Cryptogenic organizing pneumonia associated with pregnancy:A case report

lNTRODUCTlON

Cryptogenic organizing pneumonia(COP)is a diffuse infiltrating lung disease,wherein granulation tissue proliferates in the small bronchiolar epithelium damaged owing to various causes and consequently obstructs alveolar ducts and alveoli[1,2].The occurrence of COP in pregnancy is extremely rare and pregnancy-related physiological changes may worsen respiratory complications in COP.Previously reported cases had pre-onset underlying diseases such as asthma,fungal infection and Crohn's disease that can cause inflammatory condition(Table 1).Here,we report the fourth case of COP in a pregnant woman without underlying medical history initially diagnosed with community-acquired pneumonia that did not improve with antibiotic treatment.

CASE PRESENTATlON

Chief complaints

A 35-year-old woman,gravida 2,para 1,presented with concerns of chest wall pain with cough,sputum,dyspnea,and mild fever of 37.7 °C at 11 wk of gestation.

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History of present illness

The mild cough was started ten days ago with gradually aggravated feature.

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History of past illness

Her obstetric history included spontaneous vaginal delivery at 40 wk of gestation,with no special medical history.In particular,there was no history of previous pulmonary diseases.

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

Personal and family history

A 7.5 pack-year history of smoking was noted before the first pregnancy by an antenatal evaluation.

Physical examination

On admission,the patient’s blood pressure was 120/80 mmHg,body temperature was 37.7 °C,pulse rate was 108/min,oxygen saturation was 96%,and respiratory rate was 28/min with a rale in the right lower lung area.

Laboratory examinations

Laboratory tests indicated a white blood cell count of 7.59 × 10/μL(74.9% neutrophils,10.8%lymphocytes,and 3.5% monocytes)with an absolute neutrophil count of 5680 cells/μL,a hemoglobin level of 12.1 g/dL,a platelet count of 284 × 10/μL,and an elevated C-reactive protein level of 4.77 mg/dL.

Imaging examinations

Chest radiography showed increased patchy opacities in the right lower lobe(Figure 1),and computed tomography(CT)revealed some patchy lobular consolidation and peripheral ground-glass opacities(GGOs)in the posterior and lateral basal segments of the right lower lobe(Figure 2).The pulmonary function test showed a forced vital capacity(FVC)of 2.87 L(77% of predicted),forced expiratory volume in one second(FEV1)of 2.35 L(74% of predicted),FEV/FVC ratio of 82%,and peak expiratory flow of 6.19 L/s.The tidal flow-volume curve revealed minimal obstructive lung disease.An ultrasound examination showed that appropriate fetal growth for the gestational age,a normal amount of amnioticfluid,and no specific abnormal findings.

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FlNAL DlAGNOSlS

The increased lymphocyte count(40%)and a decrease in the CD4/CD8 ratio(0.6)with the presence of macrophages(25%)and neutrophils(8%)in BAL suggested a diagnosis of COP.

TREATMENT

This research did not receive any specific grant from funding agencies in the public,commercial,or notfor-profit sectors.

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OUTCOME AND FOLLOW-UP

Post-discharge,the patient did not express any special events during pregnancy and gave birth by vaginal delivery at 39+4 d of gestation(male,3370 g;Apgar scores of 8 and 9 at 1 and 5 min,respectively).Transbronchial lung biopsy was conducted after delivery without any complications,and the proliferation of granulation tissue into the bronchioles and alveolar duct indicated COP(Figure 3).

DlSCUSSlON

The authors have no conflict of interest.

The authors have read the CARE Checklist(2016),and the manuscript was prepared and revised according to the CARE Checklist(2016).

In general,imaging approaches are employed to diagnose COP.Chest radiography for COP has three characteristic features:multiple alveolar opacities(typical COP),solitary opacity(focal COP),and infiltrative opacities(infiltrative COP).Bilateral multiple opacities are more common than solitary patterns[6,7].In our patient,bilateral patchy opacities were observed in both lower lobes.Thin-section CT scans have a correct diagnosis rate of 79% with histologically proved COP[8].CT findings for COP are patchy GGOs in the subpleural and/or peribronchovascular area(80%),airspace consolidation in bilateral lower lobes(71%),wall thickening and cylindrical dilatation of air bronchogram(71%),ill-defined small nodular opacities(50%),and pleural effusion(in a third of patients)[9].The specific multifocal patchy airspace consolidation,GGOs,and bilateral pleural effusion were observed.

Corticosteroids are administered as the initial treatment for COP and are effective for both typical and focal COP.The recommended treatment regimens include initial dosages of 0.75-1.5 mg/kg prednisolone for 3 mo with gradual reduction according to clinical symptom improvement[10].In this case,we began with a low initial oral dose of prednisolone of 0.5 mg/kg/d after the patient’s second admission because corticosteroid use in first trimester can be associated with the development of an orofacial cleft.Fortunately,symptoms improved after 5 d of the low-dose administration and maintenance therapy was continued for 5 more days.This rare case is about the COP diagnosed in pregnant women without underlying medical conditions.In addition,it suggests a diagnostic value of COP,which is less effective in conevntional initial treatment.In this case,a pregnant woman was initially diagnosed with community-acquired pneumonia and treated with antibiotics;her symptoms seemed to improve temporarily but then recurred with greater severity.

CONCLUSlON

COP has similar clinical features with other types of pneumonia and in particular,chest radiographic differentiation of COP could be difficult.The progressive condition indicates a specific clinical aspect of COP;thus,it is important to differentiate COP from other atypical pneumonia that recur despite initial antibiotic treatment.

ACKNOWLEDGEMENTS

We began steroid treatment with prednisolone(0.5 mg/kg/d),and progressive improvement of radiological findings was noted.Dyspnea improved after 3 d of steroid treatment,and other symptoms were reduced on the 5day of steroid administration.

FOOTNOTES

Kim YS designed and projected the case report;Lee YJ organized data of cases and collected and analyzed previous reports;Lee YJ and Kim YS wrote and revised the manuscript;all authors contributed to editorial changes in the manuscript;all authors read and approved the final manuscript.

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The prevalence of COP is unknown and is mainly observed in individuals aged 50-60 years[3].COP occurrence during pregnancy is extremely rare,but it could be more severe owing to physiologic changes in pregnant women,such as an elevated diaphragm,increased oxygen demand,decreased functional residual capacity,and decreased chest wall compliance[4].Thus,previous reports have recommended close antenatal care and regular pulmonary function tests to reduce respiratory complications during pregnancy;further,elective preterm delivery can be an option in more severe cases[5].

The clinical features of COP in the case described above were not notably different from the general clinical features of COP.The respiratory symptoms began with a flu-like illness with cough,mild fever,malaise and progression of shortened breathing to dyspnea[2].Although a quarter of patients with COP had no special physical findings[3],inhalation rales or crackling were observed in the physical examination in this case.

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Young Joo Lee 0000-0001-5294-7368;Young Sun Kim 0000-0002-1725-0968.

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