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We report a case of global testicular infarction associated with epididymitis. A 26-year-old man with a history of clean intermittent self-catheterization since he was 1 year old presented to our hospital with left scrotal pain and swelling. He was diagnosed with epididymitis and was prescribed levofloxacin. On the next day, he returned with worsened symptoms of left scrotal pain, swelling, and fever. He was admitted because of his severe symptoms and high C-reactive protein level in the blood test. Antimicrobial therapy with intravenous flomoxef and analgesic treatment with pentazocine and loxoprofen were started but the symptoms did not improve. The color-Doppler ultrasound repeated on the 1st, 4th, and 5th day of admission showed left epididymal hypervascularity but it did not indicate testicular hypovascularity in any examinations. On the 6th day of admission, a contrast-magnetic resonance imaging (MRI) scan revealed no contrast enhancement in the left testis and high orchiectomy was performed. On pathological examination, abscess of the entire epididymis and generalized necrosis of the testes were observed. Inflammatory cell infiltration and thrombus formation were observed in almost all veins of the testis and spermatic cord, and the diagnosis of global testicular infarction associated with epididymitis was made. Global testicular infarction has been reported as a rare complication of epididymitis and should be considered in the case of atypical course of epididymitis.
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Epididimitis , Adulto , Epidídimo , Epididimitis/tratamiento farmacológico , Humanos , Lactante , Infarto/diagnóstico por imagen , Infarto/etiología , Masculino , Orquiectomía , Testículo/diagnóstico por imagenRESUMEN
A 64-year-old woman was diagnosed with diffuse large B-cell lymphoma (DLBCL) in 2013. After eight courses of R-CHOP therapy followed by local irradiation of the remaining retroperitoneal soft tissue shadow, complete response was confirmed on 18F-2-fluoro-2-deoxyglucose-positron emission tomography/computed tomography (FDG-PET/CT). Early in 2016, patient's serum LDH and soluble IL-2 receptor levels elevated. With suspected recurrence of DLBCL, FDG-PET/CT was performed that showed no lymphadenopathy or abnormal FDG uptake. By the end of July 2016, the patient developed fever and night sweating. Intravascular large B-cell lymphoma (IVLBCL) was suspected, and the patient underwent random skin biopsies, which revealed large atypical cells infiltrating peripheral and intravascular regions of the subcutaneous adipose tissue. Cell morphology, immunostaining, and PCR analysis of the immunoglobulin heavy chain gene suggested the recurrence of DLBCL. Despite salvage chemotherapy and autologous peripheral stem cell transplantation with high-dose chemotherapy, approximately 15 months later, DLBCL recurred and involved the lungs. The patient again received chemotherapy and achieved a second remission. Because DLBCL may recur like intravascular lymphoma, the same tests used for IVLBCL diagnosis are required in cases of suspected recurrence of DLBCL based on clinical and laboratory findings.
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Linfoma de Células B Grandes Difuso/diagnóstico , Neoplasias Vasculares/diagnóstico , Anciano , Anticuerpos Monoclonales de Origen Murino , Protocolos de Quimioterapia Combinada Antineoplásica , Ciclofosfamida , Doxorrubicina , Femenino , Fluorodesoxiglucosa F18 , Humanos , Linfoma de Células B Grandes Difuso/terapia , Tomografía Computarizada por Tomografía de Emisión de Positrones , Tomografía de Emisión de Positrones , Prednisona , Recurrencia , Inducción de Remisión , Rituximab , Terapia Recuperativa , Trasplante de Células Madre , Neoplasias Vasculares/terapia , VincristinaRESUMEN
Metastatic glioblastoma is a rare condition, and several studies have reported the involvement of multiple organs including the lymph nodes, liver, and lung. The lung and pleura are reportedly the most frequent sites of metastasis, and diagnosis using less invasive tools such as cytological analysis with fine needle aspiration biopsy is challenging. Cytological analysis of fluid specimens tends to be negative because of the small number of cells obtained, whereas the cell block technique reportedly has higher sensitivity because of a decrease in cellular dispersion. Herein, the authors describe a patient with a history of diffuse astrocytoma who developed intractable, progressive accumulation of pleural fluid. Initial cytological analysis of the pleural effusion obtained by thoracocentesis was negative, but reanalysis using the cell block technique revealed the presence of glioblastoma cells. This is the first report to suggest the effectiveness of the cell block technique in the diagnosis of extracranial glioblastoma using pleural effusion. In patients with a history of glioma, the presence of extremely intractable pleural effusion warrants cytological analysis of the fluid using this technique in order to initiate appropriate chemotherapy.
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Neoplasias Encefálicas/diagnóstico por imagen , Glioblastoma/diagnóstico por imagen , Neoplasias Pulmonares/diagnóstico por imagen , Neoplasias Pulmonares/secundario , Derrame Pleural/diagnóstico por imagen , Anciano , Neoplasias Encefálicas/cirugía , Resultado Fatal , Femenino , Glioblastoma/cirugía , Humanos , Neoplasias Pulmonares/cirugía , Derrame Pleural/cirugíaRESUMEN
Choroid plexus hyperplasia/papilloma and resulting hyperproduction of cerebrospinal fluid is a rare cause of hydrocephalus. In these patients, intractable ascites can occur after a ventriculoperitoneal (VP) shunting operation. However, shunt-related hydrocele is a rare complication of VP shunting. Previous reports have indicated catheter-tip migration to the scrotum as a cause of hydrocele. Here, we present the first documented case of choroid plexus hyperplasia that led to intractable ascites after shunting and a resulting hydrocele without catheter-tip migration into the scrotum.
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Ascitis , Plexo Coroideo/patología , Plexo Coroideo/cirugía , Hidrocefalia/cirugía , Hiperplasia/complicaciones , Derivación Ventriculoperitoneal/efectos adversos , Ascitis/etiología , Humanos , Hidrocefalia/diagnóstico por imagen , Hidrocefalia/etiología , Lactante , Imagen por Resonancia Magnética , Masculino , Procedimientos Neuroquirúrgicos/efectos adversos , Tomografía Computarizada por Rayos XRESUMEN
A 75-year-old woman with stage IVB (cT2bN3M1b) lung adenocarcinoma was administered nivolumab, ipilimumab, carboplatin, and paclitaxel. Fourteen days after receiving chemotherapy, she experienced an impaired consciousness and a cerebrospinal fluid analysis revealed high protein levels and pleocytosis. She was diagnosed with nivolumab- and ipilimumab-induced encephalitis and was treated with corticosteroids which were tapered to 10 mg/day, with no symptom recurrence. She died 18 weeks after the initial presentation, as the cancer worsened. An autopsy showed encephalitis and CD8+ lymphocyte infiltration around the blood vessels. Thus, immune-related adverse events should be suspected and treatment should be initiated for patients presenting with an impaired consciousness when concurrently being treated with nivolumab and ipilimumab.
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OBJECTIVE: In this study, we examine how advancements in novel antirheumatic drugs affect the clinicopathologic features of lymphoproliferative disorder (LPD) in patients with rheumatoid arthritis (RA). METHODS: In this multicenter study across 53 hospitals in Japan, we characterized patients with RA who developed LPDs and visited the hospitals between January 1999 and March 2021. The statistical tools used included Fisher's exact test, the Mann-Whitney U-test, the log-rank test, logistic regression analysis, and Cox proportional hazards models. RESULTS: Overall, 752 patients with RA-associated LPD (RA-LPD) and 770 with sporadic LPD were included in the study. We observed significant differences in the clinicopathologic features between patients with RA-LPD and those with sporadic LPD. Histopathological analysis revealed a high frequency of LPD-associated immunosuppressive conditions. Furthermore, patients with RA-LPD were evaluated based on the antirheumatic drugs administered. The methotrexate (MTX) plus tacrolimus and MTX plus tumor necrosis factor inhibitor (TNFi) groups had different affected site frequencies and histologic subtypes than the MTX-only group. Moreover, MTX and TNFi may synergistically affect susceptibility to Epstein-Barr virus infection. In case of antirheumatic drugs administered after LPD onset, tocilizumab (TCZ)-only therapy was associated with lower frequency of regrowth after spontaneous regression than other regimens. CONCLUSION: Antirheumatic drugs administered before LPD onset may influence the clinicopathologic features of RA-LPD, with patterns changing over time. Furthermore, TCZ-only regimens are recommended after LPD onset.
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Antirreumáticos , Artritis Reumatoide , Trastornos Linfoproliferativos , Metotrexato , Inhibidores del Factor de Necrosis Tumoral , Humanos , Artritis Reumatoide/tratamiento farmacológico , Artritis Reumatoide/complicaciones , Antirreumáticos/uso terapéutico , Antirreumáticos/efectos adversos , Trastornos Linfoproliferativos/inducido químicamente , Masculino , Femenino , Persona de Mediana Edad , Metotrexato/uso terapéutico , Anciano , Inhibidores del Factor de Necrosis Tumoral/uso terapéutico , Inhibidores del Factor de Necrosis Tumoral/efectos adversos , Japón , Tacrolimus/uso terapéutico , Tacrolimus/efectos adversos , Quimioterapia Combinada , Infecciones por Virus de Epstein-Barr/complicaciones , AdultoRESUMEN
Birt-Hogg-Dubé (BHD) syndrome is an autosomal dominant disorder caused by germline mutations in the folliculin gene (FLCN). It is characterized by skin tumors, multiple lung cysts, and renal tumors. Active genetic testing and appropriate periodic examinations of family lines of patients with BHD syndrome have not been widely performed. In this report, we present our experience regarding the diagnosis of asymptomatic family members with BHD syndrome. The proband was a 65-year-old female with a family history of colorectal cancer and spontaneous pneumothorax that affected her father. Computed tomography revealed an approximately 10 cm-sized tumor protruding from the upper pole of the left kidney, a buried tumor approximately 1.5 cm in length in the right kidney, and multiple pulmonary cysts. The patient underwent laparoscopic radical left nephrectomy. Pathological examination indicated that the resected tumor was a chromophobe renal cell carcinoma. After the surgery, there was no evidence of local recurrence or metastasis. The size of the tumor in the right kidney was monitored, but it did not increase. On FLCN genetic examination, targeted next generation sequencing revealed a partial deletion of exon 14, thus confirming the diagnosis of the patient to be BHD syndrome that caused the previously unreported pathogenic variant. Three years after the surgery, we conducted genetic counseling for the proposita and her three children. Genetic examination, performed at the request of the second daughter, confirmed that she carried the same genetic variant as her mother. This diagnosis prompted the second daughter to begin managing her health via periodic imaging tests.
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Síndrome de Birt-Hogg-Dubé , Neoplasias Renales , Proteínas Proto-Oncogénicas , Proteínas Supresoras de Tumor , Anciano , Femenino , Humanos , Enfermedades Asintomáticas , Síndrome de Birt-Hogg-Dubé/genética , Síndrome de Birt-Hogg-Dubé/diagnóstico , Carcinoma de Células Renales/genética , Carcinoma de Células Renales/diagnóstico , Carcinoma de Células Renales/patología , Carcinoma de Células Renales/diagnóstico por imagen , Carcinoma de Células Renales/cirugía , Pruebas Genéticas , Mutación de Línea Germinal , Heterocigoto , Neoplasias Renales/genética , Neoplasias Renales/diagnóstico , Neoplasias Renales/patología , Neoplasias Renales/cirugía , Nefrectomía , Linaje , Proteínas Proto-Oncogénicas/genética , Proteínas Supresoras de Tumor/genéticaRESUMEN
BACKGROUND: Pulmonary veno-occlusive disease (PVOD) and pulmonary capillary hemangiomatosis (PCH) are rare causes of pulmonary hypertension. There is no proven medical therapy to treat these diseases, and lung transplantation is thought to be the only cure. Administration of vasodilators including epoprostenol sometimes causes massive pulmonary edema and could be fatal in these patients. METHODS AND RESULTS: Eight patients were treated with epoprostenol for 387.3±116.3 days (range, 102-1,063 days), who were finally diagnosed with PVOD or PCH by pathological examination. The maximum dose of epoprostenol given was 55.3±10.7 ng·kg(-1)·min(-1) (range, 21.0-110.5 ng·kg(-1)·min(-1)). With careful management, epoprostenol therapy significantly improved the 6-min walk distance (97.5±39.2 to 329.4±34.6 m, P<0.001) and plasma brain natriuretic peptide levels (381.3±136.8 to 55.2±14.4 pg/ml, P<0.05). The cardiac index significantly increased from 2.1±0.1 to 2.9±0.3 L·min(-1)·m(-2) (P<0.05). However, pulmonary artery pressure and pulmonary vascular resistance were not significantly reduced. For 4 patients, epoprostenol therapy acted as a bridge to lung transplantation. For the other patients who had no chance to undergo lung transplantation, epoprostenol therapy was applied for 528.0±216.6 days and the maximum dose was 63.9±19.0 ng·kg(-1)·min(-1). CONCLUSIONS: This study data suggest that cautious application of epoprostenol can be considered as a therapeutic option in patients with PVOD and PCH.
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Antihipertensivos/uso terapéutico , Epoprostenol/uso terapéutico , Hemangioma Capilar/tratamiento farmacológico , Hipertensión Pulmonar/tratamiento farmacológico , Neoplasias Pulmonares/tratamiento farmacológico , Enfermedad Veno-Oclusiva Pulmonar/tratamiento farmacológico , Vasodilatadores/uso terapéutico , Adolescente , Adulto , Antihipertensivos/efectos adversos , Biomarcadores/sangre , Presión Sanguínea/efectos de los fármacos , Niño , Epoprostenol/efectos adversos , Tolerancia al Ejercicio/efectos de los fármacos , Femenino , Hemangioma Capilar/sangre , Hemangioma Capilar/complicaciones , Hemangioma Capilar/diagnóstico , Hemangioma Capilar/fisiopatología , Humanos , Hipertensión Pulmonar/sangre , Hipertensión Pulmonar/diagnóstico , Hipertensión Pulmonar/etiología , Hipertensión Pulmonar/fisiopatología , Japón , Neoplasias Pulmonares/sangre , Neoplasias Pulmonares/complicaciones , Neoplasias Pulmonares/diagnóstico , Neoplasias Pulmonares/fisiopatología , Trasplante de Pulmón , Masculino , Péptido Natriurético Encefálico/sangre , Enfermedad Veno-Oclusiva Pulmonar/sangre , Enfermedad Veno-Oclusiva Pulmonar/complicaciones , Enfermedad Veno-Oclusiva Pulmonar/fisiopatología , Recuperación de la Función , Factores de Tiempo , Resultado del Tratamiento , Resistencia Vascular/efectos de los fármacos , Vasodilatadores/efectos adversosRESUMEN
Pancreatic cancer is not easy to detect at its early stages due to difficulties in identifying symptoms at these stages. As it progresses, abdominal pain, loss of appetite, abdominal distension, jaundice and pain in the back, especially the lower back, might develop. Moreover, sudden onset or worsening of diabetes mellitus may be seen, which often prompts screening for the detection of pancreatic cancer. Since it rapidly spreads to surrounding tissues and organs, pancreatic cancer has a poor prognosis. However, metastasis to the bladder is rare, with few cases diagnosed on the basis of detecting gross hematuria. The current study presents a case of gross hematuria and exacerbated diabetes in a 90-year-old woman. Cystoscopy revealed a non-papillary tumor in the posterior bladder wall. Pathological examination of bladder tumor specimens obtained via transurethral resection revealed adenocarcinoma. Subsequent systemic examinations revealed primary pancreatic cancer that had metastasized to the bladder. To the best of our knowledge, this is the second reported case of pancreatic cancer diagnosed based on the detection of gross hematuria due to bladder metastasis, since 1992.
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Leptomeningeal metastasis (LM) is a rare but devastating cancer complication. LM occurs when cancer spreads into the leptomeningeal layer or cerebrospinal fluid. Intracranial magnetic resonance (MR) images of LM are characterized by the diffuse enhancement of the leptomeninges along the cerebral sulci, cerebellar folia, and cranial nerves. Here, we report an extremely rare case of LM with an atypical MR image revealing tumor mass confinement to the arachnoid membrane. The case involves an 85-year-old man who was referred to our hospital with a three-day history of dysarthria. Radiological examination revealed a solid lesion with heterogeneous enhancement and a cystic component in the extra-axial region of the right parietal lobe. Upon subsequent general examination, multiple lung cancer metastases were suspected. The patient underwent gross total resection of the brain mass in the right parietal region. Although the tumor slightly adhered to the dura mater, it was sharply demarcated from the surrounding parenchyma and pia mater. Based on pathological examination, the tumor was diagnosed as small cell lung cancer metastasis. This metastatic brain tumor was exclusively confined to the arachnoid membrane and, except for a few blood vessels, the dura mater was not infiltrated by metastatic tumor cells. To our knowledge, this is the first reported case of LM in which the tumor mass is confined only to the arachnoid membrane. Thus, in cases with atypical MR images, a general examination considering the possibility of LM is important for prompt and accurate diagnosis.
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Introduction: Extramammary myofibroblastomas are extremely rare. Case presentation: The patient was an 88-year-old male. He presented for evaluation of frequent urination and a "pushing up" sensation from the groin during defecation. Thorough physical and radiographic examinations revealed a retroperitoneal tumor on the right side of the rectum. The pathologic examination of the biopsy tissue showed that the tumor was unlikely to be malignant. Nevertheless, the patient was symptomatic and thus underwent a laparoscopic tumor resection through a transperitoneal approach. The tumor was circumscribed with a solid capsule. Based on the pathologic findings, which included immunostaining, the tumor was diagnosed as a myofibroblastoma. There was no evidence of a recurrence 6 months postoperatively. Conclusion: We present this case with the clinical course and surgical findings, and discuss the possibility of establishing a preoperative pathologic diagnosis of a myofibroblastoma.
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A 74-year-old man was admitted to our hospital because of systemic lymphadenopathy, weight loss, and a fever at night that had persisted for approximately 1 month. Blood tests revealed extreme peripheral blood plasmacytosis and hypergammaglobulinemia. A lymph node biopsy showed angioimmunoblastic T-cell lymphoma (AITL). Based on the history of methotrexate (MTX) administration, the established diagnosis was MTX-associated lymphoproliferative disorder (MTX-LPD). After MTX was discontinued, the lymphadenopathy spontaneously regressed and the plasmacytosis disappeared. He had no disease progression for three years. We found that AITL as an MTX-LPD can cause plasmacytosis, and the prognosis of this disease may not be poor.
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Linfadenopatía Inmunoblástica , Linfadenopatía , Linfoma de Células T , Trastornos Linfoproliferativos , Anciano , Humanos , Linfadenopatía Inmunoblástica/inducido químicamente , Linfadenopatía Inmunoblástica/diagnóstico , Linfadenopatía/inducido químicamente , Linfadenopatía/complicaciones , Linfoma de Células T/inducido químicamente , Linfoma de Células T/tratamiento farmacológico , Trastornos Linfoproliferativos/inducido químicamente , Trastornos Linfoproliferativos/complicaciones , Trastornos Linfoproliferativos/diagnóstico , Masculino , Metotrexato/efectos adversosRESUMEN
A 69-year-old man with refractory lung adenocarcinoma was treated with gemcitabine and vinorelbine. Dyspnea and hypertension developed after the 17th cycle of chemotherapy. Laboratory findings revealed intravascular hemolysis and renal dysfunction. Thrombotic microangiopathy (TMA) was confirmed by renal biopsy. Antihypertensive and steroid therapies were ineffective. After plasmapheresis, intravascular hemolysis and renal dysfunction gradually improved. However, the disease progressed, and he died 6 months after TMA diagnosis. Autopsy revealed similar pathological findings to those of the renal biopsy. It is important to discontinue gemcitabine at the onset of TMA and consider TMA when using gemcitabine for long periods.
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A 63-year-old man with pulmonary adenocarcinoma was treated with nivolumab. High fever developed within several hours after the first administration of nivolumab; subsequently, serum creatinine levels kept increasing daily. We diagnosed acute kidney injury (AKI) as an immune-related adverse event; the patient was initially treated with 50 mg prednisolone, and the dose was then tapered. Renal biopsy pathologically revealed tubulointerstitial inflammation with strong infiltration of only T cells that were CD3+, CD4+, and CD8+. The infiltration of CD163+ M2 macrophage was also observed. AKI within 1 week after the administration of nivolumab seems to be rare; therefore, the present case provides important findings useful in daily clinical practice.
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Neoplasias de los Genitales Masculinos , Neoplasias Hepáticas , Enfermedad de Paget Extramamaria , Neoplasias del Colon Sigmoide , Humanos , Masculino , Antígeno Carcinoembrionario , Neoplasias de los Genitales Masculinos/patología , Genitales/patología , Queratina-19 , Neoplasias Hepáticas/diagnóstico , Enfermedad de Paget Extramamaria/diagnóstico , Enfermedad de Paget Extramamaria/patología , AncianoRESUMEN
BACKGROUND: Posttransplant lymphoproliferative disorder (PTLD) is a rare condition occurring after organ transplantation. PTLD comprises 4 subtypes, of which Hodgkin lymphoma (HL) type and HL-like type (currently included in polymorphic type) account for only about 1%-3% of cases. Primary central nervous system PTLD is also rare; most cases are Epstein-Barr virus-positive, B-cell PTLD. To our knowledge, no case of HL-like PTLD has been documented. CASE DESCRIPTION: A 43-year-old woman who underwent kidney transplantation for IgA nephropathy 14 years previously presented to the emergency department with seizure. Gadolinium-enhanced T1-weighted magnetic resonance imaging showed a ring-enhancing mass in the left temporal lobe. Gross total removal of the tumor was performed, and pathologic examination revealed findings consistent with HL-like PTLD. The patient's immunosuppressants were subsequently reduced, and she received postoperative systemic therapy with rituximab and radiation therapy. Follow-up magnetic resonance imaging showed no signs of relapse. CONCLUSIONS: This represents an extremely rare case of a patient with HL-like PTLD occurring as a primary central nervous system lesion.
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Neoplasias del Sistema Nervioso Central/etiología , Enfermedad de Hodgkin/etiología , Trasplante de Riñón/efectos adversos , Trastornos Linfoproliferativos/etiología , Complicaciones Posoperatorias/etiología , Adulto , Neoplasias del Sistema Nervioso Central/diagnóstico por imagen , Neoplasias del Sistema Nervioso Central/terapia , Femenino , Enfermedad de Hodgkin/diagnóstico por imagen , Enfermedad de Hodgkin/terapia , Humanos , Trastornos Linfoproliferativos/diagnóstico por imagen , Trastornos Linfoproliferativos/terapia , Complicaciones Posoperatorias/diagnóstico por imagen , Complicaciones Posoperatorias/terapiaRESUMEN
This is the first report about a patient with concomitant polymyositis (PM), myasthenia gravis (MG), and aplastic anemia (AA). A 54-year-old male developed myalgia and muscle weakness, which gradually progressed over 2 months. He was persistently affected by MG and AA. Brachium magnetic resonance imaging showed increased signal intensity in the left triceps and deltoid muscles on short tau inversion recovery images. A muscle biopsy examination revealed perifascicular atrophication and inflammatory myopathy. We diagnosed the patient with PM combined with MG and AA. He was successfully treated with an autologous bone marrow transplantation (BMT). The present case suggests that BMT is a therapeutic option for PM, MG, and AA.
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A 66-year-old, hepatitis C virus (HCV)-positive woman was admitted to our hospital with oliguria, systemic edema, and rapid deterioration of renal function. Laboratory examination showed increased serum creatinine and decreased serum albumin levels, complement activity, and cryoglobulin positivity. The HCV RNA genotype was found to be 1b, and the viral load was high. Kidney biopsy examination showed type I membranoproliferative glomerulonephritis with capillary deposition of IgM and C3, indicating HCV-related glomerulonephritis. After hospitalization, hemodialysis was immediately required because of uremia and oliguria. Her renal function did not improve despite corticosteroid therapy. To treat the increasing HCV load, virus removal and eradication by double-filtration plasmapheresis therapy without interferon was performed, since the patient was allergic to interferon therapy. This treatment improved renal function and allowed the withdrawal from hemodialysis. This report presents a case of successful VRAD without interferon therapy in a patient with HCV-related glomerulonephritis and acute kidney injury that required hemodialysis.
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Neonatal Bartter syndrome (NBS) is an inherited renal tubular disorder associated with hypokalemic alkalosis. Here we report a case of genetically diagnosed NBS. Polyhydramnios was noted at 26 weeks. A boy was born at 31 weeks and 1 day, weighed 1344 g, and had an Apgar score of 8/8. We initiated indomethacin (IND) at a dose of 0.2 mg/kg/d on day 31, and increased it to approximately 3 mg/kg/d. However, his urinary calcium (Ca) levels remained unchanged. At 4 months of age, nephrocalcinosis was detected by ultrasound. The placenta weighed 700 g (+2.7 standard deviations). Although the proportion of terminal villi was consistent with the gestational age, many of them exhibited poorly dilated capillaries. Hemosiderin pigment was seen throughout the amniochorionic connective tissue and along about 50% of the trophoblast basement membrane (TBM). Von Kossa stain revealed the corresponding area of mineralization along the TBM. In our opinion, urinary Ca levels were high and did not change after IND initiation, indicating that nephrocalcinosis may be inevitable. Enhanced inflow of maternal plasma through the basement membrane would cause Ca deposition, given that the same finding was obtained in the case with polyhydramnios. The same mechanism would also explain the hemosiderin pigment distribution.