitis, or iridocyclitis. The most frequently performed correction for these complications is surgery.
Key words: allergy, Fuchs’ syndrome, atypical localization, corticosteroids
Аddress for correspondence: Georgy Tchernev, M. D., Department of Dermatology and Immunology, Municipal Hospital, Auenweg 38, 06847 Dessau, tel. 0049 1622643815 (GSM), e-mail: georgi_tchernev@yahoo.de




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