Limits...
Bilateral pleural effusion and interstitial lung disease as unusual manifestations of Kikuchi-Fujimoto disease: case report and literature review.

Garcia-Zamalloa A, Taboada-Gomez J, Bernardo-Galán P, Magdalena FM, Zaldumbide-Dueñas L, Ugarte-Maiztegui M - BMC Pulm Med (2010)

Bottom Line: All of them resolved with prednisone and after two years of following up he remains asymptomatic and without evidence of any other associated disease.Pleural effusion and interstitial lung disease are very uncommon manifestations of KFD.In our experience, treatment with oral prednisone was effective.

View Article: PubMed Central - HTML - PubMed

Affiliation: Mendaro Hospital, Gipuzkoa, Spain. ALBERTO.GARCIA-ZAMALLOAZAMALLOA@OSAKIDETZA.NET

ABSTRACT

Background: Kikuchi-Fujimoto's disease (KFD), also called histiocytic necrotizing lymphadenitis, is a rare, idiopathic and self-limited condition usually characterized by cervical lymphadenopathy and fever, most often affecting young patients. Aetiology is unknown. Differential diagnosis includes mainly malignant lymphoma, tuberculous lymphadenitis and systemic lupus erythematosus (SLE), so early diagnosis is crucial. Pleuropulmonary involvement due to isolated KFD has been seldom reported.

Case presentation: a 32-year-old man, on treatment for iatrogenic hypothyroidism, was admitted due to high grade fever and painful cervical lymphadenopathies. KFD was diagnosed by lymph node biopsy. Some days after admission the patient got worse, he developed generalized lymphadenopathy, bilateral pleural effusion and interstitial lung disease. All of them resolved with prednisone and after two years of following up he remains asymptomatic and without evidence of any other associated disease.

Conclusion: Pleural effusion and interstitial lung disease are very uncommon manifestations of KFD. In our experience, treatment with oral prednisone was effective.

Show MeSH

Related in: MedlinePlus

On admission both lung fields were clear and one isolated mediastinal lymph node was observed.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
getmorefigures.php?uid=PMC2991292&req=5

Figure 1: On admission both lung fields were clear and one isolated mediastinal lymph node was observed.

Mentions: A 32-year-old Caucasian man, with a history of hyperthyroidism six months previous due to toxic multinodular goiter, treated with radioactive iodine, who developed iatrogenic hypothyroidism and on initiation of substitutive treatment, was admitted due to persistent fever (39-40°C), malaise and painful cervical lymphadenopathies that had been present for two weeks. Laboratory findings were as follows: haematocrit 34%, haemoglobin 12 g/dl, leukocyte count 3400/mm3 (neutrophils 88.2%, lymphocytes 8.7%, monocytes 2.8%), platelet count 246000/mm3, erythrocyte sedimentation rate 63 mm/h (normal range 0-20 mm/h), alanine transferase (ALT) 176 IU/L (0-40 IU/L), aspartate aminotransferase (AST) 89 IU/L (0-40 IU/L), alkaline phosphatase (AlkP) 176 IU/L (40-129 IU/L) and gamma-glutamyl transpeptidase (GGT) 657 IU/L (10-50 IU/L); serum lactic dehydrogenase (LDH) 1896 IU/L (240-480 IU/L), thyroid-stimulating hormone (TSH) 73 μIU/ml (0.27-4.2 μIU/ml) and thyroxine (T4) 0,25 ng/dl (NR:0.93-1.71). Blood cultures and serologic tests were found to be negative for human immunodeficiency virus (HIV), hepatitis B virus (HBV), hepatitis C virus (HCV), Epstein-Barr virus (EBV), cytomegalovirus (CMV), herpes simplex virus (HSV), Rubella, Toxoplasma, parvovirus B19, Yersinia enterocolitica, Salmonella and Brucella. Serum antinuclear antibody (ANA) and rheumatoid factor were also negative. On admission, chest X-ray was clear in both lung fields and computed tomography of the neck, thorax and abdomen revealed only lymphadenopathy affecting bilateral cervical and one mediastinal lymph nodes (Figure 1). Cervical lymph node surgical biopsy was performed on the second day after admission. On macroscopic examination it appeared homogeneous, of medium consistency, dark grey colour and 2 × 1.2 × 1.1 cm in size. The patient's condition worsened over the following days, with high-grade daily fever, increasing size of cervical lymph nodes, the development of axillary lymphadenopathy and mild dyspnoea on exertion, with decreased breath sounds in both lung bases on physical examination. A second computed tomography of the thorax revealed generalized axillary, mediastinal and hilar lymphadenopathy, interstitial infiltrate in both lungs and bilateral pleural effusion (Figure 2). Thoracocentesis was performed with the following results in pleural fluid: pH 7.39 (6.8-7.6), glucose 102 mg/dl (60-100), proteins 3.2 g/dl (0-3), LDH 1694 IU/L (0-200), 50 leukocytes/ml (0-300); stains and cultures for bacteria and mycobacteria were negative and malignant cells were not found on cytologic examination. Cervical lymph node biopsy revealed necrotizing lymphadenitis with prominent areas of cortical and paracortical necrosis and distortion of the nodal architecture (Figure 3), abundant non-neutrophilic karyorrhexis and large numbers of various types of histiocytes at the margins of the necrotic areas, performing phagocytosis of cellular debris. Stimulated lymphocytes and immunoblasts were observed around these areas, along with reduced numbers of plasma cells and no neutrophils (Figure 4). Lymphoma cells were absent and stains and tissue cultures for bacteria, fungi and mycobacteria were negative. Prednisone therapy was started on the 7th day (after receiving negative viral results) at a dose of 1 mg/kg/day, with rapid improvement: the patient became afebrile on day 10, cervical and axillary swelling and tenderness began to decrease, dyspnoea disappeared and respiratory auscultation normalized. The daily dose of thyroxine was slowly increased by the endocrinologist. Prior to discharge, chest X-ray was normal. Tapering doses of prednisone were prescribed throughout the subsequent two months; all biochemical and haematological parameters normalized except for TSH, which reached normal values four months later. After two years of follow-up appointments the patient remains asymptomatic, physical examination and chest X-ray are absolutely normal and serum antinuclear antibodies remain negative.


Bilateral pleural effusion and interstitial lung disease as unusual manifestations of Kikuchi-Fujimoto disease: case report and literature review.

Garcia-Zamalloa A, Taboada-Gomez J, Bernardo-Galán P, Magdalena FM, Zaldumbide-Dueñas L, Ugarte-Maiztegui M - BMC Pulm Med (2010)

On admission both lung fields were clear and one isolated mediastinal lymph node was observed.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC2991292&req=5

Figure 1: On admission both lung fields were clear and one isolated mediastinal lymph node was observed.
Mentions: A 32-year-old Caucasian man, with a history of hyperthyroidism six months previous due to toxic multinodular goiter, treated with radioactive iodine, who developed iatrogenic hypothyroidism and on initiation of substitutive treatment, was admitted due to persistent fever (39-40°C), malaise and painful cervical lymphadenopathies that had been present for two weeks. Laboratory findings were as follows: haematocrit 34%, haemoglobin 12 g/dl, leukocyte count 3400/mm3 (neutrophils 88.2%, lymphocytes 8.7%, monocytes 2.8%), platelet count 246000/mm3, erythrocyte sedimentation rate 63 mm/h (normal range 0-20 mm/h), alanine transferase (ALT) 176 IU/L (0-40 IU/L), aspartate aminotransferase (AST) 89 IU/L (0-40 IU/L), alkaline phosphatase (AlkP) 176 IU/L (40-129 IU/L) and gamma-glutamyl transpeptidase (GGT) 657 IU/L (10-50 IU/L); serum lactic dehydrogenase (LDH) 1896 IU/L (240-480 IU/L), thyroid-stimulating hormone (TSH) 73 μIU/ml (0.27-4.2 μIU/ml) and thyroxine (T4) 0,25 ng/dl (NR:0.93-1.71). Blood cultures and serologic tests were found to be negative for human immunodeficiency virus (HIV), hepatitis B virus (HBV), hepatitis C virus (HCV), Epstein-Barr virus (EBV), cytomegalovirus (CMV), herpes simplex virus (HSV), Rubella, Toxoplasma, parvovirus B19, Yersinia enterocolitica, Salmonella and Brucella. Serum antinuclear antibody (ANA) and rheumatoid factor were also negative. On admission, chest X-ray was clear in both lung fields and computed tomography of the neck, thorax and abdomen revealed only lymphadenopathy affecting bilateral cervical and one mediastinal lymph nodes (Figure 1). Cervical lymph node surgical biopsy was performed on the second day after admission. On macroscopic examination it appeared homogeneous, of medium consistency, dark grey colour and 2 × 1.2 × 1.1 cm in size. The patient's condition worsened over the following days, with high-grade daily fever, increasing size of cervical lymph nodes, the development of axillary lymphadenopathy and mild dyspnoea on exertion, with decreased breath sounds in both lung bases on physical examination. A second computed tomography of the thorax revealed generalized axillary, mediastinal and hilar lymphadenopathy, interstitial infiltrate in both lungs and bilateral pleural effusion (Figure 2). Thoracocentesis was performed with the following results in pleural fluid: pH 7.39 (6.8-7.6), glucose 102 mg/dl (60-100), proteins 3.2 g/dl (0-3), LDH 1694 IU/L (0-200), 50 leukocytes/ml (0-300); stains and cultures for bacteria and mycobacteria were negative and malignant cells were not found on cytologic examination. Cervical lymph node biopsy revealed necrotizing lymphadenitis with prominent areas of cortical and paracortical necrosis and distortion of the nodal architecture (Figure 3), abundant non-neutrophilic karyorrhexis and large numbers of various types of histiocytes at the margins of the necrotic areas, performing phagocytosis of cellular debris. Stimulated lymphocytes and immunoblasts were observed around these areas, along with reduced numbers of plasma cells and no neutrophils (Figure 4). Lymphoma cells were absent and stains and tissue cultures for bacteria, fungi and mycobacteria were negative. Prednisone therapy was started on the 7th day (after receiving negative viral results) at a dose of 1 mg/kg/day, with rapid improvement: the patient became afebrile on day 10, cervical and axillary swelling and tenderness began to decrease, dyspnoea disappeared and respiratory auscultation normalized. The daily dose of thyroxine was slowly increased by the endocrinologist. Prior to discharge, chest X-ray was normal. Tapering doses of prednisone were prescribed throughout the subsequent two months; all biochemical and haematological parameters normalized except for TSH, which reached normal values four months later. After two years of follow-up appointments the patient remains asymptomatic, physical examination and chest X-ray are absolutely normal and serum antinuclear antibodies remain negative.

Bottom Line: All of them resolved with prednisone and after two years of following up he remains asymptomatic and without evidence of any other associated disease.Pleural effusion and interstitial lung disease are very uncommon manifestations of KFD.In our experience, treatment with oral prednisone was effective.

View Article: PubMed Central - HTML - PubMed

Affiliation: Mendaro Hospital, Gipuzkoa, Spain. ALBERTO.GARCIA-ZAMALLOAZAMALLOA@OSAKIDETZA.NET

ABSTRACT

Background: Kikuchi-Fujimoto's disease (KFD), also called histiocytic necrotizing lymphadenitis, is a rare, idiopathic and self-limited condition usually characterized by cervical lymphadenopathy and fever, most often affecting young patients. Aetiology is unknown. Differential diagnosis includes mainly malignant lymphoma, tuberculous lymphadenitis and systemic lupus erythematosus (SLE), so early diagnosis is crucial. Pleuropulmonary involvement due to isolated KFD has been seldom reported.

Case presentation: a 32-year-old man, on treatment for iatrogenic hypothyroidism, was admitted due to high grade fever and painful cervical lymphadenopathies. KFD was diagnosed by lymph node biopsy. Some days after admission the patient got worse, he developed generalized lymphadenopathy, bilateral pleural effusion and interstitial lung disease. All of them resolved with prednisone and after two years of following up he remains asymptomatic and without evidence of any other associated disease.

Conclusion: Pleural effusion and interstitial lung disease are very uncommon manifestations of KFD. In our experience, treatment with oral prednisone was effective.

Show MeSH
Related in: MedlinePlus